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A 7-year-old boy was referred to Pediatric Department of Mashhad Dental School because of severe mobility in the second primary left mandibular molar. On evaluation of his medical history, the parents reported no systemic disorder. Laboratory findings, which included a complete blood cell count and electrolyte, calcium, phosphorus, and alkaline phosphatase values, were normal. There was no history of dental treatment or trauma. His oral hygiene was excellent, and there was no abnormal finding on extraoral and intraoral examination except for Class II malocclusion []. The patient was caries-free and had no parafunctional habits such as bruxism and no wear facets, or premature contacts were detected; the only chief complaint was increased mobility of the lower left second molar. There was no family history of early exfoliation of primary teeth, abnormal root resorption or spontaneous loss of permanent teeth. There was no history of hypersensitivity of the patient's teeth to thermal stimuli, spontaneous pain, or pain with mastication.\nAn orthopantomogram (OPG) X-ray was taken for thorough evaluation of the patient's dentition, and posteroanterior (PA) views were obtained for a more detailed examination of the affected tooth. On radiographic examination, there was no sign of caries or any other abnormal finding except root resorption in both of the second mandibular molars. OPG and PA views showed extensive root resorption of the left second mandibular molar and mild external root resorption of the right second mandibular molar [].\nOn clinical examination, the color and texture of the gingival tissue around the involved teeth were normal. Except for the right mandibular primary second molar that had severe mobility, the mobility of the other teeth was within normal range. The pulp test revealed pulp vitality of the right mandibular primary second molar, but we could not test the left affected tooth because as we were taking the familial history, the patient wiggled and pulled the tooth out with his hand. On macroscopic examination of the exfoliated second molar, the roots were thoroughly resorbed, and the caries-free crown was undermined [].\nHistological evaluation of the exfoliated tooth was impossible due to complete resorption of roots, but histological evaluation of the soft tissue removed from the socket of the exfoliated tooth showed nonspecific chronic inflammation []. Langerhans cells were found on microscopic examination, so immunohistochemical staining with CD1A was performed. However, a negative result for this test ruled out Langerhans cell disease.\nOn the basis of the history, oral examination, and radiographic evaluation, and because there was no specific cause for this condition, a diagnosis of localized IRR was made, and after placement of a band and loop space maintainer, the patient was followed for 18 months [].\nIn the follow-up period over 18 months, the right involved tooth was clinically asymptomatic; hence, the patient's father did not agree to take new PA radiographs to determine if there was any progression of the root resorption. |
A 64-year-old Japanese woman was admitted to our hospital seven years ago with a complaint of pain in her right hip joint. Radiographic analysis revealed an osteolytic tumor of her right pelvis and a tumor in her right lower lung field. The histological findings of a biopsy specimen obtained from the bone and pulmonary tumors showed adenocarcinoma. Immunohistochemical tests showed that the tumor cells stained positive for thyroid transcription factor-1. Therefore, we diagnosed our patient with advanced lung cancer (cT2N2M1). She received systemic chemotherapy with carboplatin and paclitaxel, starting one month after diagnosis after palliative irradiation of the pelvic lesion. After completion of four consecutive courses of chemotherapy, a partial response was achieved. However, local recurrence occurred six months later. Because docetaxel, gemcitabine and vinorelbine were all insufficient for inhibiting disease progression, gefitinib was administered as the fourth regimen, starting one year after diagnosis. A tumor response was subsequently observed and the treatment was continued. However, a routine brain magnetic resonance imaging scan showed a de novo metastatic lesion in her left frontal lobe two years after diagnosis. In accordance with our patient's wishes, gefitinib administration was continued after surgical resection of the brain tumor. Although the primary lesion did not exhibit regrowth, additional brain and pulmonary metastases in her right lung were observed four years after diagnosis. Erlotinib was administered as the fifth regimen following stereotactic radiosurgery for the brain tumor. Significant growth of the pulmonary metastatic lesion was observed one year later, although the other lesions did not demonstrate regrowth. We repeated a bronchoscopy for the pulmonary metastatic lesion to investigate the EGFR gene mutation status.\nWe used a combination of the peptide nucleic acid-locked nucleic acid polymerase chain reaction (PNA-LNA PCR) clamp method and the direct sequencing method for determining the EGFR gene mutation status []. The result of the PNA-LNA PCR clamp assay for the EGFR gene showed a double-activating mutation consisting of an in-frame deletion mutation in exon 19 and an L858R point mutation in exon 21. The mutation identified in exon 19 was consistent with I744-R748del and two subsequent substitution mutations, E749I (GAA to ATT) and A750K (GCA to AAA). To shed light on the sequential changes in the EGFR mutation status, we also analyzed a series of paraffin-embedded samples obtained from this patient's tumors. The histological findings of the analyzed samples clearly demonstrated the presence of adenocarcinoma cells (Figure ). The results are summarized in Table . Genetic analysis of the specimen from the primary pulmonary tumor at diagnosis showed a wild-type EGFR gene. The specimen from the metastatic bone tumor had an exon 19 deletion identical to that in the metastatic pulmonary tumor. L858R was not observed in the bone tumor. The resected brain tumor harbored T790M in addition to the exon 19 deletion. In contrast, T790M was not found in the specimen from the pulmonary metastatic tumor. These findings strongly suggest genetic instability and heterogeneity of the lung tumor in this case.\nWe used oral TS-1 as the next regimen and achieved a good tumor response. However, she relapsed again. She received a salvage chemotherapy regimen comprising carboplatin and pemetrexed with bevacizumab, but this failed to inhibit tumor progression. We re-biopsied the pulmonary nodule by using computed tomography-guided needle biopsy. Gene analysis of the re-biopsy specimen revealed a unique deletion mutation in exon 19 and T790M. The L858R mutation was not found in this specimen. Subsequently, our patient received thoracic irradiation for the pulmonary nodule for locoregional control. |
A 32-year-old, previously healthy, African American man presented to an emergency department 45 minutes after the acute onset of left facial droop and right-sided weakness (Fig. ). A thorough history confirmed an episode 1-week prior, during which he developed sudden onset of dizziness associated with nausea and vomiting that resolved within hours. He denied any past medical or surgical history and was taking no medications. He has no family history of tumors. In the emergency room, his vital signs were within normal limits. His physical examination was significant for a left facial droop and right hemiparesis. Auscultation of his chest revealed a regular rate and rhythm with no appreciable murmur. No additional significant findings were noted. Stroke protocol was initiated. A chest X-ray was normal and an electrocardiogram showed normal sinus rhythm. A head computed tomography (CT) scan was negative for signs of intracranial hemorrhage. He was subsequently started on tissue plasminogen activator (tPA) therapy. Magnetic resonance imaging (MRI) of his brain demonstrated a right basal ganglia infarct and an old left cerebral infarct. A carotid ultrasound was negative. TTE demonstrated a 1 cm by 1 cm mass on the posterior leaflet of the mitral valve with a moderate mitral regurgitation In addition, TTE revealed a questionable mass on the left coronary cusp of the aortic valve. These findings were confirmed with TEE (Fig. ), which verified no sign of endocarditis and no atrial septal defect. A complete hypercoagulable workup was negative. Stroke protocol continued with the working diagnosis of cerebrovascular accident secondary to emboli from the mitral valve mass. Within 24 hours, he regained function of the right side of his body and had complete resolution of symptoms. He was diagnosed as having transient ischemic attack (TIA) and discussion was undertaken regarding surgical excision of his mitral valve mass.\nA median sternotomy was performed and cardiopulmonary bypass was employed via aortic and bicaval cannulation with full anticoagulation. His aorta was cross-clamped and his heart arrested with retrograde cardioplegia. The aortic valve was examined through an ascending aortotomy and all three valve leaflets appeared normal. A left atriotomy was made and the mass was easily identified on the posterior mitral valve leaflet adjacent to the mitral valve annulus (Fig. ). The mass was excised and a frozen section confirmed globular myxoma cells with abundant eosinophilic cytoplasm consistent with myxoma. The valve leaflet was reconstructed with an autologous pericardium patch and the annulus was supported using a running DeVega-type suture. The valve appeared normal and was tested; no regurgitation was noted. His left atrium and aorta were closed. His aorta was unclamped, after aggressive venting and de-airing maneuvers, and his heart returned to normal sinus rhythm with successful weaning from cardiopulmonary bypass. Anticoagulation was reversed with protamine and his chest was closed after placement of drains and pacing wires. At the conclusion of the operation, TEE confirmed appropriate mitral valve function and normal aortic valve with no evidence of a mass and no regurgitation at either location. |
A 32-yr-old Korean male patient was admitted to Inha University Hospital, Incheon, Korea due to dyspnea and right chest pain for 1 day. On physical examination, the breath sound in the right lung fields was decreased without shifting of the maximal point of the cardiac impulse. He had a history of the right pneumothorax one and a half years ago, which was treated with tube thoracostomy. He had no history of pulmonary or bronchial tuberculosis before this admission. He was a smoker. The posteroanterior chest radiograph revealed increased radiolucency along with overinflated lung parenchyma and sparse vasculature in the upper half of the right lung. Also note v-shaped branching opacity was in the right parahilar area. Pneumothorax was associated in the right lower pleural cavity (). A 32 Fr chest tube was inserted into the right pleural cavity. The chest CT taken in the state of full expansion of the right lung after tube thoracostomy showed a branching soft tissue density in the region of the posterior segment of the right upper lobe. The orifice of the posterior segmental bronchus was visualized but the orifice of the subsegmental branch of the posterior segmental bronchus could not be visualized separate from the origin (). There were no endobronchial lesions in the bronchial tree on the bronchoscope and each orifice of the segmental bronchi of the right lung was seen normal. His forced expiratory volume at 1-sec (FEV1) was 3.49 L (91%) and forced vital capacity (FVC) was 4.57 L (100%). Perfusion lung scan showed a perfusion defect in the right upper lobe. Exploratory thoracotomy was done via muscle sparing vertical thoracotomy skin incision. There was a localized emphysematous change in the posterior segmental area of the right upper lobe with the apical pleural adhesion and the remaining lungs were normal. Right upper lobectomy was successfully undertaken. The pathological findings of the resected right upper lobe showed overinflation of the posterior segment. There were no obstructed lesions of the orifices of the three segmental bronchi of the right upper lobe. However, one of the subsegmental branch of the posterior segmental bronchus was obstructed and there was a 2.5×1.5×1.5 cm sized cystic mass containing brownish mucus material at the distal portion of the obstructed subsegmental bronchus. The cystic mass was not connected with other bronchial trees. Microscopically the distal air spaces of the atretic segmental bronchus showed overinflation only. However the foci of the subpleural bullae in the overinflated segment were observed (). His postoperative course was uneventful. He was discharged on the post-operative 7th day. He has been well 1.6 yr after the operation. |
A 75-year-old woman was referred to our hospital complaining of a huge swelling in the left side of her face and neck. She stated that this mass had begun to develop 15 years ago at which it was only the size of a small almond at the left side of her neck. The mass was painless and gradually increasing in size but the patient refused to get medical consultation throughout these years till she started to feel serious difficulty in breathing.\nClinical examinations revealed a huge firm mass in the left side of the face and neck crossing the midline reaching the right side of the neck and inferiorly till reaching below the left inframammary sulcus. The mass itself was not tender, with nodular bosselated outer surface and the surrounding skin was congested in some areas (, ).\nThe origin of the mass couldn’t be determined clinically whether it arised from parotid, submandibular or even thyroid gland. A core-needle biopsy suggested a salivary glandular origin.\nCT scan with contrast of the neck was done and revealed a huge mass (34 × 20 × 26 cm) arising from left side of the neck and extended to parapharyngeal and sublingual spaces with displacement of trachea to the right side. It was found separated from thyroid gland and surrounding structures. CT angiography was also done and showed that the main arterial supply was from the facial branch of the left external carotid artery, common, external and internal carotid arteries on both sided which were patent and of normal shape (, ).\nThe patient was prepared for surgery. Anesthesia team used fibreoptic intubation. We started with a left inferolateral incision to separate the tumor from the neck great vessels. After securing them, we continued the dissection both superiorly and inferiorly in the medial direction to the right side (, ). There was no evidence of surrounding tissue infiltration. We were able to remove the tumor completely which weighed 8.1 kg (). The tumor had a very wide base therefore the defect couldn’t be closed primarily and we used a thiersch graft ().\nThe histopathology report grossly stated that the mass had lobulated irregular outer surface, and was covered by a skin flap measuring 30 × 10 cm which was grossly unremarkable. Cut section was heterogeneous showing yellowish friable areas admixed with whitish glistening areas, with wide areas of haemorrhage and cystic degeneration. Ten paraffin blocks were prepared. Microscopic analysis yielded a biphasic benign mixed tumoral proliferation formed of epithelial and myoepithelial components that were embedded against a myxochondroid matrix. The epithelial component comprised ductal structures lined by bland looking cuboidal cells. These were associated with clear plumpy, rounded epitheloid and plasmacytoid, as well as spindle shaped myoepithelial cells admixed with cartilaginous foci against myxoid background (, , ). Multiple areas of infarction necrosis were seen with ghosts of cells appearing, admixed with areas of dystrophic calcification (, ). There was no detected atypia or malignancy in all dissected areas with no evident mitotic figures nor metaplastic elements. The tumor had very low proliferation index as proved by ki67. It exhibited nuclear positivity in only about 2% of tumor cells ().\nThe patient was discharged three days postoperatively in a good general condition with marvelous improvement of her respiration and facial appearance.\nTo date, the largest ever recorded PA arising from the submandibular gland was 22 × 25 × 19 cm []. Our case measured 34 × 20 × 26 cm and weighed 8.1 kg. |
This is a case of a 59-year-old male who presented to a fracture clinic with a 7 month history of an infected surgical site wound overlying the left tibia, which was thought to be complicated by underlying osteomyelitis. During his initial consultation, the patient reported that, 7 months prior, he was hit by a motorcyclist while crossing a road in Bangkok (Thailand), where he had sustained a displaced spiral fracture of the left distal tibia. He was subsequently operated on in Thailand, where he underwent an open reduction and internal fixation. Upon return to the UK his surgical site wound began to discharge, over the course of a 7 month period prior to presentation to our institution. The patient reported that he had received multiple empirical courses of antibiotics for a non-healing surgical site wound. He had no significant past medical history, nil reported allergies and was fully mobile prior to this incident.\nThe patient was admitted to hospital (Day 1) for further management of his infected surgical site wound. Initial laboratory analyses including a full blood count, liver function tests and serum urea and electrolyte tests were conducted upon admission. All results were unremarkable. An X-ray of the left tibia and fibula performed on admission indicated periprosthetic lucencies. There was no evidence of healing at the site of the proximal fracture aligning with the initial clinical presentation of osteomyelitis.\nThe patient was subsequently taken to theatre on Day 4 for removal of all metalwork (including 12 screws), debridement and deep tissue and bone sampling for bacterial culture. During this operation vancomycin was incorporated in bone cement and intravenous (IV) ceftriaxone (2 g/q24h) was initiated pre-operatively. Purulent discharge was encountered from the skin and deeper skin layers from the distal tibia during the procedure. A CT scan of the left lower leg was performed the following day and suggested osteomyelitis of the medial aspect of the left distal tibia. Twenty-four hours after the operation, the infection team were consulted, and IV ceftriaxone was switched to IV vancomycin and oral ciprofloxacin (750 mg/q12h) was added to broaden empirical aerobic Gram-negative antimicrobial cover for a confirmed osteomyelitis. Vacuum-assisted closure (VAC) therapy was applied to the wound to aid healing and the patient continued to remain clinically stable and afebrile.\nOn Day 7 a pan-resistant Pseudomonas aeruginosa was isolated from a rectal swab screening for carbapenem-resistant organisms, which showed resistance to gentamicin, meropenem, ceftazidime, ciprofloxacin and piperacillin/tazobactam. An IMP1 MBL gene was detected from the isolated organism suggesting that the patient was colonized with a carbapenemase-producing organism. Six days after the operation, preliminary culture results of the bone samples revealed a polymicrobial infection. Nine bone samples of the left tibia grew P. aeruginosa whilst five of the samples also grew Morganella morganii. One of the bone samples also grew Staphylococcus epidermidis. The IV vancomycin was continued (whilst waiting for final phenotypic susceptibilities) together with oral ciprofloxacin, and the patient did not display any signs of clinical deterioration whilst waiting for extended susceptibilities. The patient returned to theatre on Day 12 for further bone sampling of the left tibia and debridement of the wound.\nFinal culture of bone samples on Day 17 revealed a pan-resistant P. aeruginosa and confirmed resistance to ceftolozane/tazobactam, ceftazidime/avibactam, gentamicin, aztreonam, cefepime, ceftazidime, meropenem, piperacillin/tazobactam and ciprofloxacin, whilst showing susceptibility to amikacin, colistin and cefiderocol. The P. aeruginosa was found to harbour the IMP MBL gene. M. morganii was susceptible to ciprofloxacin, gentamicin, temocillin, ertapenem and co-trimoxazole. Antibiotic susceptibility was confirmed by the reference laboratory. On Day 17, antimicrobial therapy was rationalized on the basis of this culture result with the addition of high dose IV colistin (9 million units loading followed by 3 million units/q8h), continuation of oral ciprofloxacin and cessation of IV vancomycin.\nSix days into IV colistin therapy, the patient developed an acute kidney injury (AKI) with a rapid rise in creatinine. A baseline creatinine of 65 µmol/L rose to 160 µmol/L (reference value: 60–125 µmol/L) and was classified as an AKI stage 3 as per the RIFLE and KDIGO systems. The nephrology team were consulted who made the diagnosis of acute tubular necrosis (ATN) secondary to colistin on the basis of medical imaging (renal ultrasound) and clinical presentation of the AKI. Colistin therapy was suspended, and oral ciprofloxacin therapy was continued to provide antimicrobial treatment for the M. morganii isolated. The AKI slowly began to recover 4 days after colistin cessation.\nDue to the lack of treatment options and possible risk of amputation if there was further progression of the infection, compassionate use of cefiderocol was pursued. Approval was granted by the manufacturer (Shionogi) and consent for use was gained from the patient. Cefiderocol susceptibility testing was performed using disc diffusion and the P. aeruginosa was deemed to be susceptible. The patient initiated cefiderocol therapy (1.5 g/q8h infused over 3 h) 10 days after discontinuation of colistin therapy on Day 32, which was dosed according to renal function as per the protocol established by Shionogi (creatinine = 139 µmol/L, creatinine clearance = 40 mL/min). Oral ciprofloxacin was continued to ensure the M. morganii isolated was adequately treated. During the following weeks, there was modest clinical improvement of the surgical site wound. The patient’s renal function returned to baseline with complete resolution 17 days post-discontinuation of colistin therapy. The patient reported no drug-related effects or infusion site reactions and weekly monitoring of bloods showed no untoward effects. Thirteen days after initiation of cefiderocol, the renal function had sufficiently improved to allow the dose of cefiderocol to be increased to 2 g/q8h. Cefiderocol and ciprofloxacin were both discontinued after completing 28 days of treatment, 60 days after he was initially admitted to hospital, and he was discharged home once medically stable.\nHe was reviewed in an outpatient clinic 3 months later and there was no evidence of persistence or relapse of infection. The patient reported significant improvement in pain and swelling following the surgery and completion of antibiotic therapy. The post treatment X-ray showed improved bony remodelling over the tibia and fibula with good bony alignment and no adverse features. Physiotherapy support was continued for 4 months following treatment which resulted in good functional mobility and improved proprioception. He was subsequently discharged from the physiotherapy service once he had regained the ability to fully bear weight on the left leg. The patient has since remained off antibiotics without clinical evidence of infection and has returned to work. |
A 27-year-old woman presented with a nodule at the lower abdominal caesarean section scar. She had undergone surgery 4 years before. The tumor appeared over the caesarean section scar 4 years after surgery, which gradually increased in size. The patient described cyclical pain at the site of the mass, which coincided with her normal menstrual cycle. She had no previous history of endometriosis. On physical examination, the patient had a firm mass with restricted mobility along the right upper lateral aspect of the caesarean section scar with a black appearance. The mass measured about 5 cm × 5 cm in size and was clinically diagnosed as a tumor on the abdominal wall. It was clinically diagnosed as either metastatic deposit or skin appendage tumor. The patient was found physically fit otherwise.\nFNAC was performed and the aspirate was obtained using a disposable 10 mL syringe and 22 gauge needle. The material was collected on glass slides and was wet fixed and then stained by the Papanicolaou method, while the remaining aspirate was allowed to clot. To facilitate clotting, few drops of blood from the finger prick of the patient was added on the aspirate and the clot was transferred to a formalin vial. This method of cell block preparation has been practiced in our institute since long.[]\nA cell block was made from formalin-fixed sediment. Sections were cut and stained with the hematoxylin – eosin stain.\nSmear preparation of the FNAC sample showed extensive areas of hemorrhage and scattered small cells with compact nuclei and scanty cytoplasm, resembling stromal cells and columnar epithelial cells, which represented the endometrial glands. Hemosiderin pigment-laden macrophages were also seen []. The diagnosis given on FNAC was suggestive of scar endometriosis.\nThe cell block findings were confirmatory of endometriosis as they revealed endometrial lining columnar epithelium with subepithelial dense and compact endometrial stroma along with extensive areas of hemorrhage [].\nHistopathological findings after complete excision of the nodule also confirmed endometriosis [Figure and ]. |
A healthy 8year old male child presented with a history of a gradually increasing swelling around the right shoulder joint since 2 months. It had been painful right from the beginning and associated with mild restriction of movements. He denied any history of trauma or any other systemic complaints. The swelling progressed in size over next month. On examination there was a tender swelling on the proximal anteromedial aspect of shoulder. The swelling was not fixed to underlying bone and overlying skin. The mobility of swelling was less in longitudinal direction. Abduction and forward flexion of the shoulder were painfully restricted.\nRadiological examination revealed multiple well defined swellings on anteromedial aspect of proximal humerus with calcified wall. One of the swelling was approximately 2cm X 2cm. This picture was suggestive of calcified hydatid cyst or chondrocalcinosis. Total blood count was 12000 with predominance of leucocytes with normal eosinophilic count. Ultrasound of abdomen and chest showed no evidence of hydatid cyst. Ultrasound guided aspiration of the large swelling was suggestive of inflammation with no evidence of infection.\nMRI showed well defined lesion with calcific wall with multiple loose bodies. These swelling were arising from biceps tendon sheath. MRI reported normal articular cartilage of glenohumeral articulation without any loose bodies inside the joint. Radiologist opined in favor of a synovial chondromatosis and gave a differential diagnosis of calcified hydatid cyst. There is only one reported case of extra articular synovial chondromatosis in subacromial bursa around shoulder.\nConsidering patients symptoms we decided to excise the swelling. Under general anaesthesia the lesion was approached through delto pectoral approach. The proximal part of pectoralis major insertion was released to expose the swelling. Well defined swelling was arising from biceps tendon sheath, with multiple other small lesions. No communication was seen to shoulder joint. All the swellings were excised. To ensure complete excision intra operative image intensifier was used. Child reported complete resolution of pain at three week after the surgery.\nHistopathology revealed circumscribed lobulated nodules of osteocartilaginous tissue with the stroma consisting of cellular fibroblastic tissue. Some fragments were covered with synovial tissue. There was no evidence of infection or malignancy in the specimen, and this was consistent with the appearance of synovial chondromatosis.\nThe child was symptom free at 1 year after the surgery. There was no pain and limitation of motion then. |
A 45 year-old male patient reported with the chief complaint of pain, swelling, and pus discharge from the maxillary left canine of one month's duration. The tooth was previously sensitive to hot and cold, and had recently developed spontaneous pain and pus discharge. Periodontal probing depths were 8 mm mesially, 6 mm labially, and 7.5 mm distally [Figure –]. A periapical radiograph showed a widening of the periodontal ligament space in the periapical area with an infrabony defect on the mesial aspect of the tooth []. An endoperio lesion associated with maxillary left canine was diagnosed.\nAn access cavity was prepared and the root canal system was cleaned and shaped in the first session with abundant 5.25% sodium hypochlorite irrigation. The canal was dressed temporarily with calcium hydroxide and the access cavity sealed with IRM cement. The patient was asked to stop all analgesic drugs and was given an appointment in seven days to continue the root canal treatment. He was also asked to contact the dental centre if there were any complaints. He was completely comfortable without any need for analgesia. The patient returned after a week and the absence of pain or signs of inflammation indicated that the final filling could be placed. This was completed with gutta-percha and a root canal sealer. A one-month recall revealed a stable situation and disappearance of pain, however, the pockets persisted around the tooth. It was decided to correct the defect after one month after the endodontic therapy, using autologous platelet concentrate mixed with an alloplastic bone graft substitute. The site was surgically opened up for debridement and a circumferential defect was evident around the tooth []. The platelet-rich concentrate was mixed with an alloplastic bone graft substitute to obtain a gel-like consistency [Figure and ]. This gel was placed to cover the exposed root and fill the defect [].\nThe clinical appearance of the tooth had improved considerably at the time of evaluation three and six months following treatment. The periodontal pockets had reduced from 8 mm to 0.5 mm mesially, from 6 mm to 1 mm labially, and from 7.5 mm to 1 mm distally [Figure –]. Radiographic evidence showed a significant bony fill []. The results were stable and maintained at the end of nine-months' follow-up. |
A 72-year-old male with a distant history of alcohol abuse presented with approximately 6 months of memory loss, difficulty expressing himself, and a progressively worsening gait. A magnetic resonance imaging (MRI) of the brain revealed a 2.5 × 3.7 × 2.5 cm heterogeneously enhancing cystic mass centered within the cerebellar vermis with mass effect on the fourth ventricle and mild ventriculomegaly [Figure and ] as well as an 8 × 6 × 4 mm satellite lesion within the right superior cerebellar peduncle [Figure and ]. On examination at an outpatient clinic, the patient had a slight expressive aphasia but was otherwise neurologically intact. At that time, the differential diagnosis of the lesion included primary glioma versus metastatic or granulomatous disease and the decision was made for surgical resection of the lesion.\nRoutine preoperative testing found the patient to be in rapid atrial fibrillation and he was sent to the emergency room. While undergoing evaluation of the arrhythmia, his rhythm converted to a junctional bradycardia, necessitating placement of transvenous pacemaker. In the absence of other identifiable etiologies for his sudden bradycardia, his cardiac symptoms were attributed to intracranial hypertension secondary to the obstructive hydrocephalus caused by the vermian lesion. A computed tomography (CT) scan of the head revealed ventriculomegaly but no frank hydrocephalus and the patient was taken to the operating room for placement of a ventriculoperitoneal (VP) shunt. Intraoperatively, he was found to have elevated intracranial pressure. His postoperative course was notable for immediate resolution of the cardiac arrhythmia.\nTwo days following placement of the VP shunt, the patient underwent a suboccipital craniectomy for resection of the cerebellar lesion. Intraoperatively, the lesion was found to be soft and mildly vascular with clean margins relative to the surrounding brain. Pathology specimens were sent for frozen histological analysis and were thought to be most consistent with a diagnosis of glioma. A postoperative MRI revealed a subtotal resection of the lesion. Permanent section of the lesion demonstrated a glial neoplasm composed of astrocytes with elongated, bipolar cytoplasmic processes, forming fascicles and focally, loose perivascular patterns, dispersed within an abundant myxoid background [Figure and ]. The tumor cells had round to irregular, hyperchromatic nuclei, with prominent perinuclear halos infiltrating the molecular layer of the adjacent cerebellar folia. They were strongly immunoreactive for GFAP and WT-1 with a MIB-1 labeling index of greater than 5% [Figure and ]. The lesion was diagnosed as a WHO grade 2 PMA. Postoperatively, the patient did well and was discharged to the inpatient rehabilitation service on postoperative day 4 and discharged home 12 days later with residual dizziness and gait imbalance. Following discharge, the patient refused further medical or surgical treatment. His neurologic status and overall health gradually deteriorated and he expired 11 months after initial symptom presentation and 4 months after surgery. |
A 75-year-old male patient was sent to our hospital suffering from left hip pain sustainedly after an accidental fall. Radiographic imaging revealed that this patient was affected by Crowe type IV DDH combined with an Evans type III intertrochanteric fracture of left femur (Fig. a). The patient reported a history of left hip trauma at the age of 10, at which time he did not accept proper treatment. The resultant left hip deformities had since caused the patient to walk with a limp. Prior to his fall, the patient was able to bear weight and work with moderate left hip pain. Physical examination revealed the left leg to be 4 cm shorter than the right leg, with local tenderness being detected in the left hip. In addition, percussive pain in the left femur in the axial direction and pain with internal and external rotation were noted. As passive activity caused serious pain, we were unable to measure the degrees of left hip motion. The patient had been diagnosed with hypertension for six years, and regularly took blood pressure control medications. Computed tomography scans revealed the high posterior dislocation of the left femoral head up to the level of the greater sciatic foramen(Fig. b). The original acetabulum was smaller and shallower (Fig. b). In order to better preoperatively evaluate the acetabulum and femur deformities in this patient, a 3D printed model was constructed (Fig. c).\nThe patient was offered two surgical options: THA or internal fixation to treat only the intertrochanteric fracture. After careful consideration and discussions with family members and the surgeon, the patient selected to undergo THA, and also provided informed consent for the publication of his case, which has not been reported previously to our knowledge.\nUnder general anesthesia, the patient was placed in the lateral decubitus position. The operation was performed via a posterior approach with an incision length of almost 20 cm. The external rotators were first detached, and then the femur neck was removed. After resection of the elongated hypertrophic joint capsule, the original acetabulum was clearly exposed and gradually reamed to 50 mm. Next, the 50 mm acetabular cup and a 28 mm polyethylene liner were placed in an appropriate anatomic arrangement with three screws. To clearly expose the proximal femoral canal, the femoral great trochanter fragment was overturned along the fracture line. Approximately 2 cm below the lesser trochanter, a 4 cm-long femoral shortening transverse osteotomy was performed by resecting the femur in accordance with preoperative planning. A modular S-rom femur stem was then installed from the level of intertrochanteric fracture end into the canal, and the femur was then de-rotated and the resected cylindric bone segment was cut longitudinally, after which these two pieces were bound onto the osteotomy site with wires. Equipped with 32 mm short metal head, the stem was easily reduced, after which the intertrochanteric fracture was reduced and stably fixed with steel wires.\nPostoperatively, the patient was administered intravenous antibiotics and prophylactic anti-thrombotic treatment. Three days postoperatively, the patient was encouraged to stand and to walk with the aid of a walker. Postoperative plain radiographic images revealed that the original rotational center of the hip was restored and that the inclination and anteversion of the cup had been restored (Fig. a, b). The patient completed baseline visual analog scale (VAS) score and modified Harris hip score (mHHS) assessments via retrospective questionnaire, and also completed these assessments at 3 and 6 and 10 months postoperatively. Both VAS and mHHS scores were significantly improved at these postoperative follow-up time points (Table ). Radiographic and computed tomography scans conducted upon most recent follow-up revealed that the prosthesis was properly positioned and the intertrochanteric fracture and subtrochanteric osteotomy had healed effectively (Figs. c, d, ). Leg length discrepancy (LLD) measurements through the full length weight bearing radiograph of lower extremities showed the left limb to be 1 cm shorter than the right limb(Fig. e). Even though there was a little heterotopic ossification occurred on greater trochanter of left femur,the patient almost felt no pain and could walk up and down stairs without any assistance. |
A 74-year-old male patient was admitted to our hospital in March 2017 to undergo liver resection to treat a malignant hepatic lesion diagnosed with CT and PET and a fine-needle biopsy positive for squamous carcinoma. The hepatic tumour discovered during follow-up for a previous bladder cancer submitted to endoscopic surgery three years before measured 22 mm in diameter and was located in the VIII Couinaud's segment [] of the liver in association with three smaller hypodense liver lesions with a focal dilatation of peripheral biliary tree ().\nThe case is discussed with radiologists, oncologists, and pathologists of our hospital. Even if the lesion had been the single site of disease; due to the proximity/doubtful infiltration of the lesion to the biliary tree, we decided to submit the patient to an explorative staging laparotomy and possible palliative surgery.\nOur internal protocol states that during the preadmission every patient who is a candidate for a liver resection is subjected to a routine liver function test with ICG to determinate the most appropriate surgical procedures []: 0,5 mg/Kg ICG are routinely injected intravenously up to seven days before surgery to evaluate the ICG retention rate at 15 min (R15). In our case 45 mg of ICG was intravenously administrated to test hepatic function, ten days before the surgery (patient R15 = 8.9).\nThanks to the ICG property of being fluorescent with the light emitted from the photodynamic eye of the laparoscopic system in our possession, it is possible to visualize the lesion during the surgical procedure. To this target, timing of administration and dose of ICG are key points.\nSeveral studies have demonstrated that the effective dose of ICG depends on the timing of injection; in particular, if the function liver test had been performed more than 7 days before surgery it would have been necessary to administer an adjunctive dose (0,1 mg/Kg) the day before []. In this case, it was necessary to administrate an adjunctive dose of ICG the day before the surgery (9 mg of ICG injected intravenously). After laparotomy, exploration of the abdominal cavity, and exposure of the liver, we easily confirmed the superficial lesion in the VIII Couinaud's segment. The liver surface has been analysed with the fluorescent imaging system. The fluorescing tumour has been clearly identified and defined on the liver surface, as shown in . We have also identified that a large area of fluorescent parenchyma that gets from the peripheral of the lesion up to the portal pedicle such as the neoplasia would interest the right biliary tree in the form of neoplastic lymphangitis (). This datum was not preoperatively known.\nA right hepatectomy would have been the oncologically correct surgical procedure due to the infiltration of right biliary duct. Considering the probable metastatic nature of the lesion, the absence of a clearly primary lesion, the age, the comorbidities, and the small size of residual liver, we have decided to perform an atypical segmental resection of S8 associated with cholecystectomy and lymphadenectomy of the hepatic pedicle nodes, including the area of impaired biliary excretion.\nAt the histological examination, the lesion, the lymph nodes of the hepatic pedicle region, and the right biliary branch, respectively, resulted in hepatic metastases from squamous cell carcinoma and sites of metastatic location. As expected the resection margin was interested by neoplasia.\nIn particular, the histological examination showed the following:Macroscopical exam: the neoplasia, in a site, appears to be in contact with the resection margin Microscopical exam: parenchymal hepatic section that showed metastatic localization of squamous carcinoma moderately differentiated. The neoplasia interest the surgery resection margin.\nIn this case, fluorescent imaging has revealed a fluorescing ring around the hepatic metastasis (). The fluorescence of the cholestatic area was shown on the cut surface (). |
A 64-year-old man with a 3-month history of non-specific neurological symptoms, such as vertigo and syncope, was admitted to our hospital. He presented with cardiovascular risk factors: grade 3 hypertension and hypercholesterolemia, and also with bilateral stenoses of the common iliac arteries. Doppler sonography revealed occlusions of the BCT and of the left SA, and stenosis of the left ICA caused by a long atherosclerotic plaque, with increased systolic and diastolic flow velocities to 350 cm/s and 120 cm/s, respectively. In the past, in another medical centre, he underwent an unsuccessful attempt of endovascular revascularization of the left SA.\nAfter a thorough diagnostic evaluation, including assessment of the patient's neurological status and the above-mentioned sonographic examination, we decided to perform catheter angiography of the arteries supplying the brain, in order to obtain more information about cerebral hemodynamics. This examination revealed occlusion of the BCT localized about 1 cm from the aorta, occlusion of the left SA localized about 2 cm from the aorta, and also an 80–90% stenosis of the left ICA (). Morphology of these lesions was typical of atherosclerosis and not of fibromuscular dysplasia or Takayasu disease. Moreover, a detailed analysis of angiographic pictures revealed the presence of a double steal syndrome. First, there was reversed flow via collateral from the left ECA to the left SA, and then to the left vertebral artery (VA) and the basilar artery. This collateral network constituted the main blood supply to the posterior part of the brain, since there was no inflow of injected contrast from the left carotid artery through the Willis circle to the basilar and cerebellar arteries ().\nTaking into account this unique hemodynamic pattern, and also the general condition of the patient, we decided to reconstruct the arteries using endovascular technique in two stages. Also, as the first procedure we decided to improve the inflow to the posterior part of the brain, reconstructing the left SA and restoring the proper flow in the left VA. An additional benefit of such a therapeutic scenario would be the possibility of adequate management of arterial hypertension (the patient presented with a history of severe arterial hypertension, yet because of bilateral occlusions of the SAs it was impossible to measure his arterial pressure non-invasively; arterial pressure measured intraoperatively in the aorta was 240/140 mm Hg). Reconstruction of stenosed ICA and of occluded BCT was planned as the second step. In our opinion, alternative therapeutic scenarios were associated with a risk of intraprocedural stroke in the posterior brain territory or other severe complications. For example, stenting of the left ICA performed as a first step could decrease the flow through the ECA, which in turn would compromise the cerebral circulation in the posterior part of the brain. Similarly, to prevent intraoperative stroke of the brainstem and cerebellum, surgical endarterectomy of the left ICA would require the use of an atypical shunt into the ECA, with potential technical problems associated with such a modified procedure. The first endovascular reconstruction was performed using femoral and radial artery vascular accesses. After cannulation of the femoral artery, a 6F 90-cm-long introducer sheath (Cook, Cook Medical Inc., Bloomington, IN, USA) was positioned in the aortic arch. We then navigated through the occluded left SA using Terumo Glidewire (Terumo, Tokyo, Japan) and V-18 ControlWire (Boston Scientific, Natick, MA, USA) hydrophilic-coated guidewires, and afterwards using 5F diagnostic catheters: VERT Beacon Tip Torcon NB Advantage Catheter (Cook Medical Inc, Bloomington, IN, USA) and Judkins Right (Cordis, Fremont, CA, USA). Once we had navigated through the occlusion, via the left radial access (5F sheath) we introduced a Hi-Torque coronary guidewire (Abbott Vascular, Abbott Park, IL, USA) into the left VA, and then via the femoral access we implanted a balloon-expandable 8 × 59 mm Omnilink stent (Abbott Vascular, Abbott Park, IL, USA) into the left SA. Since the proximal portion of the left SA was patent, without atherosclerotic plaques, we did not implant the subclavian stent typically, i.e. with a protrusion of the stent into the aorta. In this particular case such a protrusion was not needed, and, moreover, a protruding stent could make future endovascular repair technically difficult. The expanded stent also covered the origin of the left VA, since atherosclerotic plaque occluding the left SA was also located in this area. Then, through the mesh wall of the subclavian stent over the 0.014-inch guidewire we introduced a 4.5 × 20 mm Sterling Monorail angioplastic balloon (Boston Scientific, Natick, MA, USA) to the VA, and a 7 × 59 mm angioplastic balloon (Omnilink, Abbott Vascular, Abbott Park, IL, USA) to the stent. Afterwards we inflated both balloons, using the kissing-balloon technique, under the pressure of 10 bar. As a result, there was a good flow through the left SA and the left VA, with normalized circulation in the left upper extremity, as well as in the posterior part of the brain. Yet, there was still a reversed flow in the right VA ().\nThe second stage of endovascular treatment, aimed primarily at the repair of the left ICA, was performed 40 days later. Control angiography confirmed the success of previous interventions, but still with the signs of right-sided subclavian steal syndrome and stenosis of the left ICA. We decided to reconstruct the lesions of the left ICA during a single procedure via the right femoral access. We introduced a 6 Fr 90-cm-long introducer sheath (Cook, Cook Medical Inc., Bloomington, IN, USA) to the left common carotid artery and we performed angioplasty of the left ICA using a 3.5–5.5 mm FilterWire EZ distal protection system (Boston Scientific, Natick, MA, USA) and successfully implanted a 7 × 30 mm self-expandable Carotid Wallstent stent (Boston Scientific, Natick, MA, USA) (). There was good flow in both carotid and vertebral arteries after the procedure.\nThe patient was discharged after 4 days of hospitalization with the recommendation of dual antiplatelet therapy (aspirin 75 mg/day and clopidogrel 75 mg/day), antihypertensive therapy (ramipril 5 mg/day and torasemide 2.5 mg/day) and cholesterol-lowering treatment (atorvastatin 40 mg/day). Control Doppler sonography performed 30 days later demonstrated good flow through implanted stents with no sonographic signs of stenosis.\nInitially the reconstruction of the occluded BCT was planned as the third step, using a distal protection system, such as the Spider FX embolic protection device (ev3 Endovascular, Plymouth, MN, USA). Still, the timing of such an endovascular repair of the BCT depended on the neurological status of the patient, primarily the existence of symptoms of brain ischemia. Since, for the time being, the patient is free of neurological symptoms, we decided to manage him conservatively. |
A 50-year-old man was admitted to the General Hospital of the People's Liberation Army after complaining of a month of dysphagia, six months of vomiting, and infrequent regurgitation of a fresh mass into the oral cavity (Fig a). No treatment was provided until the dysphagia became worse. Barium swallow demonstrated a giant neoplasm which almost occupied the whole esophageal lumen, 22 cm apart from the incisor and 18 cm in length. The mucosal surface of the neoplasm was smooth, suggesting a giant submucosal neoplasm in the esophagus with a high probability of an esophageal polyp (Fig b). Chest computed tomography (CT) demonstrated a huge neoplasm, extending from the cervical esophagus to the cardia; the largest diameter of the polyp was 6 cm (Fig c). The lumen of esophagus was also distended. Ultrasonic endoscopy showed a neoplasm originating from the submucosa of the cervical esophagus, the base of which was about 5 cm in diameter, occupying almost half of the esophageal lumen (Fig d). These data indicated an esophageal polyp measuring 18 cm in length and 5–6 cm in diameter, extending from the cervical esophagus to the stomach cavity. After preparation, the patient was treated via gastroscopy under general anesthesia. Because of the large dimension of the polyp, it could not be completely removed; therefore, it was cut into small pieces by snare and removed piece by piece. The procedure lasted about three hours and approximately two-thirds of the tumor was removed. Surgery was discontinued after heavy edema in the esophageal mucosa. After surgery, the patient was supported with nutritional therapy (oral food and water were not permitted), and orally treated with mannitol, dexamethasone, and gentamycin to reduce swelling and prevent infection. Gastroscopy was repeated a week later, resulting in the total removal of the tumor. The patient left the hospital and no abnormality was found by gastroscopy one month later (Fig ). Pathological examination confirmed that a fibrovascular polyp was successfully removed. The patient was followed up for more than two years and no recurrence was observed. |
A 10-year-old male child was brought by his parents with a 1-month history of irritability, restlessness, and increased anxiety on separation from parents. These symptoms were abrupt in onset, with the child suddenly refusing to play with his sister and other children in his neighborhood. He began asking his parents to be around him and cried if they did not listen. He insisted that his parents and siblings did not move away from him even for a minute to the point of not allowing them to go to another room. Within 2 days, the behavior increased to such an extent, that he would cry at the thought of separation from his parents and siblings. At one point, when the parents resisted his behavior, he got very angry and scratched himself on his face leading to abrasions. However, such symptoms were very discrete and were present only for some time during a day (usually from 1 to 5 pm everyday) with no exacerbating or relieving factors. For the rest of the day, though he appeared slightly anxious, he did not have as much separation anxiety.\nHis parents noticed that the child had difficulty completing any work at home like helping his mother in her chores, which he would do with ease otherwise. His handwriting had deteriorated and he was unable to complete his homework since a month. Over the next 4-5 days, his anxious behavior persisted even though his parents were present around him. He was taken to a psychiatrist where he was prescribed oral escitalopram (5 mg) and clonazepam (0.25 mg bid). However, despite being on these drugs for 15 days there was no improvement after which the child was brought to us.\nOn detailed probing, there was no recent stressor in the child's life either at home or school. The parents also revealed that the child stopped going to school for 5 days before the onset of these symptoms and that he was having mild grade fever for those 5 days, which receded without medications.\nHis birth and developmental history was normal. He was good in his studies and there was no past history of school refusal, truancy or failures or bullying at school. Physical examination revealed healed scar marks on his face, which occurred when he scratched himself in a fit of anger on one occasion. His systemic examination was normal. On mental state examination, he was anxious and irritable. Throughout the interview, he was clinging to his parents and did not allow separation from them.\nHe was advised complete blood counts, erythrocyte sedimentation rate (4 mm/h), liver function tests, renal function tests, 24 h urine copper levels, serum copper levels (9 μg/dl), serum ceruloplasmin levels (10 mg/dl) and anti-streptolysin-O (ASO) titers. Ophthalmological consultation was sought for a slit-lamp examination for Kayser–Fleischer ring. All his investigations were within normal limits, but his ASO titers were elevated (800 IU/ml). A pediatric consultation was then sought for the raised ASO titers. A detailed history by the pediatrician revealed multiple episodes of throat infection in the past 1 year.\nHe was started on oral cefixime (100 mg bid) with oral acetaminophen (40 mg/kg) for 10 days. On follow-up after 10 days, parents reported complete remission of his symptoms within 5 days of starting medications. |
A 37-year-old woman (gravida 2, para 1) was referred to our fetal unit at 39 weeks' gestation for evaluation of what appeared to be a small cystic mass in the fetal thorax. The pregnancy had been otherwise uneventful. Family history was unremarkable. Amniocentesis for karyotyping had been performed at 16 weeks for advanced maternal age and revealed a normal karyotype (46, XY). According to Italian guidelines, the patient had undergone ultrasound examinations at 20 and 32 weeks' gestation, and the stomach was normally seen located on the left in the abdomen. Routine ultrasound examination performed at 39 weeks showed a fetus with normal growth. The ultrasound examination was performed because, in our hospital, all patients are evaluated with a clinical and ultrasound examination at 39 weeks of gestation. On the transverse section of the thorax the heart appeared normally located, without mediastinal shift, and the lungs appeared to be of normal echogenicity and volume. A round cystic image, diameter 8 mm, was visible behind the heart in the posterior mediastinum (); in sagittal section it appeared to be in continuity with a small stomach located below the diaphragm (). During the ultrasound exam period, no modification of the cystic structure was detected. One possible diagnosis was hiatal herniation of the stomach into the thorax and counseling about the diagnostic suspicion of hiatal hernia, the differential diagnosis, and the likely postnatal complications was performed.\nAt 40 weeks' gestation, the patient was admitted for spontaneous labor. A male newborn, weighing 3460 g, was delivered vaginally with Apgar scores of 9 and 10 at 1st and 5th min, respectively. Postdelivery chest radiography revealed no pulmonary abnormalities and a normal diaphragmatic profile ().\nThe upper gastrointestinal tract series using barium confirmed the prenatal diagnosis of CHH, categorized as type I. During the examination, gastroesophageal reflux was observed, together with a sliding up and down of the stomach into the fetal thorax. The newborn was discharged with prophylactic therapy with ranitidine and domperidon. At six-month follow-up the baby is growing well with sporadic episodes of reflux and he is still on therapy. |
A 56-year-old Caucasian man presented to the hospital with a three-day history of a red, painful rash to the first and second digits on the right foot, suggestive of ischemia. The patient stated that three days prior to the development of the rash, he was experiencing numbness and tingling at the site of the eventual lesion. An abdominal aortogram with lower extremity runoff showed patent bilateral renal arteries, patent bilateral common and external iliac arteries, patent right profunda, and three vessel runoff with an intact pedal arch flow. The physical exam was notable for right foot first and second digit duskiness with extreme pain to palpation, along with left foot and leg erythema (Figure ). Bilateral dorsalis and posterior tibial arteries were palpable. He denied any B symptoms and did not demonstrate any lymphadenopathy on an exam. The rest of the patient’s physical exam was normal. His vital signs were also within the normal range. The patient’s history was notable for CLL diagnosed in 2011 through routine lab work showing leukocytosis, with subsequent confirmation via a bone marrow biopsy. He also had a history of untreated HCV thought to be contracted through his extensive intravenous drug use in the 1990s. He was treated for the HCV in 2012 with ribavirin and beta interferon and was cleared of the virus. As the CLL was asymptomatic and not appearing to progress, no treatment for this was commenced and he was under watchful monitoring by his outpatient oncologist.\nAs vasculitis was suspected on initial rheumatologic evaluation, the patient was started on prednisone 60 mg daily as workup commenced. A biopsy of the right foot ischemic lesion revealed ischemic necrosis of the epidermis and dermis, with vascular congestion but no evidence of vasculitis and negative direct immunofluorescence. Labs were obtained on admission and during his hospitalization (Tables -). A computerized tomography (CT) scan of his chest, abdomen, and pelvis was negative for malignancy. A transesophageal echocardiogram did not reveal any thrombus. A bone marrow biopsy showed CLL persistence, with an absolute neoplastic lymphocyte count of 19 k/ul. Interestingly, the peripheral blood smears showed an abundance of proteinaceous blue-gray material consistent with cryoglobulins (Figure ). Four days into the patient’s admission, he developed pain and swelling in his left foot, along with erythema. As the lesions on his left foot continued to worsen, he was treated with a course of high-dose intravenous (IV) methylprednisolone. As it was felt the patient had developed CLL-associated vasculitis, an initial dose of rituximab 375 mg/m2 was given according to the CLL dosing guideline. The day after rituximab infusion, bendamustine 100 mg/m2 was given over the course of the following two days. Despite these therapies, the patient developed erythema of his left hand and forearm (Figure ) and his fingers rapidly became necrotic (Figure ). He also developed ischemia and necrosis of his left upper ear. Due to the aggressive spread of cryoglobulinemic vasculitis, plasmapheresis was commenced on a near-daily basis. Plasmapheresis was striking for the amount of sediment in the collection bag effluent (Figures -). Given the positive cryoglobulin screen, the sediment was thought to be the large quantities of cryoglobulins the patient was producing. Two weeks after his first rituximab dose, he was given a second rituximab dose. He was then discharged home on prednisone 60 mg daily and plans for the continuation of outpatient plasmapheresis and the continuation of combination rituximab and bendamustine treatment. Six days after discharge, he was readmitted for worsening pain and necrosis to his left foot and left hand. The ischemia and necrosis were felt to be secondary to the ongoing cryoglobulinemic vasculitis attack. He was treated with another course of rituximab. Three days later, cyclophosphamide 1000 mg IV was given. The patient continued to deteriorate and he was given rituximab 375mg/m2 and cyclophosphamide 1000 mg together 17 days after the initial cyclophosphamide dose. He continued with near-daily plasmapheresis as well as oral steroids during this period. The patient eventually stabilized and did not develop new areas of ischemia. Unfortunately, the areas of prior necrosis were deemed unsalvageable and he subsequently underwent amputation of the distal digits of his left hand with a left forearm fasciotomy, left foot amputation, and amputation of the first two digits of his right foot. The patient was eventually transitioned to oral cyclophosphamide 75 mg daily as well as obinutuzumab (CD-20 directed cytolytic antibody) 1000 mg monthly infusion as the treatment for his CLL. Rituximab was deemed ineffective and discontinued. He also currently undergoes plasmapheresis once a week. He has so far not developed any new necrotic lesions. Aside from the physical exam, trending the Kappa/Lambda (K/L) light chain ratio has been a way of gauging the patient’s response to treatment. As seen in Table , the patient’s Lambda light chain number has reduced to a normal range during his treatment course, with subsequent normalization of the K/L ratio. Immunofixation on each cryoglobulin screen showed immunoglobulin M (IgM) monoclonal protein with Lambda light chain specificity. Immunofixation and electrophoresis of the cryoprecipitate revealed type I cryoglobulinemia. The cryoglobulin screen eventually turned negative, as noted in Table , attesting to the patient's treatment success thus far. Monitoring CD 19/20 counts is another likely method of assessing treatment efficacy but has, so far, not been pursued by the oncology service. |
A 59-year-old male patient with chronic alcoholism and hepatitis B virus carrier was diagnosed with alcoholic liver cirrhosis and hepatocellular carcinoma (HCC) two years ago. Then, he received transcatheter arterial chemoembolization therapy three times and has been living without recurrence. The patient visited our emergency department with the symptoms of headache beginning 10 days prior and progressive left hemiparesis, altered mentality occurring two days prior. He was afebrile and his vital signs were stable. There were no leukocytosis and C-reactive protein (CRP) was 4.04 mg/L of blood. Upon a neurological examination, he was drowsy with disorientation and revealed decreased upper and lower extremities motor power to grade IV. DWI of the brain was performed because of suspicion of cerebral infarction. It showed a multi-lobulated cystic mass lesion and associated mild edema located in the right parieto-occipital lobe. We considered the possibility of a metastatic brain tumor at the first impression owing to negative diffusion restriction sign and a history of HCC. Contrast enhanced MRI combined with DWI revealed a multi-lobulated cystic rim-enhancing mass with surrounding edema and hypointensity in the cystic cavity on the DWI (). Stereotactic biopsy with aspiration was performed on the assumption of HCC multiple metastasis in the brain and the result revealed BA involving multiple bacterial colonies. However, because the bacteria was not cultured, an initial antimicrobial therapy was started on the basis of the standard empirical treatment that consists of vancomycin plus a third-generation cephalosporin and metronidazole. Despite the use of the above antimicrobial therapy, clinical deterioration with an increasing abscess size on cranial imaging () made further stereotactic aspiration and cultures including fungus, parasite and tuberculosis mycobacterium. The amount of vancomycin dosage was increased in order to increase the CSF concentration of vancomycin but intermittent spiking fever continued and patient's clinical symptoms did not improve. Even though there were no bacterial growth in the cultures, considering the situation that antimicrobialresistant gram-positive strains is increased, we had to change the previous antibiotics to linezolid which is excellent on CSF penetration. Then, infection signs including fever, erythrocyte sedimentation rate, CRP, and leukocytosis were decreased immediately. The abscess size was also reduced in follow up brain CT after about 4 weeks of using the antibiotics () and the post-linezolid clinical course was uneventful. This case was approved by the Institutional Review Board of the hospital (EMCIRB18-77). |
A 33-year-old female gravida 2 para 0110 presented to the obstetric and gynecologic clinic to establish prenatal care. Her estimated gestational age determined by last menstrual period was 10 weeks 6 days. A diagnosis of missed abortion was made after ultrasound revealed a crown rump length of 9.2 mm, consistent with a gestational age of 7 weeks 0 days, with no fetal heartbeat identified.\nThe patient had a history of normal 28-day menstrual cycles and used a combined oral contraceptive pill prior to this pregnancy. She reported a history of polycystic ovary syndrome, human papillomavirus infection, and CIN grade 3 (CIN 3) diagnosed 3 years prior. Two years prior to the current visit, the patient received cryotherapy treatment for CIN 3 that failed to resolve her cervical dysplasia as evidenced by a repeat Pap smear. She was offered CKC but declined, and instead opted for homeopathic treatment with topical black salve. She reported that her cervical dysplasia was confirmed as resolved by Pap smear after one treatment with black salve; however, she applied another treatment that resulted in significant vaginal pain.\nPelvic examination at presentation revealed normal external genitalia without lesions and normal hair distribution. The vagina was moist and well rugated without lesions or discharge. The vaginal canal appeared shortened, and the cervix appeared pink and flush with the vaginal cuff. The uterus was of normal size, mobile, without tenderness, and with no evidence of adnexal masses or tenderness. She was prescribed oral misoprostol 800 μg for management of missed abortion, and she passed fetal tissue without complication.\nThree months after her initial presentation, the patient returned to the clinic with complaints of oligomenorrhea, with her last menstrual period occurring 38 days prior to this visit. She denied any fevers or pelvic pain. The patient was afebrile, and vital signs were normal. Pelvic examination findings were identical to those from the previous examination. A uterine sound was placed through the external cervical os, and purulent material with a small amount of red blood was passed through the cervix. Human chorionic gonadotropin level was <1.2 mIU/mL. Transabdominal ultrasound revealed no sonographic abnormality of the uterus.\nAerobic cultures were collected at the time of examination. Three days later, the cultures grew Klebsiella pneumoniae sensitive to ciprofloxacin. The patient was treated with ciprofloxacin 500 mg orally twice daily for 10 days and instructed to return if signs or symptoms of pelvic abscess developed.\nThe patient returned to clinic 6 weeks later with continuing oligomenorrhea to discuss a management plan. Hysteroscopy was scheduled 5 days later for evaluation and management of cervical stenosis. On the day of the procedure, visual examination revealed a shortened vaginal canal measuring approximately 5 cm in length and a cervix that appeared flush against the vagina. The cervix was serially dilated to accommodate a 5-mm rigid hysteroscope. Once through the dilated os, the hysteroscope entered a blind pouch measuring approximately 3.5 cm in length, and the true cervix was visible on the right anterior vaginal wall (). Two attempts to advance the hysteroscope through the cervical os failed because of the distorted anatomy. The procedure was terminated after the second attempt, and the hysteroscope was withdrawn without complication.\nThe patient was referred to the urogynecology service to discuss management of her newly diagnosed vaginal stenosis. Vaginoscopy and hysteroscopy were scheduled for 3 months later. Vaginoscopy findings were consistent with the prior attempted hysteroscopy. Hysteroscopy revealed a normal cervix leading to an arcuate uterus with normal-appearing ostia. Postoperatively, the patient was recommended to use a cervical dilator daily for 5 to 10 minutes. Future plans were to perform a vaginal adhesiolysis and to place an indwelling vaginal stent to relieve the stenosis. |
A 40-year-old obese male presented to our Level 1 Trauma Center following multiple gunshot wounds (GSWs) to the left upper and lower extremities. The patient was hemodynamically stable and presented with a Glasgow Coma Scale of 15. On exam, there were 4 GSWs with 2 in the left forearm and 2 in the upper left thigh. The patient had decreased range of motion and paresthesia of the left lower extremity but adequate capillary refill in all extremities. There were palpable pulses of the left femoral, popliteal, dorsalis pedis, and posterior tibial arteries. Initial emergency department imaging demonstrated no fractures in the left upper and lower extremities but shrapnel was appreciated. A CT angiogram of the abdomen with runoff demonstrated a left traumatic arteriovenous fistula involving the left deep femoral artery and left common femoral vein with adjacent bullet fragment (, , ). There was also shrapnel in the soft tissues anterior to the pubic symphysis with associated hematoma and soft tissue inflammation (). Distal vasculature were intact and patent.\nThe patient was taken to the operating room and underwent an exploration of the left groin, repair of the traumatic AV fistula, removal of bullet fragment in the suprapubic region. Intraoperatively, proximal and distal control of the deep femoral artery was achieved and the fistula was identified just distal to the take off. The vein had significant perivascular soft tissue injury with greater than 50% venous wall disruption, a grade IV injury. The vein was ligated with combination of sutures and clips. The arterial defect was debrided to healthy tissue and repaired primarily using interrupted 5–0 polypropylene sutures without stenosis (, ). The left lower extremity was placed in a compression stocking with palpable distal pulses and normal capillary refill.\nThe patient recovered from his injuries with resolution of his paresthesia's and was able to ambulate. He was discharged home on postoperative day 4 on aspirin and a compression stocking. On last follow up 6 months from the initial trauma the patient is asymptomatic and doing well. |
A 47-year-old Asian woman had bilateral uneventful cataract surgery for central cortical opacity. Phacoemulsification via clear corneal incision and implantation of hydrophilic acrylic intraocular lens (IOL) with polymethylmethacrylate modified C-loop haptics were done in the left eye first and the right eye later with interval of 2 days. The left eye was operated first among three operative cases of cataract and the right eye was operated third among four operative cases of cataract. Preoperatively, the cornea was clear with normal specular microscopic finding, and inflammation was not detected in the anterior chamber of both eyes. The patient had not have any systemic or ocular disease other than diabetes that was controlled well under medication, and diabetic retinopathy was not presented in both eyes.\nOn postoperative day 1 of each eyes, right eye had grade 1+ white blood cells in the anterior segment with uncorrected visual acuity (UCVA) of 20/25, while left eye had trace of white blood cells with UCVA of 20/25. Levofloxacin and prednisolone acetate 1% eye drops were prescribed 4 times a day in both eyes. On postoperative day 7 of right eye, anterior inflammation improved and prednisolone acetate 1% was switched to fluorometholone 0.02% in both eyes. On postoperative day 11 of the right eye, the patient visited appealing mild right ocular pain, and the UCVA was dropped to 20/30 presenting diffuse corneal edema. Grade 2+ white blood cells and puff balls were shown in the anterior chamber and inflammatory plaques on the surface of IOL were also found []. However, the cell was not found in the vitreous and it was so clear enough to examine the retina []. Intraocular pressure of right eye was 17 mm Hg. In the left eye, cell reaction was not found in the anterior chamber and the vitreous. Since anterior segment inflammation had been controlled with prednisolone acetate 1% immediately after cataract surgery and the vitreous did not present inflammatory cells, the patient was recommended to use prednisolone acetate 1% every 2 h and continue to use levofloxacin in the right eye. On postoperative day 14 of right eye, cellular reaction decreased to grade 1+ and plaques of IOL surface disappeared partially. Intraocular pressure was 15 mm Hg and UCVA was 20/25 in the right eye. The patient was recommended to use prednisolone acetate 1% every 2-4 h until next visit. On postoperative 1 month of right eye, the white blood cells and inflammatory plaques were not observed, and visual acuity improved to 20/20. On specular microscopic examination, the right eye had lower endothelial cell density than the left eye (2343 cells/mm2 vs. 2758 cells/mm2), higher mean cell area (426 ± 116 μm2 vs. 362 ± 57 μm2), and higher coefficient variation (27 vs. 15), []. The left eye had a normal uncomplicated course during whole treatment period of right eye. |
A 1 year and 4 month-old male baby was admitted in our department with history of delayed developmental milestone, mental retardation, abnormal limb movement, generalized hypotonia and abnormal head movements with nystagmus. There was also history of abnormal breathing pattern with episodes of alternate rapid breathing and normal breathing. There was history of feeding difficulties on and off and frequent chest infection from the early months of life.\nPast history revealed that the child was born by normal vaginal delivery in a hospital at term pregnancy, with history suggestive of birth asphyxia. The birth weight of the baby was 3 kg. His parents had a consanguineous marriage. From the early days of life, his mother noticed feeding difficulties and frequent chest infections of her baby. She also noticed abnormal movement of his head and peculiar eye movement with deviation of the eyes, but relative paucity of movement of the limbs. There was history of episodic rapid breathing lasting for 2-3 min with frequent opening of the mouth and protrusion of the tongue interspersed with normal breathing. There was weak cry and floppiness of all the muscles from the first few months of life. But, no convulsion was noted. Hearing was normal as the child was able to turn the head toward the source of sound. Symptoms were progressively increasing. Prenatal history was uneventful. No similar illness in any siblings in the family was reported. Immunization was incomplete. There was gross delay in development of mental and motor milestones.\nPhysical examination revealed a hypotonic child with constant efforts of abnormal head movement sideways and limb movements, opened mouth with protruded tongue [], and the child showed abnormal breathing pattern with hyperpnea and tachypnea. Head circumference and other anthropometric examinations were normal. No morphological abnormality was detected. Cardiovascular and respiratory system examinations were normal. No organomegaly was present. Ocular examinations revealed bilateral divergent squint, inability to follow moving object, restricted upward gaze of eye and bilateral horizontal gaze-evoked nystagmus. Retinoscopy revealed refractory error of both eyes. Ophthalmoscopic examination was normal and no pigment changes in the retina were detected; optic disc was also normal. Complete blood count, renal function test and liver function test were normal.\nAxial T1- and T2-weighted magnetic resonance imaging (MRI) images showed hypoplastic cerebellar vermis with hypoplasia of the superior cerebellar peduncle resembling the “Molar Tooth Sign” in the mid-brain [Figures and ]. MRI sagital T1-weighted image revealed partial agenesis of cerebellum agenesis and hypoplasia of the superior cerebeller peduncle was noted []. MRI axial (at the level of post-fossa) T2-weighted image revealed hyperintensity of basal cisterns communicating with the fourth ventricle suggestive of vermian agenesis []. Renal ultrasound showed no abnormality. |
A 9-year-old female in mixed dentition was reported with a chief complaint of missing upper front tooth. The patient had a convex profile and asymmetric smile. Intraorally, the patient had a class 1 molar relationship bilaterally with crowding in maxillary and mandibular arch (). Three supernumerary teeth between upper right central incisor and upper left lateral incisor teeth had been diagnosed radiographically and surgically removed 5 months earlier. The maxillary left central incisor was impacted and the adjacent teeth had drifted into the unoccupied space. The impacted tooth was tipped mesially with the crown height at the middle third of the adjacent incisor (). The cause of impaction was established as an obstruction to the path of eruption due to supernumerary teeth.\nThe follow-up radiograph taken 5 months after extraction of supernumerary tooth showed no improvement of the vertical position of the affected incisors, so it was decided to proceed with active orthodontic eruption. A Nance holding appliance and lower lingual arch were placed to maintain space in the arches. The maxillary first premolars and deciduous canines were extracted to relieve crowding and facilitate canine eruption in the maxillary arch. The permanent teeth were bonded in the maxillary arch after the eruption of the canines to align the teeth. An open coil spring was placed post alignment to create space for the impacted incisor. A closed eruption technique was planned as the tooth was apical to the mucogingival junction. Orthodontic vertical traction was applied 1-week post-surgery using an e-chain tied to a modified 0.018 SS wire containing a helix. Over the next 6 months, the progress of incisor eruption was evaluated monthly. Once sufficiently erupted, the crown was bonded with an incisor bracket to align the maxillary arch (). A lower right central incisor was extracted to relieve the crowding in the lower arch. The active treatment took 18 months. At the end of treatment, the upper and lower arches were aligned, there was a class 2 molar relation and class 1 canine relation (). |
The patient was a 66-year-old male with advanced malignant prostate cancer, bone metastases, and kidney failure requiring dialysis. While an inpatient at a cancer hospital, he was transported to the intensive care unit (ICU) for catheter placement and a hemodialysis session. The professional on duty chose a left subclavian vein access, using anatomic landmarks. The blood aspirate at puncture appeared to be venous and the guidewire was advanced without difficulties, but after dilation of the tract and insertion of the catheter, retrograde pulsating flow was observed. Inadvertent positioning in the left subclavian artery (LSA) was confirmed by blood gas analysis and Doppler ultrasound ( ). The examination ruled out the possibility of injuries to the carotid or vertebral vessels, which had normal morphology and blood flow. Physical examination found 4+ brachial and radial pulses. The device was left in place and the patient was transferred to a hospital with vascular and endovascular surgery services. Inherent problems within the Brazilian National Health Service (SUS - Sistema Único de Saúde) delayed the transfer by 18 days. Since there was a risk of fatal complications, the catheter was not removed from the LSA and the patient was not given anticoagulation because of a recent history of melena. After transfer, the catheter was removed, but endovascular repair was not possible because a thrombus was seen in the arterial lumen. There was no bleeding or formation of hematoma, and left upper limb perfusion was maintained, although the brachial pulse was rated 2+ and the distal pulses were absent at that time. The patient was transferred back to the cancer hospital. Doppler vascular echography was conducted again, showing a subacute thrombus in the LSA, where flow was monophasic ( \n ), constituting subocclusion. The arterial thrombosis was in topography distal of the emergence of the vertebral artery, in which flow was laminar, anterograde and with velocities within the limits of normality ( ). At the subclavian-axillary transition, an arterial branch was observed with reversed flow that, based on topography, may have been the dorsal scapular artery ( ). The axillary ( ) and brachial arteries were patent and exhibited slow, low resistance flow, as did the radial and ulnar arteries. The conduct adopted in this case was watching and waiting since, in addition to the contraindication to anticoagulation already mentioned, the patient’s level of morbidity was elevated for an attempt at open revascularization and predictive indicators of the success of a possible bypass were unfavorable: the time elapsed since thrombus formation (22 days), the poor prognosis of the patient’s cancer, and the presence of kidney failure. The patient was observed for a further 2 weeks and did not show any sign of cyanosis, pain at rest, or trophic lesions. He was discharged from hospital for palliative home care. |
A 22-year-old female from a distant Southern Philippines city was presented with a large, painful mass in her right leg. A biopsy 6 years earlier had revealed an intraosseous haemangioma, but the patient had refused amputation and did not seek further consultation.\nThe physical examination showed a large, tender, bony mass over the anteromedial portion of the proximal right leg. The patient retained full motion of the ipsilateral ankle and knee joints. There were no other masses (). The radiographs showed an extensive lesion of the right tibial shaft extending from the proximal epiphyseal line to the distal third of the tibia with an associated huge anterior soft tissue mass. There was a lattice-like pattern with horizontally and vertically oriented striations within the entire lesion (). Magnetic resonance T2-weighted images showed multiple high signal intensity lobules with multiple septations containing several round areas of low signal intensity ( to ).\nA repeat open biopsy confirmed the histopathology to be that of a haemangioma of the bone. During the biopsy, the mass was noted to be extremely vascular. The continuous intense bleeding was controlled only by packing the biopsy cavity with bone cement.\nIn anticipation of increased intraoperative bleeding, the patient was referred for embolisation 2 days before surgery. At surgery, the tumour mass was exposed with an incision beginning medial parapatellar proximally, extending distally to include the previous biopsy site and proceeding distally over the medial tibia just above the ankle joint. After detaching the medial gastrocnemius muscle origin and pes anserinus insertions, the popliteal vessels were identified and protected. Given the long duration of the tumour, the extensive bleeding on biopsy and that the patient resided far away from the treating establishment, thus making regular follow-up monitoring impractical, the agreement reached pre-operatively with the patient was to achieve a marginal resection as far as possible even if it meant a longer segment of bone would be resected distally in order to decrease the chance of a local recurrence. Proximally, however, based on the magnetic resonance images, barely 1 cm of normal tibia remained and resection at this level would have included the tibial tubercle. A cut was therefore made 4 cm distal to the plateau, preserving the tibial tubercle. At this level, the cortex had not been infiltrated; but since tumour had extended beyond this point within the intramedullary canal, curettage and high-speed burring of the medullary contents were undertaken, leaving only the outer cortical shell of the proximal tibial epiphysis. Distally, the tibia was cut 1 cm beyond the intramedullary extent of the tumour, leaving 3 cm of distal tibia (). The entire resected specimen measured 24 cm in length and 12 cm in maximum diameter ().\nIt was decided that this huge tibial defect would be reconstructed with the ipsilateral fibula. The remaining proximal and distal tibia were aligned and fixed with tensioned transfixation wires clamped to an Ilizarov circular frame. Added stability was achieved with olive wires. The fibula was then fixed with three olive wires with the olives on the lateral side of the fibula. On the medial side, Ilizarov components were configured to pull the fibula via olive wires anteromedially. The fibula was percutaneously osteotomised at two levels corresponding to the cuts of the remaining portions of the proximal and distal tibia ( to ).\nGradual medial translation of the fibula at the rate of 1 mm/day was initiated after a week of latency. Bone grafting at the two docking sites of the fibula to the tibia was undertaken a month later. At 6 months, radiographs showed healing at both distal and proximal ends (). The fixator was then removed and a posterior splint applied to protect the leg.\nWeight-bearing with bilateral axillary crutches commenced at 16 months, and fibular hypertrophy was noted on radiographs at 22 months. Radiographs at 38 months showed union at proximal and distal graft–host sites with fibular hypertrophy (). At review, the patient has no pain, has full knee extension and has up to 100° of knee flexion (). There is a 1.5-cm leg length discrepancy, which does not affect walking. And at latest follow-up at 45 months, the patient is fully weight-bearing without any assistive device. There are no signs of local recurrence, and the patient has resumed previous activities with an MTS score of 26, scoring 5 for pain, 4 on function, 4 on emotional, 4 on supports, 4 on walking and lastly, 5 on gait. The final histopathology confirmed the diagnosis of haemangioma. |
An 89-year-old fit female with a history of chronic back pain and an appendectomy during her youth completed using a McBurney incision presented with a one-day history of spontaneous pain in her right flank without any fever, chills, or other symptoms. At the time of her admission, she was not in distress, she was not febrile, and her vital signs were within normal values. On clinical examination, there was swelling with a red area measuring 12 cm × 4 cm and tenderness of the right flank around her appendectomy scar. Crepitus could be felt diffusely on her right and left flanks and the periumbilical and epigastric regions upon palpation. Blood test showed the presence of mild inflammation, with a CRP value of 7 mg/l (within normal values) and an elevated white blood cell count of 18 G/l. The rest of the laboratory results were normal. Emergency ultrasonography was unhelpful because of air interference. An abdominal CT scan () showed diffuse subcutaneous abdominal emphysema extending to the pelvis on the left side that was more pronounced on the right inguinal fossa with a bowel loop in contact with the abdominal wall. An emergency laparotomy centered on the McBurney incision showed feces and pus within the subcutaneous compartment. Furthermore, at the level of the aponeurosis of the external oblique muscle, an inflammatory diverticulum could be seen fistulizing between the lumen of the sigmoid colon loop and the necrotic subcutaneous tissue. We subsequently diagnosed intraoperatively a subcutaneous abscess and emphysema with an enteroparietal fistula caused by a ruptured sigmoid diverticulum in an incisional hernia. The necrotic tissues were excised, and the punctiform sigmoid colon fistula was closed. Revision of the rest of the sigmoid showed important adhesions between the sigmoid colon and the parietal peritoneum of the right flank and between the caecum and the sigmoid colon, respectively. The sigmoid colon also showed diffused diverticulosis with no inflammation. The cutaneous and subcutaneous tissues were left open and dressed with a negative pressure-assisted closure device on postoperative day 1. The patient received intravenous antibiotherapy for two weeks with quinolones and a third-generation cephalosporin at first which was then switched to aztreonam due to an allergic reaction. Bacteriological studies showed polymicrobial digestive bacteria (i.e., Escherichia coli, Streptococcus equinus, and Enterococcus). Subsequently, there was good clinical and biological evolution. At two weeks postoperation, she was reoperated on for closure of the wound. She was discharged from the hospital three weeks after her initial surgical intervention with the indication to continue antibiotics for a total of four weeks. |
The authors report a case of a 29-year-old Saudi woman who was G4T2P0A1L2 at 21 weeks of gestation. She was free from medical illness and she had had no previous surgical procedures. She is a housewife; she never smoked tobacco or drank alcohol, and she had no history of recent travel to endemic or pandemic areas. She was referred based on an antenatal ultrasound finding that showed multiple fetal anomalies. This ultrasound had been conducted at another hospital for evaluation and management. Her past obstetrical history was uneventful with two normal term vaginal deliveries and a history of first trimester unexplained miscarriages. She is married to a first-degree cousin working in a governmental institute; there is no history of genetic or congenital anomaly in either of their families.\nHer current pregnancy was spontaneous with no history of illicit drug use or exposure to infection or radiation. Her initial early antenatal scan diagnosis showed suspicion of possible fetal diaphragmatic hernia and required further validation which was not possible at the maternal–fetal medicine (MFM) unit at the hospital which also did not have available sonographic specialists. During her first antenatal visit at 21 weeks + 0 day of gestation, the results of her anatomy scan revealed a single viable fetus with estimated fetal weight (EFW) on 50th percentile with normal biometry measurements.\nFurther detailed anatomy scan findings revealed a male fetus with both kidneys appearing small in size, hyperechoic dysplastic, both ureters were dilated, urinary bladder looked abnormal in shape with thickened bladder wall, and umbilical cord at fetal insertion side appeared thickened. In addition, the diaphragm was seen clearly separating the chest from the abdominal compartments with no evidence of diaphragmatic hernia. Both feet were clubbed and open hands were seen with no other anomalies or any soft marker seen (see Fig. .) Based on the multiple fetal structural anomalies discovered, the couple was counseled about the scan findings and advised for further workup, such as: perinatal invasive testing; toxoplasmosis, other (syphilis, varicella-zoster, parvovirus B19), rubella, cytomegalovirus, and herpes (TORCH) screening; and fetal echocardiogram to exclude syndromic or chromosomal causes. This would support reaching a better diagnosis and allow for further discussion on the options available such as the continuity of the pregnancy or termination based on the severity of the fetal condition (see Table ).\nOur patient had some social issues and was also following her condition in another institute and only revisited our center at 32 weeks and 4 days of gestation. At our center another follow-up scan revealed a single viable fetus, cephalic in presentation, anhydramnios with normal head and femoral length biometry. Unfortunately, the abdominal circumference (AC) was not taken due to the extremely distended abdominal wall that prevented any further visualization by ultrasound. The right kidney measured 3.4 × 1.1 cm with a small cyst, the left kidney measured 2.9 × 1.3 cm with bilateral hugely dilated ureter and urinary bladder (mega cyst) (see Fig. ).\nTORCH screen test results were non-reactive. Amniocentesis was performed and showed normal chromosomal results. A fetal echocardiogram allowed for limited examination due to anhydramnios; however, no obvious cardiac anomalies were noted. Lungs appeared compressed due to severely distended abdomen from the progressively enlarged urinary system, otherwise no other abnormal findings noted. The couple was counseled by the MFM team about the worsening condition from the recent scan findings and were told about the poor fetal prognosis and the high mortality rate, secondary to severe lung compression with the presence of anhydramnios which would lead to lung hypoplasia and cause fetal demise.\nIt was explained that the entire urinary system was affected with severe dilatation causing severe abdominal wall dilatation and for this reason measuring fetal AC had been difficult antenatally. Options were discussed with the couple:Termination of pregnancy to avoid obstetrical complication during labor which is fetal abdominal dystocia as it was difficult to measure the abdominal wall antenatally with the severe progressive renal system dilation with advancing gestational age versus To wait until term pregnancy while knowing the poor fetal prognosis\nFurthermore, antenatal interventions were offered to the couple including tapping of the fetal bladder and ureters prior to induction of labor and to then send the amniotic fluid sample for further genetic testing. Our patient’s case was initially discussed by a multidisciplinary team which included a perinatologist and a neonatologist before finally making a combined agreement and alignment with the couple who decided to terminate the pregnancy; a caesarian section would be preserved for maternal indication and comfort care post-delivery to born infant were also explained.\nAt 32 weeks and 5 days of gestation, tapping of the fetal bladder and ureter was performed and samples of amniotic fluid were sent for whole exome sequencing (WES) test; however, unfortunately, after waiting a few weeks for the results, no results could be determined due to a laboratory error.\nOur patient underwent induction of labor to terminate the pregnancy and delivered vaginally a male neonate with Apgar score of 2 in 1 minute and 5 in 5 minutes, weighing 1800 grams without any complications. The vital signs revealed blood pressure of 90/60, pulse 100 beats /minute, and temperature of 36 °C. Clinical examination of the newborn revealed distended abdomen and thin wrinkled skin, retracted chest, cryptorchidism, and clubbed feet; no facial anomalies were noted and the features were most likely to be suggestive of PBS (see Fig. ). The newborn died 2 hours post-delivery.\nThe placenta was sent for a histopathology examination as a part of the workup and the result revealed normal findings.\nA postmortem examination was not offered to the couple since this is not conducted in the center. The couple was counseled prior to discharge regarding future pregnancy plans, despite low reoccurrence. It was also highlighted to them the importance of having early prenatal testing in a center in which there were well-trained sonographers and a high risk in pregnancy unit available. They were also informed about the lack of result of WES test due to laboratory error and they were fine. |
A 9-year-old boy was brought to the emergency department of our hospital because of a 5-day history of fever, vomiting, and right hip pain localized to the anterior and medial aspect of his right hip. He was examined and received a diagnosis of flu and viral gastroenteritis. His right hip pain was attributed to sportive activities without trauma, and the patient was discharged with symptomatic treatment. The family returned to the emergency department twice more in the following 3 days because of his worsening symptoms. Four days after the onset of the fever, a reduction in the range of motion of the child's right elbow appeared, in addition to vomiting and limping.\nUpon admittance to our hospital, the child was febrile, and could only manage to walk a few steps without supports, with an important antalgic gait. The spontaneous rest position of his hip was in slight flexion with external rotation. His hip range of motion was restricted in all directions, but predominantly in internal rotation. In addition, the patient presented pain and swelling of his right elbow, with a restriction in motion of the joint.\nThe patient's laboratory results showed a white blood cell count of 17,100 cells/mm3 with 86% neutrophils, C-reactive protein to 200 mg/L, and erythrocyte sedimentation rate 65 mm/h. A conventional radiograph of the hip and the elbow showed no significant abnormality and a diagnosis of hip and elbow septic arthritis was therefore suspected, and the child underwent aspiration of both the cited joints. Aspiration of the elbow returned 7 mL of pus, and thus drainage and irrigation of the joint were performed using a lateral approach. Conversely, no effusion was found in the hip aspiration. The patient was immediately started on empiric intravenous antibiotics (clavulanic acid/amoxicillin). The following day, an MRI of the hip was performed in order to explain the pain and range of motion restriction of the right hip, which were unusual since the hip aspiration was normal. The MRI revealed an osteomyelitis of the ischio-pubic area associated with a subperiosteal abscess (Fig. ). A 0.6 × 3 cm low attenuation mass with an enhancing rim within the right obturator externus muscle was also identified (Fig. ). Scintigraphy was performed to eliminate other septic localizations.\nStreptococcus pyogenes (group A) was cultivated from the joint fluid of the elbow, whereas the blood and urine cultures remained negative. Three days after the elbow arthrotomy, a surgical treatment was performed on the patient's right hip in order to evacuate the subperiosteal abscess and muscular collection because of the persistence of the patient's symptoms and inflammatory syndrome despite effective intravenous antibiotics. An incision and drainage was performed through the medial approach. This revealed bloodstained pus of the ischiopubic area. The right elbow was also examined again during this operation. Clavulanic acid/amoxycillin therapy was continued intravenously for a further 2 weeks, then the boy was switched to an oral treatment for 4 additional weeks. The patient showed steady improvement, his temperature returned to normal within 24 hours, along with a gradual improvement of his hip and elbow pain. All the patient's biological parameters returned to normal within a week. |
A 30-year-old woman visited the hospital due to right lower quadrant pain over the period of 1 week. A laparoscopic myomectomy was performed 4 years ago because of a broad ligament leiomyoma, which was about 10 cm in size. Laboratory findings included a routine blood examination, and a C-Reactive Protein (CRP) test, with tumor markers all found to be within normal ranges. The pelvic Magnetic Resonance Imaging (MRI) scan showed some nodules at the posterior margin of the bladder that were considered to be endometriosis, and some pelvic effusion that was significant on the right side. It was also noted that there was a mass shadow at the lower right ureter (medial to the iliopsoas muscle), with distention of the lower right ureter (Fig. ). The patient also underwent computed tomography (CT) scans to enable the practitioners to observe the size of the abdominal mass and its surroundings. The CT images revealed a region occupying the middle right ureter that was considered to be a retroperitoneal aggressive fibroma, which led to severe hydronephrosis on the right kidney and upper ureter, and a right pelvic effusion (Fig. ). After more detailed examinations were conducted, there were no obvious abnormalities detected in the brain, heart, liver, gallbladder, spleen, pancreas or blood. The color Doppler ultrasound demonstrated that there was a hypoechoic mass next to the right iliac vessels that was closely related to the adjacent ureter. This resulted in severe hydronephrosis of the right kidney and a right upper ureteral dilatation (Fig. ). Ndzengue et al. [] reported a case of a pelvic desmoid tumor simulating a uterine leiomyoma recurrence. The patient that presented at our hospital had a history of uterine leiomyoma. We subsequently organized a multidisciplinary consultation to determine the next stage of her treatment plan. According to the patient’s surgical history, the next step would be determined after reviewing the results of the last surgical pathological wax, because the pathological nature of the retroperitoneal mass was uncertain.\nConsequently, a laparoscopic pelvic tumor resection with assistance from a Da Vinci robot was successfully conducted. A local peritoneal protuberance was observed in the right iliac vascular area. The parietal peritoneum was incised above the mass and it was carefully freed along the edge of the mass. The size of the tumor was about 6.0 cm × 5.0 cm × 3.0 cm. It had invasively grown and it was wrapped around the right ureter and the right ovarian arteries and veins. It was stuck to the psoas muscle and the iliac vessels. According to preoperative preparations and intraoperative conditions, a decision was made to cut off the right ureter, the right ovarian arteries and veins, and completely remove the tumor and the two masses that were located in front of the broad ligament on the right hand side of the uterus. The sizes of the masses were approximately 1.5 cm × 1.0 cm × 1.0 cm. The right ureter was anastomosed and put into a double J tube. A pathological diagnosis of an intraoperative frozen sample determined that it was a spindle cell soft tissue tumor, and the two masses were leiomyomas. Postoperative pathology tests of the pelvic mass determined that it was aggressive fibromatosis that had invaded the ureteral wall (Fig. ). The uterine surface nodules were also leiomyomas. Microscopically, the tumor cells were arranged sparsely in a spindle shape with blood vessels of different sizes found in the interstitial tissue. Immunohistochemical findings were found to be partially positive for smooth muscle actin (SMA) and desmin, and less than 5% ki-67 of positive cells were seen in the lesion. A detailed re-examination was performed 3 months after the initial surgery, to determine the structure and function of the ovaries. The transvaginal color Doppler ultrasonography was able to determine that the ovaries were normal in size with several follicular echoes. The blood supply to the right ovary was good. There were no obvious abnormalities in the uterus or pelvic cavity (Fig. ). The pelvic MRI and the CT scan of the whole abdomen determined that there were no abnormal lesions in the pelvis. Simultaneously, the endocrine function of the patient’s ovaries was found to be normal, and she was able to self-maturate after removing the double J tube. |
In September 2010 a 78 years old man was diagnosed with an adenocarcinoma of the right-sided colon. Initial staging by abdominal and pulmonary CT revealed a lesion in the right-sided colon penetrating the visceral peritoneum with infiltration of the abdominal wall (cT4b) and a single metastatic lesion in the liver, in segment VII, with a diameter of 1.5 cm (M1a) according to a stage IVA disease.\nAfter right-sided hemicolectomy together with a simultaneous atypical resection of the single liver metastasis adjuvant chemotherapy (CTX) with capecitabine and oxaliplatin was started. After 5 cycles of adjuvant CTX the patient complained about a considerable increase of his abdominal girth. An abdominal CT in March 2011 yielded peritoneal nodules and ascites (Figure ).\nAfter confirmation of a mutated K-RAS gene a palliative first line CTX with capecitabine and irinotecan in combination with bevacizumab was started. However, treatment was interrupted right after the first cycle because of an episode with neutropenic fever and resumption of CTX was refused by the patient until June 2011.\nCT imaging in June 2011 showed a disease progression with a new solitary pulmonary lesion with a diameter of 3.3 × 2.8 cm (Figure a) and ascites in all 4 quadrants of the abdomen (Figure b). As the patient refused biopsy of the pulmonary lesion and the radiology report stated that the lung lesion was radiological consistent with a pulmonary metastasis a biopsy was not performed. Tumor markers CEA and CA 19.9 were within normal range at the time of diagnosis and during the whole course of disease. Clinically, the amount of ascites was increasing and required abdominal punctures on a weekly basis. Therefore the first line CTX with capecitabine, irinotecan and bevacizumab was reinitiated. Despite administration of granulocyte colony stimulation factor another episode of neutropenic fever occurred and CTX was discontinued after a total of 3 cycles in August 2011. As the patient refused to receive any further CTX and because of ascites being the predominant clinical symptom an immunotherapy with i.p. catumaxomab was started in December 2011 (4 consecutive i.p. infusions of catumaxomab on days 0, 3, 7 and 10 at increasing doses of 10, 20, 50 and 150 μg). Treatment was well tolerated without any side effects. Surveillance by CT scan in April, July and October 2012 showed a partial response of the pulmonary lesion and no paracentesis was necessary until January 2013 (Figure a and b). While the pulmonary lesion showed a further regression in size and could finally only be detected as a scar (Figure a), a CT scan in January 2013 showed a progressive peritoneal disease with ascites and a consecutive hydronephrosis III° of the right kidney (Figure b). As the patient still refused any further CTX we decided to restart i.p. treatment with catumaxomab in January 2013. |
A 53-year-old male patient presented with symptoms of progressive left lower extremity myoclonus and muscle rigidity that started 3 days ago. The left lower extremity myoclonus and stiffness started suddenly without any apparent cause with a frequency of once every few minutes. They significantly worsened on the second day with an increase in frequency of once every few seconds and persisted even during sleep. After admission and on the fourth day, similar symptoms started to appear on the patient's right lower limb. Those symptoms were accompanied by a severe joint stiffness and the inability to straighten it, to stand or walk. The Patient denied any prior history of chronic conditions such as diabetes mellitus, Grave disease or similar symptoms among his family members. He also denied any prior surgery.\nThe physical examination indicated a bilateral muscle twitching of lower extremities, an elevated muscle tone, a positive knee reflex (++++) and positive bilateral Babinski sign. The electromyography results displayed signs of motor unit discharge. Meanwhile, the brain, cervical, thoracic, and lumbar spine MRI scans were all negative. Paraneoplastic antibodies, GAD-65, GAD-67 antibody, and glycine receptor antibody were all tested and reported negative. Additionally, the chest CT and PET-CT scans were also negative.\nThe patient was given an intravenous infusion of gamma globulin 0.4 mg/kg with additional symptomatic treatments such as baclofen and clonazepam on the fourth day after admission. No obvious adverse reaction was observed after the initial treatment. He showed signs of remission after 5 days of treatment with total disappearance of his muscle twitching. However, no significant changes in muscle stiffness were seen after the initial treatment and the patient was still unable to walk or stand.\nTwo months after the onset of the condition and after a thorough discussion with the patient, he received injections of botulinum toxin A in both lower limbs partially alleviation his muscle stiffness. Additionally, he was able to stand and walk but with a spastic gait.\nInformed consent was obtained from the patient for the description, data utilization, and publication of this report. |
An 82-year-old woman with hypertension and atrial fibrillation on warfarin presented with 2 h of acute onset epigastric pain with radiation to the chest, jaw, and left arm. Initial vitals were within normal limits. She was in moderate distress. Labs were significant for a subtherapeutic international normalized ratio (INR). Electrocardiogram revealed ST segment elevations in the inferolateral leads. After Allen’s test was done, she was deemed an appropriate candidate for approach via right radial artery and taken for urgent cardiac catheterization. The patient was found to have an acute thrombus in the distal posterior descending artery (PDA) and posterior left ventricular (PLV) branch. She was treated with angioplasty and aspiration thrombectomy, and the acute thrombi were thought to be secondary to thromboembolic phenomenon. The patient received dual antiplatelet therapy following the procedure, and a pneumatic band was placed over the patient’s right wrist and remained in place until it was removed per protocol. The following day, patient reported pain at the right wrist. On exam, a pulsatile 2.4-cm mass with an overlying bruit was noted. A color flow Doppler ultrasound study was performed and it revealed a radial artery pseudoaneurysm (). Initial discussions with vascular surgery, and interventional radiology did not yield a consensus approach towards resolving this complication. Nonspecific compression via pneumatic band was attempted for 18 h; however this was initially proved unsuccessful as seen on repeat Doppler. Following this, a different compressive approach was attempted. Pneumatic band was inflated until radial pulse flow was occluded as evidenced by absence of pulse oximetry wave form. The level of occlusion was achieved with 12 cc of air inflation. Air was removed in small segments until radial artery flow was noted (this was noted to be at 10 cc of air inflation of the pneumatic band). Upon dialogue with nursing staff, it was discussed that the pneumatic band should remain in place at current level of compression for 1 h and then 1 cc of air should be removed each hour until device was fully deflated (). After the allotted time course of 10 h that it took to deflate the pneumatic band, a repeat arterial Doppler ultrasound revealed complete resolution of the radial pseudoaneurysm (). She also reported improvement of pain, and was discharged that day. |
A 26 year-old man attended our ophthalmic ward in April 2017 with intermittent bleeding of the right eye, from which there was also strong odor. The patient was a heavy smoker but had no other underlying conditions. He had no history of drug-use. From his medical history it was noted that the patient had undergone a right ophthalmectomy 24 years previously due to retinoblastoma, and implantation of an artificial right eyeball in 2014 (timeline shown in Additional file ).\nOn admission, his pulse rate was between 80 and 100 beats/min. His body temperature and respiratory rate were both normal. Physical examination showed narrow conjunctival sac in right eye and the exposure of ocular prosthesis, which was discharging a yellow-green secretion along with a strong odor. The visual acuity of left eye was 0.3, and the intraocular pressure was 15 mmHg. All other characteristics of the left eye were normal. A auscultation did not show any abnormality in the lungs, and no signs of carotid murmur were found. Interestingly, laboratory investigations did not reveal abnormal inflammatory markers such as leukocytosis or any increase in neutrophils or C-reaction protein. According to clinical and laboratory investigations, infectious endocarditis was not suspected. The patient had no history of other immunosuppressive conditions, except smoking and a retinoblastoma 24 years previously. The patient did not report any direct contact with animals; however, he did work in a clothing factory so would have been contact with wool and cowhide for one month of the year. Three months had elapsed between the patient last coming into contact with wool and cowhide and the appearance of clinical symptoms. Considering the results of these investigations, partial artificial eye infection, especially anaerobic organism infection, was suspected.\nImaging workups were completed, which included chest x-ray, transthoracic echocardiography and eye magnetic resonance imaging. As shown in Fig. , eye magnetic resonance imaging revealed that the tissue surrounding the right eye prosthesis as well as the soft tissue of the lacrimal gland area were swollen, whereas the left eye appeared normal. Inflammatory disease in the right eye was therefore suspected. According to chest x-ray and transthoracic echocardiography, no obvious abnormalities in the lungs or heart were observed.\nBefore surgery, a few specimens of the right eye secretions were collected to be cultured, but no bacteria were isolated, possibly because most of the secretions had been absorbed by the artifical eye making it yellow-green in appearance. After removal of the right artificial eye with debridement (5 days after admission), both the artificial eye and specimens of the eye secretions were sent for bacterial culture under aerobic and anaerobic conditions. No bacterial growth was detected from the ophthalmic secretions, but cultures were obtained from the artificial eye. Sparse growth of β-hemolytic cocci and heavy growth of small, non-hemolytic, translucent colonies were observed on Columbia agar plates supplemented with 5% sheep blood (BioMérieux, Marcy l’Etoile, France) under aerobic conditions after 48 h. And the latter colonies only grow close to the hemolysis zone of the former one. Under anaerobic conditions, only the small, translucent colonies were detected from the artificial eye (as shown in Fig. ). Of the two colony types, the β-hemolytic cocci were confirmed as Staphylococcus aureus, whereas the small, translucent colonies stained positive in a Gram stain and occurred singly, in pairs, or in short chains (Fig. ). Catalase and oxidase reactions of the unknown colonies were negative and phenotypic characterization using the Vitek2 GP system (BioMérieux) was inconclusive. However, Matrix-assisted laser desorption/ionization time of flight (MALDI-TOF) mass spectrometry revealed a match with Helcococcus ovis DSM 21504 T DSM (log score: 1.637) according to the Brucker Maldi-Biotyper database. Identification of this organism was confirmed by 16S rRNA gene sequencing. BLAST analysis of the partial 16S rRNA gene sequence derived from our isolate (1492 nucleotides, deposited in the GeneBank database under accession number MG188744) showed 98.9% identity (15 nucleotide differences) with the 16S rRNA gene sequence of H. ovis s840–96-2 deposited in the GenBank database under accession number NR027228 by Collins and coworkers [] in 1999 when this species was first described.\nAntimicrobial susceptibility testing (AST) of both strains was performed. The disk diffusion method was carried out and with the exception of penicillin, erythromycin and clindamycin, S. aureus isolated from this case was susceptible to all other drugs including cephalosporins and fluoroquinolones. AST for Helcococcus was performed using the CLSI broth microdilution method on Mueller-Hinton II broth (BD Diagnostics, Heidelberg, Germany) supplemented with 3% (vol/vol) lysed horse blood (Oxoid, Wesel, Germany) and 0.001% (wt/vol) pyridoxal HCl (Sigma–Aldrich, Munich, Germany) incubated at 37 °C in 5% CO2 for 24 h [, ]. Streptococcus pneumoniae ATCC 49619 served as a quality control. Then, we changed the method and performed an E-test on blood agar, with S. aureus ATCC 29213 as the quality control (for reference only). The MICs (μg/L) of the drugs for this strain are reported in Table . Since no antimicrobial testing guidelines are currently available from the Clinical and Laboratory Standards Institute (CLSI) for Helcococcus, the MICs were determined in reference to the CLSI guidelines for S. aureus []. According to the CLSI [, ], this strain was susceptible to penicillin, ampicillin, teicoplanin, ceftriaxone, vancomycin, and linezolid.\nAfter admission, the patient received levofloxacin eye drops 4 times per day until being discharged from hospital. After surgery, the patient was initially treated with intra-venous cefotaxime (2.25 g/250 ml NaCl, 1/day) and ornidazole (500 mg/day) for 1 week. Two weeks after admission, the patient recovered and was discharged from hospital. Six months later, the patient returned to the hospital to finish implantation of the artificial right eyeball and no signs of infection were detected. With the patient’s consent, we collected samples from the skin around both eyes for aerobic and anaerobic culture, but only normal skin flora were detected, such as coagulase-negative Staphylococcus. After surgery, the patient was in good health and was discharged from the hospital. |
A 73-year-old Hispanic male with a history of coronary artery disease, hypothyroidism, and arthritis underwent a CT scan of the abdomen and pelvis for an unrelated complaint and was incidentally found to have a hypervascular left renal mass suspicious for malignancy. He was referred to an outpatient percutaneous transcatheter renal artery embolization, in preparation for a subsequent radical left nephrectomy with a view to decrease intraoperative bleeding at surgery. Physical examination prior to the procedure was essentially normal and did not reveal any cardiopulmonary abnormalities. The patient received intravenous conscious sedation over a period of 60 minutes consisting of 100 mcg fentanyl and 1.0 mg midazolam, administered in intermittent doses, while undergoing continuous physiologic monitoring. Per protocol [], the procedure was performed using a total of 40 mL of pure ethanol divided into three doses and administered in slow pushes at 15-minute intervals. This dose was derived based on a weight of roughly 80 kg, using the lower end of the recommended dose of 0.5–1 mL/kg. The result was a significant reduction in tumor vascularity, with approximately 10% residual renal artery tumor perfusion.\nAfter the third ethanol injection, the patient developed cough, agitation, and acute respiratory distress with hypoxemia (SpO2 of 80%; normal >92%). He required sedation and emergent intubation for airway protection. Given the concern for pulmonary embolism, pulmonary angiography was immediately performed and showed patent pulmonary arteries, without central or segmental pulmonary artery embolus or vessel occlusion. The procedure confirmed the presence of PAH with a mean pulmonary artery pressure (mPAP) of 70 mmHg (normal <25 mmHg). Acute pulmonary vasospasm due to venous shunting from the systemic intra-arterial ethanol administration was suspected. A Fogarty balloon was inflated in the renal artery to prevent further dissemination of ethanol into the systemic circulation. Nitroglycerin was administered directly into the pulmonary artery circulation at a rate of 5 mcg/min with a total dose of 1400 mcg; this resulted in reduction of mPAP from 70 mmHg to 30 mmHg. A pulmonary artery catheter was left in place for continued pulmonary artery pressure monitoring.\nThe patient was admitted to the intensive care unit for close monitoring. He was started on intravenous dexmedetomidine for sedation and analgesia. On physical examination, his vital signs were within normal limits, with no focal findings. Laboratory workup revealed mild leukocytosis (white blood cell count 11.4 k/mm cu; normal 4–11 k/mm cu), anemia (hemoglobin 8.3 g/dL; normal 13.5–17 g/dL), and elevated blood alcohol level (80 mg/dL; normal 0–20 mg/dL). There was no evidence of hemolysis, as the hemoglobin level was close to baseline and the bilirubin was normal. Electrocardiogram and cardiac enzymes were negative for acute ischemia; chest X-ray (CXR) did not show any pleural effusion, consolidation, or acute pathology; transthoracic echocardiogram (TTE) showed normal cardiac function; lower extremity ultrasound Doppler ruled out deep venous thrombosis (DVT), and recent pulmonary functions tests (PFT) showed no abnormalities in baseline respiratory function. Arterial blood gases were within normal limits and the patient was successfully weaned and extubated the following day. Pulmonary artery pressure throughout his stay stabilized at a mean of 30 mmHg before the pulmonary artery catheter was discontinued. His respiratory status remained stable and he was gradually weaned off supplemental oxygen altogether. There was no evidence of new cardiopulmonary abnormalities at the time of discharge. |
A 72-year-old Asian woman had been admitted to our department 28 days previously for treatment of an intracerebral hemorrhage (ICH). During her stay, the patient complained of an unpleasant sensation in her whole body that had been present at night since admission. When the patient was at rest, discomfort and irritation occurred in both calves. All symptoms were resolved by moving the legs. The symptoms mainly occurred in the evening, around dinner time (about 6 pm) and reached a peak during sleep. As a result, the patient could not easily fall asleep, and often woke up during the night. In addition, the patient had to move her legs 7 to 8 times a night because of the discomfort in both calves during sleep. A detailed interview revealed that unpleasant sensations when resting or sleeping had occurred since the patient was an adolescent. However, the patient reported that both her mother and sister had the same symptoms and she had concluded it was a familial characteristic. Therefore, the discomfort in the lower extremities had never been assessed, and had she had never received any treatments or medications to manage it. The patient stated that she had to move throughout the night, and that when someone else was nearby, she refrained from moving, making the discomfort worse. Therefore, she slept alone, even after she was married.\nWe diagnosed the patient with chronic persistent RLS based on the 2012 Revised International Restless Legs Syndrome Study Group Diagnostic Criteria.[ Blood tests revealed no iron deficiency or renal function abnormality. HbA1c was also confirmed to be within the normal range, and diabetes mellitus was excluded. In addition, secondary RLS was excluded because there was no history of peripheral neuropathy, Parkinson disease, or the use of medications known to block the action of dopamine. At the first visit, the Korean version of the international restless legs scale (K-IRLS) was used to evaluate the degree of subjective symptoms,[ which were found to be the most severe, with a total score of 35 points. To evaluate the effects of RLS on sleep disturbance, the insomnia severity index (ISI) was used,[ which showed a severe state of sleep disturbance with a total score of 27 points (Figs. and ).\nThe primary purpose of admission was to treat left-sided weakness caused by the ICH. However, given the persistence of RLS-related symptoms and subsequent decrease in daytime activity, the patient had difficulty participating in a scheduled rehabilitation treatment program. Therefore, to treat the left-sided weakness, the patient began a rehabilitation program and acupuncture therapy (twice a day), while herbal medicinal treatment was planned to control the symptoms of RLS. The patient had a low weight (159 cm/49 kg), an anorexic tendency, and a pale complexion, with flushing occurring in the afternoon. She had a floating and rapid pulse and a pale red tongue. We therefore determined the patient to have Xue deficiency and Qi stagnation using the identification pattern of traditional East Asian medicine, considering the symptoms of RLS and the aforementioned findings. Among the herbal complexes containing Paeoniae Radix, Dangguijakyak-san (DS, 6 g/d, Kracie; Tokishakuyakusan in Japanese, Danggui Shaoyao San in Chinese) and Shihogyeji-tang (ST, 6 g/d, Kracie; Saikokeishito in Japanese, Chaihu Guizhi Tang in Chinese), which can be used for Xue deficiency and Qi stagnation, were administered to control the symptoms of RLS (Table ). During the treatment period, other medications the patient was taking were continued, though some were adjusted as needed.\nDuring treatment, changes in the patient's symptoms and sleep status were assessed daily. The K-IRLS and ISI, which were used to evaluate the severity of RLS on day 1, were used twice more to quantitatively evaluate the changes in symptoms over time (days 21 and 47).\nOn day 1, the unpleasant sensations were present on the bilateral calves, thighs, ankles, and wrists and the total duration of sleep was 2 hours. One week after the start of DS and ST, the symptoms of RLS began to improve. After 3 weeks (day 21), a significant improvement in symptoms was observed, with the duration of sleep lasting 9 hours. In addition, on day 21, the patient reported that she awakened 2 to 4 times during the night, but that after a brief massage of her legs, she fell back to sleep immediately. On day 36, ST was discontinued given the continued improvement of symptoms. On day 47, symptoms had almost completely disappeared, with no awakening at night, and a duration of sleep averaging 10 hours or more. After day 47 (day 94), DS was also discontinued since there were no further symptoms. According to the K-IRLS score, the symptoms were rated as 11 points on day 21 (moderate) and 0 on day 47 (no symptoms). The ISI results also improved, with 9 points on day 21 and 0 points on day 47. Additionally, the sleep disturbances caused by RLS had also completely resolved. There were no adverse effects associated with DS or ST during the treatment period (Figs. and ).\nThe patient's ICH treatment continued until day 86, at which time the patient was discharged. There was no recurrence of RLS by discharge, and approximately 6 months after discharge (day 244), telephone counseling revealed symptoms had still not recurred (Figs. and ).\nOn the last day of treatment, the patient signed informed consent for publication of the case report, which was approved by the Institutional Review Board of Kyung Hee University Korean Medicine Hospital (KOMCIRB 2020-05-001-001).\nThe following is a qualitative description of the experience of the patient and her caregivers during the course of treatment.\nDay 1, patient: “If someone is lying next to me, I cannot sleep. I have to move from time to time, but it is hard because I cannot. So, I have always slept alone.” “It has been harder since the stroke. I guess I cannot move freely because of left-sided weakness.” Her husband: “My wife has too much sensitivity. In the evening, the sensitivity worsens, and she hates to have someone next to her. But all of her family members are like that.”\nOn day 7, patient: “For the first time, I felt the discomfort at night diminishing.”\nOn day 21, patient: “Previously, there was discomfort in the calf, thigh, ankle, and wrist, but now only in the calf and thigh.”\nOn day 47, patient: “I can sleep well without waking up these days.”\nOn day 86, patient: “I think I can sleep no matter who is next to me.” Her husband: “I’ve always thought of my wife as an unusually sensitive person, but I never thought it was a disease.” |
We describe the case of an 11-year-old right-hand-dominant White Portuguese girl who presented with pain in her right elbow following a fall onto the outstretched hand that occurred while she was riding a bicycle. She was unable to move her elbow actively due to pain. A physical examination revealed mild swelling and tenderness over the lateral aspect of the distal part of her humerus. The child had a restricted range of motion of her elbow, especially with flexion. Joint stability was considered normal and no neurovascular signs were observed. The diagnosis was made on a lateral and an oblique radiograph (Figures and ), which revealed a displaced fracture of the capitellum of her right elbow, a typical Hahn-Steinthal or Type I fracture. An anteroposterior radiograph of the distal part of her right humerus did not reveal a definite fracture. No other injuries were detected.\nOur patient was treated by open reduction and internal fixation. Under general anesthesia, an attempt was made at closed reduction but complete reduction could not be achieved that way. With a pneumatic tourniquet in place, a posterolateral incision was made (Figure ). Further manipulation of the fragment allowed reduction that was held in the correct position with two temporary Kirschner wires. The final fixation was performed with two 3.0 mm cannulated screws. The screws were inserted in a posteroanterior direction through the posterior surface of the lateral condyle. An intraoperative dynamic examination showed satisfactory stability of the osteosynthesis and anatomic articular congruity. A protrusion of the tip of the screw on the articular surface was carefully avoided (Figure ). Postoperatively, her elbow was immobilized in a long arm cast at a right angle, with the forearm in a neutral position for three weeks. The postoperative outcome was uneventful, allowing our patient to leave the hospital 48 hours after surgery.\nThe follow-up period was of one year with regular clinical examination and roentgenographic control. Our patient was asked about pain and daily activities. Her elbow flexion-extension and pronation-supination were evaluated and compared with the contralateral side.\nOur patient was able to perform normal elbow movements and normal daily activities without pain, but she mentioned having limited mobility after removal of the plaster cast. This initial period was followed by four months of a progressive mobilization program that was guided by a physiotherapist. On the basis of the radiographic appearance, the fracture presented convincing signs of consolidation after five weeks. At the fourth month of follow-up, the fracture was considered completely healed and our patient had a good range of motion with full flexion, pronation and supination, but lacked 15° of extension in comparison with her contralateral side (Figures , and ). There was no varus or valgus angulation. At one year of follow-up, our patient had no pain and her elbow had a restored full range of motion. Furthermore, a radiographic examination revealed no signs of avascular necrosis, physeal arrest or angular deformity (Figures and ). |
A 79-year-old woman with bilateral lower extremity weakness due to cervical myelopathy presented at our department in 2002 after multiple reconstructive procedures in both hips for developmental dysplasia of the hip. In 1993, a bulk allograft in combination with an acetabular cage and a cemented cup were used to treat the left massive acetabular bone loss. The defect was type IVb by the classification of the American Academy of Orthopaedic Surgeons () and . In 2000, the acetabular construct failed mechanically while the existing cemented femoral stem remained well fixed (Figure ). Removal of the acetabular hardware was followed by implantation of a whole acetabular allograft. The allograft was stabilized with plates and screws, and a new cemented cup was inserted. 2 years later, allograft fracture and acetabular failure occurred again.\nIn 2002, a high hip center bipolar hemiarthroplasty was performed via a standard posterior hip approach. Failed acetabular component and hardware were removed but the femoral stem was left in situ as it was found to be stable. Capsular and periacetabular scar tissues were preserved as much as possible to create a soft tissue cavity to seat the bipolar head into. A 60-mm bipolar femoral head was inserted onto the femoral component to articulate with the periacetabular soft tissues in a high hip center mode. Its position was further augmented with capsular repair around the neck of the prosthesis (capsular noose). A femoral condyle allograft was fixed to the ilium to serve as posterior superior acetabular wall. Postoperatively, the patient was advised to gradually increase her weight bearing using a walker or crutches. No casts or braces were applied. Within 6 months, the bipolar component migrated out of the acetabulum and articulated with the iliac soft tissues (Figure ). Although the patient had limb shortening, she had no pain and declined further surgery.\nThe right hip required 7 reconstructive procedures, which led to pelvic discontinuity (type IVb) and resection arthroplasty in 1999 (Figure ). In 2000, a re-implantation was performed by using a reinforcement ring with a cemented polyethylene cup and a long cemented femoral prosthesis. 4 years later the acetabular construct failed. A 56-mm diameter bipolar head was inserted onto the previously implanted and stable femoral stem and articulated with the soft tissues adjacent to the lateral ilium (Figure ). After surgery, the patient was able to transfer independently and ambulate short distances in her home with a walker.\nAt 3 years postoperatively (right hip) and 5 years postoperatively (left hip) the patient had no pain, relatively equal leg lengths, and could sit comfortably. Due to complete loss of lower extremity motor function associated with failed spine surgery and cervical myelopathy, the patient was non-ambulatory. However, the Harris hip score (HHS) of the patient’s left hip had improved from 39 preoperatively to 58 postoperatively. Similarly, the HHS of the patient’s right hip increased from 14 preoperatively to 58 postoperatively. |
A 46-year-old female patient presented to our outpatient clinic with postprandial nausea and vomiting. She stated that these complaints had started 6 mo earlier and had recently become worse. Her past medical history was not remarkable. On physical examination she only had tenderness in the epigastric region. Her biochemical parameters and tumor markers were within normal limits. Oral and intravenous contrast enhanced computerized tomography revealed a mass lesion with an approximate size of 100 mm × 80 mm that originated from the body of the pancreas and extended inferiorly (Figures and ).\nAs the mass did not invade vascular structures, a surgical intervention was planned. The abdominal cavity was entered via midline incision. After opening the gastrocolic ligament, the diameter of the dense was approximately 120 mm × 100 mm. It was noted that it originated from the pancreatic body, caused severe adhesions with adjacent tissues, and formed a conglomerated structure together with the fourth part of the duodenum and proximal jejunal loops. The mass was also severely adhered to the prepyloric antrum of the stomach. First, dense adhesions between the stomach and the mass were dissected with sharp dissection. Then, the extremely close anatomic relations of the mass with both the portal vein and the superior mesenteric artery were cut with sharp dissection. The conglomerated fourth part of the duodenum, proximal jejunum, distal pancreas, and the spleen were removed en-bloc. Then, an end-to-end anastomosis was formed between the third part of duodenum and proximal jejunum (Figure ). Supportive serosal stitches were placed along the anastomosis line. A jejunal tube extending to the proximal part of the anastomosis was placed in order to protect the anastomosis. The patient was discharged uneventfully.\nThe histopathological examination of the pathology specimen revealed a lesion with an approximate diameter of 55 mm and an appearance consistent with fibromatosis, which originated from the muscularis propria layer of the duodenum and extended into the pancreatic parenchyma (Figures and ). Immunohistochemically, the tumor was positive for vimentin (strong staining), beta catenin, cluster of differentiation 99 (CD99), smooth muscle actin (weak staining), calponin (patchy staining), and Ki67 proliferation index (5%) whereas it was negative for B-cell lymphoma 2, CD68, low molecular weight keratin, high molecular weight keratin, CD117, and pan-cytokeratin. Additionally, a hydatid cyst lesion with a diameter of 10 mm was detected in the neighborhood of the tumor (Figure ). The patient was administered etodolac for a total of 3 mo at the postoperative period. The tumor did not recur for a period of 24 mo postoperatively. |
A 46-year-old male presented with sudden visual disturbance that started one month prior to presentation. The patient had been having problems with blurred vision, loss of peripheral vision, and floaters in his left eye only. He was specific in that his symptoms were only occurring out of his left eye. He stated that his symptoms started with his central vision loss and then he could not see out of the peripheral part of his vision. He did have a history of ocular migraines, but these symptoms were different than his usual migraines. These new symptoms were persistent and not intermittent. In addition, he had just had a full eye examination by a neuro-ophthalmologist the week before and no eye abnormalities were found. The patient denied any events that provoked or alleviated the complaint. There was no significant medical and social history related to his chief complaint. He worked as a construction worker and reported a significant amount of stress in his life. Magnetic resonance imaging (MRI) of the brain and carotid ultrasound were recently done and were reported as normal.\nPast medical history included a history of lumbar back pain, gastroesophageal reflux, allergic rhinitis, and depression. He had a minor surgery as a child for his knees and a broken ankle. He was married, non-smoker, and worked full time as a construction worker. Family history was otherwise unremarkable. He had no recent foreign travel history. Review of systems was otherwise normal.\nOn physical examination, his vitals were normal. He was awake, alert, and oriented, and his mental status was normal. He had a normal eye examination except that he had a monocular left-sided inferior visual field defect (confirmed by formal visual field testing). Right eye examination was normal including visual eye fields. The remainder of the neurological and physical examination was normal.\nInitially, as the eye examination was normal and the symptoms were monocular, an assumption was made that the symptoms were from stress and that, with time, the symptoms would resolve.\nOn a subsequent visit one week later, the patient returned for follow-up. Despite taking some time off from work and reducing the stress level in his life, the patient still had persistent symptoms of left-sided monocular inferior quadrantanopsia. Upon insistence from the patient, a decision was made to repeat MRI of the optic nerves. In addition, the patient proceeded to tell the team that he had neglected to tell us that he was worked up for sarcoidosis a few years previously but no definitive conclusion had ever been made.\nFollow-up imaging of the optic nerves showed a small contrast-enhancing lesion on the left optic nerve. It was suspected that the patient may have sarcoidosis. He was also beginning to have some modest breathing difficulties.\nSubsequent assessment by bronchoscopy with biopsy confirmed a diagnosis of sarcoidosis. Upon further review of his MRI with neuro-radiology, a determination was made that the patient likely had NS causing the symptoms of his left eye. The patient responded well to treatment and the symptoms resolved.\nIn addition, further evaluation by both pulmonology and rheumatology helped to rule out other causes of granulomatous disease. |
Case 2 was a 70 years old woman with two histologically identical lung tumours in the right upper and lower lobes, respectively, both surgically removed with wedge resections. Both tumours were round and about 2 cm in diameter and rather well-circumscribed. A previous biopsy had suggested possible squamous cell carcinoma, but the morphology and IHC profile with >90% of the cells positive for p40, S100 and smooth muscle-specific actin (CK5 was positive in about 40% of the cells) was consistent with epithelial-myoepithelial carcinoma. See Fig. . Based on the tumours’ macroscopic appearance and peripheral location in the lung it was concluded the tumours were metastases. There was no evidence of epithelial-myoepithelial carcinoma in the salivary glands or in accessory salivary glands in the bronchi.\nThe patient had a previous history of a breast tumour originally suggested to be at first hand consistent with ductal carcinoma in situ, approximately 2 cm in size, surgically treated almost 5 years earlier. The tumours from the lung and breast were reviewed by several pathologists in Sweden and one international expert without consensus regarding the breast tumour. The morphology of the peripheral cells in the breast tumour and the cells of the lung tumours was similar although the growth pattern when comparing with the breast tumour as a whole was not perfectly identical and there were different opinions among pathologists whether the breast tumour was invasive or not. However, additional IHC staining of the breast tumour showed positive CK5, p40, p63, S100 and smooth muscle actin in the basal and peripheral cells, while the luminal cells were partly positive for CK5 (about 15% of the luminal cells) and estrogen receptor (about 20%). See Fig. . Progesterone receptor and HER2 were negative. The lung tumours were negative for estrogen and progesterone receptor and HER2.\nAfter targeted NGS analysis revealed the same PIK3CA mutation in both the breast and lung tumours there was a total agreement among the pathologists that the breast tumour was a malignant adenomyoepithelioma with metastases to the lung. In the breast there was also a PTEN mutation in a low frequency not seen in the metastases to the lung (checked for frequency less than 3%). There were no other mutations detected. See Table for full NGS data. An initial FISH for ALK gene rearrangements was inconclusive in one of the metastasis in the lung, and further FISH analyses were not performed after the result of the targeted NGS.\nExactly one year later one more metastasis, this time in the left lung, was surgically removed. It had the same morphological appearance as the previous metastases to the lung. The patient was still alive 18 months later with no evidence of any more metastases but with a suspicion of local relapse of the metastasis in the right upper lobe on a CT scan. |
A 65-year-old Caucasian female with a past medical history of hypertension, gastroesophageal reflux disease, iron deficiency anemia, and rheumatoid arthritis was referred for exposed left mandibular bone and a persistent neck fistula 3 weeks after extraction of tooth #20. She had a concomitant extraoral incision and drainage for a presumed submandibular abscess by an outside practitioner. The patient reported a history of being on bisphosphonates for osteoporosis. She was on Risedronate (Actonel) for a total of 4 years and was then switched to yearly Zoledronic Acid (Reclast) injections for a period of two years with the last dose being roughly 1 year prior to the extraction. She was however started on Denosumab (Prolia) subcutaneously roughly 1 week prior to her extraction.\nOn physical exam, the patient had a 1 cm cutaneous fistula in left submandibular region. Intraorally there was an area of exposed bone roughly 1 cm in size on the buccal aspect of the mandible in the area of tooth #20; there was a separate area of exposed bone on the lingual aspect. There was minimal amount of thin purulent drainage intraorally and on the neck bandage. A diagnosis of MRONJ was made based on her clinical presentation, and she was scheduled for follow-up with imaging to discuss her treatment options. At follow-up 10 days later, the patient's lesions had increased in size and the two areas of exposed bone were confluent. There was another 5 mm area of exposed bone in the symphysis region and another 5 mm area in the right retromolar pad area. The neck fistula slightly increased in size. CT scan showed sclerotic changes involving the mandible diffusely. Given the diffuse nature of her bony involvement and continued progression of bony exposure, we have elected to watch and wait and allow the necrotic bone to declare itself prior to proceeding with surgical resection and microvascular reconstruction.\nA week later, the patient was transferred from an outside hospital to our intensive care unit with a diagnosis of sepsis. Her left neck fistula had doubled in size and now had a necrotic appearance. The inferior border of the mandible was visible in the wound (). Intraorally, the area of bony exposure increased in size to 2.5–3 cm area in the left posterior mandible. The symphysis and right posterior mandible areas of bony exposure also increased in size (). Moreover the right side of her soft palate appeared to have undergone spontaneous necrosis, forming a through and through 2 cm defect into the nasopharynx. This fistula did not communicate with any areas of bony necrosis on exam and endoscopy (). The patient was started on broad spectrum antibiotics including Vancomycin, Levofloxacin, and Meropenem and ID was consulted. The patient was afebrile with a white cell count of 10.5 × 103/mm3. She suffered from tachycardia and was tachypneic and her chest X-ray showed findings suspicious of Acute Respiratory Distress Syndrome (ARDS) (). She required supplemental oxygen but no mechanical ventilation. She also received a blood transfusion at the outside hospital for hemoglobin of 5.0 g/dL and was transfused again in our facility for hemoglobin of 7.1 g/dL. She received multiple platelet transfusions for thrombocytopenia with a count 63 × 103/mm3 on arrival and a nadir of 16 × 103/mm3 (unit) during her 10-day hospital stay. The differential diagnosis by hematology was autoimmune causes due to her sepsis such as idiopathic thrombocytopenic purpura (ITP) versus drug related causes. The patient also had anion gap metabolic acidosis and was managed with fluids, insulin, dextrose, and bicarbonate and took several days to stabilize. Blood cultures eventually grew out Escherichia coli which was sensitive to the antibiotics administered. A repeat CT scan showed no abscess but showed some air in the marrow on left side of the mandible (). It also showed air in the epidural space in cervical region (). Neurosurgery recommended treatment with antibiotics.\nAs the patient progressed in her hospital course, she started to improve. Her anion gap was corrected, her counts improved, and her intra- and extraoral wounds stabilized. A PICC line was placed and the plan was to have the patient complete a 4-week course of Unasyn (Ampicillin/Sulbactam) after discharge followed by oral Augmentin (Amoxicillin/Clavulanate). Due to her generalized weakness and diffuse involvement of her mandible, the patient was deemed not a good surgical candidate at the time. There was also a concern with regard to soft tissue healing after surgery given her soft tissue wounds.\nAt follow-up 3 months after discharge, the palatal fistula was found to have completely healed. The neck wound had nearly completely healed with a pin point fistula with minimal drainage remaining. The 2 small areas of bony exposure in the symphysis and right posterior mandible had resolved. The left mandibular bony exposure was stable without progression. The patient continued to show progressive improvement on periodic follow-up. At 1 year, the soft palate and neck wounds were completely healed, and the intraoral wound had dramatically decreased in size with no purulence (). At 18 months the patient continued to do well. She essentially had near complete recovery with no surgical intervention. The patient was content with area of exposed bone in her oral cavity and was not interested in any conservative surgical debridement. |
A 36-year-old female presented to the maxillofacial surgery clinic complaining of gradually developing asymmetry of the right side of the face for the past 15 years. The progressive asymmetry of the entire right side of the face was noticed by her family members. Mandibular deflection toward the healthy side and overgrowth were noticed 15 years before, progressing gradually until they reached the present proportion. Furthermore, there was progressive development of pain in the TMJ region while opening the mouth. There was no history of trauma, vascular collagen diseases, infections, or surgery on the face or jaws. Her medical history and family history were noncontributory.\nThe extraoral examination confirmed facial asymmetry due to the downward displacement of the entire right mandible and an increase in the vertical height of the middle and lower facial thirds on the right side. The chin had deviated to the right side, and the lip line had slightly shifted downward, tilting toward the right side. Furthermore, mild tenderness was noted in her TMJs bilaterally, a clicking sound was heard during movement of the left TMJ, and tenderness was noted in the right side of the neck. The intraoral examination revealed a slight shift of the mandibular midline to the right side along with a mandibular cant. In addition, she had a posterior open bite, that is, posterior teeth of both the jaws were slightly tilted lingually to maintain the occlusion.\nThe panoramic radiographic image revealed significant uniform asymmetrical enlargement of the right mandibular condyle and elongation of the neck of the right mandibular bone, along with improper dental occlusion of the right side. The right gonial angle was characteristically rounded off and the mandibular canal was displaced to the lower border of the right mandible (Figure ). Computed tomography (CT) scan was performed to further characterize the abnormality. Three-dimensional (3D) volume-rendering CT showed asymmetry and differences in the size of both condylar heads as well as elongation of the neck of the mandibular condyle on the right side (Figure ). Accordingly, a three-phase bone scan along with single-photon emission computed tomography (SPECT) was performed to determine the activity of the condylar heads. The three-phase bone scan was unremarkable in blood flow (not shown) and blood pool (Figure ) images over the head region. However, three-hour delayed planar images demonstrated markedly increased focal tracer uptake at the right temporomandibular region (Figure ). Furthermore, there was bilaterally asymmetrical increased focal tracer uptake localized to the maxilla and mandible, which was related to the dental pathological process.\nThereafter, SPECT demonstrated intense focal activity corresponding to the right mandibular condyle, which appeared enlarged and measured 1.9 × 1.5 cm compared to the left mandibular condyle which measured 1.6 × 0.6 cm in transverse and anteroposterior dimensions. It was associated with elongation of the right mandibular neck and tilt of the mandible to the left side (Figure ). The left mandibular condyle demonstrated normal physiological tracer uptake. The relative uptake of the right mandibular condyle was markedly increased at 73.4%, while the relative uptake of the left mandibular condyle was only 26.6%. There was no evidence of erosive or sclerotic changes at the TMJ bilaterally (Figure ). Moreover, there were no erosive or sclerotic changes at the TMJs bilaterally.\nThe clinical and radiologic findings were consistent with a diagnosis of unilateral CH of the right side of the face. The patient was referred to a tertiary center to be managed accordingly. |
A 31-year-old Bosnian male with an unremarkable health history was originally seen in early October 2015 at a walk-in medical clinic after he noticed a change in his voice for 3 weeks. The patient had no smoking history, no prior occupational exposures, was an infrequent social drinker and worked as an assistant manager at a convenience store. Initially he thought the hoarseness was secondary to an upper respiratory tract infection and surprisingly did not notice the neck mass until it was pointed out to him (Fig. ). The physician who examined him at the time felt a large mass in the left anterior portion of the neck and followed up with a neck ultrasound. The neck ultrasound was unremarkable revealing a homogeneous thyroid gland with no solid or cystic soft tissue masses along with normal appearing lymph nodes on both sides of the neck.\nTwo days later he presented to the emergency department because of increasing hoarseness and mild dysphagia. The department of Otolaryngology Head and Neck Surgery was consulted. At the time of consultation, the patient appeared to be in no airway distress and had normal vital signs. On physical examination, a palpable mass was felt on the left side of the neck. The rest of the head and neck examination was unremarkable. Laboratory investigations were ordered and included CBC, serum ionized calcium, thyroid stimulating hormone, free T4 and T3 and calcitonin. These laboratory findings were all within normal limits with the exception of a slightly elevated T3 level. Direct fiberoptic nasopharyngolaryngoscopy was performed and revealed a supraglottic mass involving the left vocal cord causing immobility (Fig. ). The mass appeared to be submucosal in nature and did not affect the right vocal cord. He was then admitted for observation and further workup for diagnostic purposes.\nA CT scan with contrast was performed and showed a 5 × 5.7 cm laryngeal mass centered on the left thyroid cartilage, which was completely destroyed (Fig. ). It displayed some central decreased attenuation and was associated with early punctate calcification. The tumor extended medially and compressed the laryngeal ventricle and airway at the level of the vocal cords. Superiorly it extended to the level of the left intact hyoid bone and compressed the left pyriform sinus. There was compression and displacement of the left carotid sheath and left sternocleidomastoid with no sign of any direct invasion. A 1.1 cm diameter lymph node was seen just lateral to the jugular vein and inferior to the tumor on the left-hand side. No other bone or soft tissue abnormality was observed. At this point the appearance of the mass was very suggestive of a chondrosarcoma. Magnetic resonance imaging (MRI) revealed the same findings as the CT scan and maintained the suspicion for a chondrosarcoma.\nTo clarify the diagnosis, an open biopsy was performed. Histopathological findings from the biopsy specimen revealed uniform sheet appearance of multinucleated osteoclast-like giant cells (Figs. and ). The intervening cells between the giant cells showed band nuclear features supporting a diagnosis of giant cell tumor.\nThe patient’s case was discussed at the Saskatoon Cancer Centre Head and Neck rounds and the consensus was to proceed with a surgical approach. After consulting with various head and neck cancer specialists across Canada, reviewing the literature, and respecting the patient’s wishes it was decided to proceed with a TL. The decision to proceed with a TL versus a PL was extremely difficult. With the extreme size of the tumor and significant pharyngeal invasion, it was postulated that a partial laryngectomy in this particular case, would result in a high degree of remaining laryngeal dysfunction.\nOn December 2015, the patient underwent surgery. The following procedures were performed: total laryngectomy, left hemithyroidectomy, pharyngeal plexus neurectomy, partial pharyngectomy, anterior pharyngotomy, and creation of tracheoesophageal fistula for future voice prosthesis (Fig. ).\nAt 13 months follow-up the patient has no evidence of disease or recurrence. He is able to tolerate all textures of foods and is happy with the quality of his voice. In terms of communication he states he “doesn’t have any problems” and has been doing “very well from a psychological standpoint”. He was connected with an existing laryngectomy patient for peer support prior to surgery, which he found very helpful and gave him “a piece of mind.”\nHe currently works two jobs, the first being an assistant manager at a convenience store and second as an office clerk. He has an interdisciplinary degree in political science and business administration and is hoping to return to school in the future. One day he hopes to swim again as this was his main form of exercise and stress relief prior to surgery. The patient will have continued follow up in clinic to ensure no tumor recurrence. |
A 17-year-old female living with extreme BED and obesity presented to hospital with aggressive behaviour towards her primary caregiver (grandmother). Despite being followed in an interdisciplinary obesity management programme, her binge eating behaviours had resulted in a 45 kg weight gain over 12 months, resulting in numerous medical complications. Comorbid psychiatric diagnoses included anxiety disorders, post-traumatic stress disorder and obsessive compulsive disorder. Prior to admission she resided in a group home with intensive psychological supports. Much of her therapeutic work at the group home focused on processing trauma and increasing effective coping skills using dialectical behaviour therapy and trauma informed approaches. However, due to limited resources, patients were sent home on weekends. Removed from the structure and extra support the group home provided, she reverted to ineffective strategies to gain emotional control such as binge eating and aggressively rejecting any boundaries set by her caregiver.\nThe patient was admitted to a ward designed for individuals diagnosed with restrictive eating disorders as no acute care treatment options existed for BED. The main goals of admission were symptom interruption, safety planning and care coordination. With structured meal times, interruption of bingeing was successfully achieved within 72 hours. Much of the admission (25 days) was then, however, dedicated to coordinating a discharge plan that would allow for intensive mental health support, while also ensuring safety for the patient's caregiver. Numerous barriers to finding a suitable treatment programme for an adolescent living with BED, complex obesity and mental health comorbidities surfaced. She could not return home with her grandmother due to safety concerns and, as a result, could also not return to the group home or attend an outpatient day programme. No residential programmes for youth living with BED existed. Ultimately, she was discharged to a residential home for youth living with mental health concerns that was not specific to eating disorders. The lack of long-term care options for youth with BED and comorbid mental health concerns was striking.\nMany learning points stemmed from the juxtaposition of admitting an adolescent living with severe obesity and BED to a restrictive eating disorder unit. Logistically, there were challenges related to medical equipment including unsuitable gown sizes, blood pressure cuffs and a scale incapable of recording higher weights. Applicability of usual ward protocols requiring all meals to be finished and supplementing calories for food refusal were questioned by unit staff.\nThe patient herself demonstrated insight into this stating, “I could see the nurses struggle to come up with a plan to control my eating habits as I was the polar opposite of all their past and present patients.” Additionally, the question of whether admitting a patient living with severe obesity would psychologically affect the patients admitted with restrictive eating behaviours, and vice versa, was also raised. |
A 32-year-old woman was transferred to our hospital for a giant tumor in the mediastinum, which was detected during a health checkup. She had no overt signs or symptoms and the results of the laboratory tests were unremarkable. The abdominal ultrasonography and electrocardiography were normal. The posteroanterior chest radiography () demonstrated a huge mediastinal shadow with well defined margins that extended to the two lung fields and there was no calcification and ossification. The pre-contrast CT scan revealed a giant cystic-solid mass that measured 16 × 13 × 8 cm and it was located in the center of the anterior mediastinum and it symmetrically grew toward the two lung fields; the mass was well-circumscribed and compressed next to the aorta and the superior vena cava. The cystic lesion with a mean CT attenuation value of 15 Hounsfield unit (HU) had a slightly thickened wall. Most of the solid components that were found in the backside of the mass obscured the aorta, superior vena cava and main pulmonary trunk, and the solid components had an inhomogeneous density with a mean CT attenuation value of 42 HU (). On the post-contrast CT scan, the solid part demonstrated marked enhancement with a mean CT attenuation value of 130 HU and the cystic wall of the lesion showed moderate enhancement (). A provisional diagnosis of cystic teratoma with cancerization was made. No metastatic lesions were found on the preoperative staging, and surgical exploration and resection were then performed.\nThe patient underwent a complete surgical resection of the giant mass via a median sternotomy. A mass posterior to the sternum and chest wall was observed in the anterior mediastinum. It had adhesions to the adjacent vascular structures and the mediastinal pleura. Pathological examination during the surgical procedure indicated no malignancy, and so no further dissection was performed. The gross pathology revealed a 16 × 13.5 × 7.8 cm cystic-solid mass, and about 1,500 ml of brownish, turbid fluid was found in the cyst. Microscopically, the solid components presented as complete pancreatic tissue with a normal acinar structure and islets of pancreas (). Therefore, the pathologic diagnosis was an ectopic pancreas in the mediastinum with cystogenesis. Postoperatively, the patient recovered uneventfully and no recurrence or metastasis was found during the three months follow-up. |
The patient's clinical history started when she was 13 years old and presented with a generalized tonic–clonic seizure, which lead to a magnetic resonance imaging (MRI) of the brain with the subsequent diagnosis of multiple intracerebral CVs: a left frontal intraparenchymal one (35 mm in diameter) and a left posterior temporal one, both within the parenchyma (23 mm in diameter), and an intraventricular one (30 mm in diameter). Despite the best medical treatment, the epilepsy was not well controlled and the patient had up to three to four epileptic attacks per week. This case was discussed several times at our multidisciplinary meeting, as well as with the patients and the parents. The final decision was to remove the largest and the apparently symptomatic CV, and this decision was guided by a video-EEG (electroencephalogram). The left frontal CV was removed at the age of 14 years, with epilepsy symptoms being temporarily improved. Unfortunately, after 10 months, she started to complain of epilepsy again, with a clinical absence type behavior, pointed for temporal lobe origin type of seizures. Thus, a few months later, the left posterior temporal lesion was removed as well. The second operation gave very good medical results in terms of seizures control. The episodes dropped to one or two focal seizures per year. The third lesion, the intraventricular one, was followed up with a yearly MRI scan. At the age of 21, because the lesion had increased in size (∼8 mm) and because of the patient's desire, we decide to remove it using a transcranial interhemispheric approach. The operation was uncomplicated, and the patient was discharged home a week after the procedure. At that stage, no other lesions were present, and in the following 10 years, the follow-up MRI scans did not show any recurrence or new CVs. When she was 32 years old, on the yearly follow-up scan, a newly developed lesion was identified. This lesion, suspicious for CV, was small (6 mm) and located within the septum pellucidum. Because of its small size, the location, and the absence of symptoms, a conservative treatment option was followed. Unfortunately, the lesion doubled in size in the following 18 months and therefore the patient was very adamant about having it removed (\n). We were a bit reluctant because the patient was completely asymptomatic and had not had any epileptic attack for 10 years. Upon neurologic examination, she presented no issues. Finally, we took the decision to remove the lesion and we started to discuss how to approach it. We were wondering whether to use the same interhemispheric approach with the possibility of encountering scar tissue or if it was better to use a new surgical route such as a transcortical one. Finally, we decided to use something completely different and we opted for a transcortical endoscopic approach.\nWith the patient in the supine position through a single burr hole, placed slightly more laterally in relation to Kocher point, a purely endoscopic approach was performed and the lesion was completely removed (\n). A rigid endoscope was used and guided by the neuronavigation. Upon inspection, the lesion (\n) presented with two veins attached to it (one rostral and the other caudal). The removal began with the coagulation and dissection of the septum pellucidum superior to the CV location. After accurate coagulation and section of the caudal vein, using endoscopic forceps allowed the creation of a “pedunculated” CV. The insertion of an endoscopic rongeur in the space between the peel-away cannula and the endoscope allowed keeping the CV in place, avoiding its fluctuation in the ventricles. This maneuver allowed the exposition and easy dissection of the rostral vein, which, eventually, was coagulated and cut. The CV was then freed from the surrounding tissue and finally removed. An external ventricular drainage was precautionary left in the right ventricle just for 24 hours.\nThe histological examination revealed multiple dilated and congested vascular spaces lined by the endothelium, confirming the diagnosis of a CV.\nThe postoperative course was uneventful, and the patient was discharged home 2 days later. Serial follow-up MRI scans did not show any new or recurrent lesion at 5 years follow-up. |
The patient is a 40-year-old male, originally from India who works as a camel herder. He reports no chronic medical illnesses especially no diabetes mellitus and no immune suppressive therapy. On the day of admission, he was bitten by an otherwise healthy camel while he was tying it down. He arrived to our emergency department 3 hours after injury. He was found to be vitally stable and afebrile. He was opening eyes spontaneously, obeying command but mildly confused. No lateralizing neurologic deficits were noted on his neurological examination. On scalp inspection, he was found to have 2 small cut wounds on the left side and 3 on the right side both just above the pinna of the ear; all of these wounds are consistent with teeth-bite marks. An abrasion above the left supraorbital region was noted, likely secondary to the fall he sustained after the bite. All the wounds were dry and the skin around them was healthy looking with no subcutaneous collections. He received tetanus toxoid in the ER. Computed tomography scan of the head was obtained and showed depressed skull fracture on the left temporal region associated with pneumocephalus but no intracranial hematoma or contusion were seen (). Given his early arrival to our ER, all the wounds were irrigated with normal saline and the patient was started on intravenous flagyl and augmentin immediately to cover oral and skin flora. In addition, phenytoin was started as seizure prophylaxis, given his less-than-perfect initial level of consciousness and the depressed skull fracture over the left temporal lobe. Initial laboratory blood tests were within normal limits. After just 12 hours, the patient was found to be ill-looking with mild fever. The wounds were found to be indurated and with erythematous edges and started to show small amount of lightly purulent discharge. He still maintained his good level of consciousness with a GCS of 15 and showed no focal neurological deficit. He was taken to the operating room for debridement of all wounds. To our surprise, the purulent collection was found to be spreading well beyond the edges of the teeth-puncture wounds into the subgaleal space in the retro-auricular area bilaterally. In the left side, the 2 puncture wounds were connected together to maximize the drainage of the purulent material. The depressed skull fragment was elevated, the dura was sutured after copious irrigation, and the skin was closed primarily. In the right side, the 3 puncture wounds were connected and the pocket of purulent discharge was found to have reached the upper part of the cervical region. As such, the wounds were packed and allowed to heal by secondary intention and were attended to by daily dressing. He continued on antibiotics and wound dressing with close follow-up by our infectious disease colleagues who added rabies vaccine. He was discharged home after 10 days of intravenous antibiotics and was continued on oral antibiotics for another 2 week. He was seen in the outpatient department at that time, and his wounds were healing well with no residual infections. |
A 32-year-old female patient reported to the Department of Oral Medicine and Radiology with a complaint of a growth present on the right side of cheek since last 3 months. The patient gave a history of similar kind of growth on the right cheek 7–8 years (2007) back for which she had undergone partial surgical removal 6 years (2009) back by local dentist and report had been given with leukoplakia. After surgery, she had noticed a gradual increase in the size of the growth for which she had undergone second surgery 4 years (2011) back. During second surgery complete removal was done. In report atypical changes of cells were present in the sample was mentioned. After 1 year of complete removal of growth, it again reappeared. Then third surgery was performed, complete removal done and diagnosis was given in favor of inclusion cyst (sebaceous cyst). After third surgery, no recurrence was seen for next 2 years. Six to 7 months back she had noticed a white patch initially on the same region which had gradually been increasing and had become a growth since last 2 months. On the region of growth, she often felt mild and intermittent pain and used to chew during eating.\nThe patient reported no history of past medical details. On personal history, she reported of chewing paan masala and betel nut since last 8–9 years. She used to take 3–4 packets/day. On clinical examination mouth opening was found to be slightly reduced. Mouth opening was 37 mm blanching was present on both right and left buccal mucosa extending from retromolar region till the 16.46 region on the right side and 27, 36 regions on the left side giving marble like appearance. Diffuse brown pigmentation was present on buccal mucosa. A thick nonscrapable white lesion was present on right buccal mucosa near the commissure of lip measuring approximately 1 cm × 2 cm in diameter. On the right side, an exophytic soft tissue growth was present irt 47.48 measuring approximately 0.3 cm × 1 cm in diameter extending from distal aspect of 47 till the distal surface of 48 anteroposteriorly and suproinferiorly from the occlusal level of 48 till the mucogingival junction. Superficial mucosa of growth was hyperkeratinized and multiple projections elevated from the base. On palpation base of growth was firm with multiple superficial projections []. Reduced cheek flexibility was present on both sides with vertical, horizontal bands in retromolar region. After clinical examination, diagnosis was made in for growth as verrucous leukoplakia irt 48 with leukoplakia on right buccal mucosa and oral submucous fibrosis Grade 1. On management, we had planned for surgical removal of growth by diode laser. Before surgical procedure hemogram assessment was performed along with viral profile. Diode laser of 980 nm wavelength was used for the surgical excision. The laser was used in continuous mode at 2 watts. The growth was surgically completely excised using laser [].\nA slight char tissue in the base of the wound was seen, this char acts a bioprotective plug that has prevented bleeding and also helps to prevent infection in such wounds. The absence of bleeding was the most evident feature of this wound. The borders were rolled out, and these borders flattened out later. The excised tissue was sent for histopathological examination. The biopsy confirmed the case to be that of verrucous leukoplakia. The patient was recalled for follow-up after 7 days []. On the 7th day follow-up, granulation tissue was seen which was sign of secondary healing. The patient came after 11 and 13 days for follow-up. Gradual decrease in site of surgery and complete healing as seen [Figures and ]. After 1 month, complete resolution of the lesion was seen without recurrence and restricted mouth opening. Histopathologically, multiple soft specimens were sent to Oral Pathology Department measuring 11 mm × 2 mm, 8 mm × 4 mm, irregular in shape, slightly blackish in margin and firm. Under the microscope, the section showed high cellular stroma. On higher magnification, fragmented epithelium was seen. Epithelium showed cleft formation with parakeratin plugging. The connective tissue stroma is condensed with dense inflammatory cells mainly lymphocytes and endothelial lined blood vessels suggestive of verrucous leukoplakia (Batsakis et al. Grade 2) []. |
A Japanese male who was diagnosed as having severe hemophilia A (factor VIII activity less than 1%) due to a large thigh hematoma at 10-month old. He was diagnosed with chronic kidney disease at age of 18 on the basis of persistent proteinuria with hematuria. He was diagnosed with congestive heart failure and end-stage renal disease at age of 21. He started to undergo PD, but heart failure recurred one month later because of his poor self-management. Thereafter, he underwent combination therapy with HD (for 4 hours, once per week) and PD (1.5% Dianeal PD-2, 4 times per day, 6 days per week). Factor VIII (1000 U) was administered about three times a week (once a week after HD and irregular self-injection). He developed suddenly severe headache eight months after the beginning of the combination therapy. When he presented at the emergency room, his blood pressure was 200/100 mmHg. His blood tests showed prolonged APTT (110 second). In spite of immediate treatment by antihypertentsive medication (oral nifedipine), one hour later, he had a generalized convulsion due to intracerebral hemorrhage. The next day, his cerebral hemorrhage enlarged and his consciousness level worsened despite administration of factor VIII and intravenous anti-hypertensive medications. Emergency surgery was performed to remove the hematoma. He received a large amount of factor VIII to achieve an APTT level less than 40 seconds for one week. He regained consciousness after surgery and was managed with PD without HD to avoid cerebral edema. One month later, his renal replacement therapy was switched to HD (three times a week) from PD, since a ventriculoperitoneal shunt catheter was placed to treat his hydrocephalus. Neither clot formation nor hemorrhage was found after hemodialysis which was performed without anticoagulant agents. He received 1000 U of factor VIII before removal of the dialysis needle at the end of every HD session to maintain the APTT level between 50 sec and 60 sec. The factor VIII levels were not influenced by the hemodialysis (), indicating that factor VIII did not pass through the dialysis filter.The APTT levels under management with HD were significantly shorter than those under combination therapy (PD and HD) (). He could be discharged from hospital, and he underwent maintenance dialysis safely without anti-coagulant as outpatient thrice weekly. |
A 14-year-old girl was brought to our epilepsy department. Her first attack was noticed 6 months ago when her mother went to her bed to awaken her. She could not gain her full consciousness with blurred speech. When she was forced to stand up, parents noticed that she was not able to walk without support. She was taken to a state hospital and internalized for 3 days in intensive care unit. Biochemical tests, cranial magnetic resonance imaging, and lumbar puncture analysis were normal. Electroencephalography (EEG) showed random generalized epileptic discharges with the photic sensitivity. She was diagnosed to have idiopathic generalized epilepsy and given valproic acid (750 mg/day). She had no history of convulsions. The attack lasted for about 4 days. No changes of personality, appetite or sexual behavior were noticed by the family. No triggering factor was noticed by the parents. At the end of about 1-week, the patient was in premorbid condition without any residual symptoms.\nThe second attack occurred 4 months later, characterized same as the previous attack, but lasted for 5 days. She had a tendency to sleep, difficult to be awakened and had decreased responses to external stimuli, including pain. This attack was also decided to be of epileptic in origin; the dose of valproic acid was increased to 1250 mg/day, and antibiotics were given for infection of undetermined etiology suggested by increased serum inflammatory markers. This second attack lasted for about 4 days, similar to the previous one, without any accompanying change in personality, appetite or sexual behavior.\nFour months after this event, she had another attack and was brought to our department. She was still within attack period, which had started 3 days ago. We performed continuous video-EEG monitoring, and it revealed that the patient was in sleep []. The latest attack lasted for 5 days, with full recovery of the child without any neurological symptoms. Repeated EEGs demonstrated randomly occurring generalized epileptic discharges with the photic sensitivity [], irrespective of the clinical status of the patient. A detailed medical history revealed that she was at menarche with irregular menstruations, coinciding with the attacks. Of three attacks, two started 2 days before menstruation, and the last attack started simultaneously with the menstruation. There were no associated features of behavioral or cognitive abnormalities, compulsive eating, sexual disinhibition or delusions/hallucination. The patient, however, was confused during the attacks with some degree of feeling of unreality. There was no sign of dysautonomic features or psychiatric comorbidity. Family history revealed that her father had primary generalized epilepsy that was cured for more than 10 years.\nThe patient was diagnosed as menstruation-related recurrent hypersomnia. Co-existing generalized epileptic activity was accepted as an incidental comorbidity, and because there was no history of seizures (generalized tonic-clonic, absence, myoclonic, or any other), the patient was not diagnosed as having epilepsy. She was given carbamazepine with a daily dose of 400 mg. At the end of 19 months follow-up duration, she is still symptom-free. |
A 35-year-old female patient was presented to our center for TV replacement due to stenosis. At the age of 11 years, the patient suffered from endocarditis supported by a small ventricular septal defect. At that time, the ventricular septal defect was closed and a mechanical tricuspid valve (SJM, 28 mm) implantation and epicardial single chamber pacemaker implantation due to concomitant third degree AV block without escape rhythm were performed. At the age of 17 years, the abdominal pacemaker was replaced, due to battery depletion (Biotronik). Six months later, a second surgery followed with replacement of the dysfunctional mechanical valve for a 27 mm Porcin-Baxter biological TV. Another PM replacement (Medtronic) was performed at the age of 27 years, again, due to battery depletion. At the age of 30 years, the abdominal pacemaker was explanted and a two chamber transvenous ICD (Medtronic Egida DR) was introduced, as the patient suffered a syncope during documented sustained ventricular tachycardia. The ICD lead was implanted passing the tricuspid valve into the right ventricle. Six years later, severe TV stenosis developed, caused by an ICD lead, that stuck to the posterior leaflet of the biological valve and ventricular myocardium, and the TV had to be replaced a third time. To avoid future TV dysfunction due to lead related complications, we discussed several options with the patient. These included first TV replacement in combination with epicardial pacemaker leads and a subcutaneous ICD (S- ICD, EMBLEM™ Boston Scientific) or alternatively placing a pacing lead in the coronary sinus in combination with an S- ICD. As the patient disagreed on both options, despite a higher risk of TV dysfunction by placing another transvenous ICD lead through the TV, we discussed another option and opted for a lead sparing replacement of the TV. The TV ring was cut open on both sides beneath the original ICD lead, and the TV prosthesis was excised. A new bioprosthetic valve was implanted, leaving the ICD lead outside the ring of the new TV prosthesis. As the lead was stuck to the former TV annulus and posterior ventricular wall, no further fixation of the lead was needed. The new valve (SJM biological 28 mm) was implanted using 14 Coreknots (Figures , , ).\nThe postoperative course was unremarkable, and the patient was dismissed on the 11th postoperative day/postoperative day 11. Follow-up after 4 months showed a well functioning TV prosthesis without stenosis or regurgitation and no deterioration in pacing or sensing parameters. No artifacts or other hints to a lead dysfunction were recorded (Table ). |
A 75-year-old Caucasian male presented in September 2019 with a long-term asymptomatic mass (for approximately the last 3 years) that had increased in recent years ().\nThe patient had a medical history of laparoscopic cholecystectomy for cholelithiasis in 2008. The pathological study indicated a chronic cholecystitis with an incidental diagnosis of adenocarcinoma in situ over a 12 mm adenomatous polyp located at the bottom. The operative report mentioned that the removal of the gallbladder was difficult due to the inflammatory process, and the gallbladder was accidentally opened during the operation. It was not clear from the operative report whether an extraction bag was utilized to remove the specimen, but the histopathological study confirmed an open gallbladder. The patient did not undergo any adjuvant therapies, and there was no postoperative follow-up medical history.\nThe current physical examination revealed a mass effect in the mesogastrium of stony consistency, painless to palpation and no signs of peritonitis. An abdominal contrast-enhanced computed tomography (CT) scan was performed (). A heterogeneous mass in the abdominal wall measuring 12 × 6 cm, predominantly cystic, with septa inside, was noted affecting the rectus abdominis muscle. A subsequent magnetic resonance imaging (MRI) further delineated the mass lesion with no associated abnormality in the rest of the biliary system or pancreas ().\nGiven the presence of mucin and the history of adenocarcinoma in situ of a gallbladder polyp, a gastroscopy and a colonoscopy were performed. The colonoscopy showed tumors in the descending colon and sigma with biopsies from both sites confirming adenocarcinoma.\nThe patient underwent an elective laparotomy. We found an amorphous, well-defined mass that involved the skin, subcutaneous fat, the rectus muscle, and the posterior sheath of the rectus without affecting the peritoneum or the abdominal organs (). En bloc resection with surgical margins (R0 resection) free of tumor was performed. After that, the abdominal cavity was explored without any suspicion of malignancy, liver metastasis, or peritoneal carcinomatosis. Then, a left radical colectomy and sigmoidectomy were performed. Finally, we repaired the defect in the abdominal wall using a prosthetic mesh (GoreTex Dual Mesh, Bard®) as a bridge between the two aponeuroses. The postoperative period was uneventful. The patient was discharged on the seventh day after surgery.\nThe histopathological results were described as metastasis of adenocarcinoma of biliary origin (positive for CK7, CK18, CK19, CK20 (focal), CDX2 (focal), MUC1, MUC5AC, MUC2 (weak and focal), EMA, CA19. 9, CEA (focal), CD10 (focal), beta-catenin; negative for CA125, PAX8, PSA, TTF1, S100, estrogen and progesterone receptors, CD34, inhibition) without affecting surgical margins. The histological study of the colon revealed an adenocarcinoma in sigma T3N0M0 (according to the eighth edition of the AJCC) while the left colon showed an adenocarcinoma in situ (TisN0M0) (). The patient was evaluated by the oncology department, and no adjuvant therapy was recommended.\nDuring the follow-up (18 months), the patient has not presented evidence of recurrence, wound infection, or mesh rejection. The follow-up schedule based on an abdominal contrast-enhanced computed tomography scan and analytical blood test with tumor markers (CEA and CA 19.9) was every 6 months. |
A 30-year-old woman visited the hospital due to right lower quadrant pain over the period of 1 week. A laparoscopic myomectomy was performed 4 years ago because of a broad ligament leiomyoma, which was about 10 cm in size. Laboratory findings included a routine blood examination, and a C-Reactive Protein (CRP) test, with tumor markers all found to be within normal ranges. The pelvic Magnetic Resonance Imaging (MRI) scan showed some nodules at the posterior margin of the bladder that were considered to be endometriosis, and some pelvic effusion that was significant on the right side. It was also noted that there was a mass shadow at the lower right ureter (medial to the iliopsoas muscle), with distention of the lower right ureter (Fig. ). The patient also underwent computed tomography (CT) scans to enable the practitioners to observe the size of the abdominal mass and its surroundings. The CT images revealed a region occupying the middle right ureter that was considered to be a retroperitoneal aggressive fibroma, which led to severe hydronephrosis on the right kidney and upper ureter, and a right pelvic effusion (Fig. ). After more detailed examinations were conducted, there were no obvious abnormalities detected in the brain, heart, liver, gallbladder, spleen, pancreas or blood. The color Doppler ultrasound demonstrated that there was a hypoechoic mass next to the right iliac vessels that was closely related to the adjacent ureter. This resulted in severe hydronephrosis of the right kidney and a right upper ureteral dilatation (Fig. ). Ndzengue et al. [] reported a case of a pelvic desmoid tumor simulating a uterine leiomyoma recurrence. The patient that presented at our hospital had a history of uterine leiomyoma. We subsequently organized a multidisciplinary consultation to determine the next stage of her treatment plan. According to the patient’s surgical history, the next step would be determined after reviewing the results of the last surgical pathological wax, because the pathological nature of the retroperitoneal mass was uncertain.\nConsequently, a laparoscopic pelvic tumor resection with assistance from a Da Vinci robot was successfully conducted. A local peritoneal protuberance was observed in the right iliac vascular area. The parietal peritoneum was incised above the mass and it was carefully freed along the edge of the mass. The size of the tumor was about 6.0 cm × 5.0 cm × 3.0 cm. It had invasively grown and it was wrapped around the right ureter and the right ovarian arteries and veins. It was stuck to the psoas muscle and the iliac vessels. According to preoperative preparations and intraoperative conditions, a decision was made to cut off the right ureter, the right ovarian arteries and veins, and completely remove the tumor and the two masses that were located in front of the broad ligament on the right hand side of the uterus. The sizes of the masses were approximately 1.5 cm × 1.0 cm × 1.0 cm. The right ureter was anastomosed and put into a double J tube. A pathological diagnosis of an intraoperative frozen sample determined that it was a spindle cell soft tissue tumor, and the two masses were leiomyomas. Postoperative pathology tests of the pelvic mass determined that it was aggressive fibromatosis that had invaded the ureteral wall (Fig. ). The uterine surface nodules were also leiomyomas. Microscopically, the tumor cells were arranged sparsely in a spindle shape with blood vessels of different sizes found in the interstitial tissue. Immunohistochemical findings were found to be partially positive for smooth muscle actin (SMA) and desmin, and less than 5% ki-67 of positive cells were seen in the lesion. A detailed re-examination was performed 3 months after the initial surgery, to determine the structure and function of the ovaries. The transvaginal color Doppler ultrasonography was able to determine that the ovaries were normal in size with several follicular echoes. The blood supply to the right ovary was good. There were no obvious abnormalities in the uterus or pelvic cavity (Fig. ). The pelvic MRI and the CT scan of the whole abdomen determined that there were no abnormal lesions in the pelvis. Simultaneously, the endocrine function of the patient’s ovaries was found to be normal, and she was able to self-maturate after removing the double J tube. |
A 62-year-old man was referred to our hospital because of recurrent syncope. The patient had syncopal episodes while drinking fluids starting seven years ago. He reported the feeling of a foreign body sensation at his mid sternal area after drinking a cold beverage, and then suddenly lost consciousness. The duration of unconsciousness was a few seconds. There were no seizure-like movements or incontinence during the attack, and he felt no numbness or motor weakness before or after the events. The patient reported more than twelve episodes of syncope before presenting to the cardiology outpatient clinic at Samsung Medical Center. A skin laceration was noted on his forehead from the last episode of syncope.\nThe patient was diagnosed with atrial fibrillation seven to eight years ago, and had been taking warfarin since then. He was also diagnosed with diabetes mellitus three years ago. His diabetes was well controlled with oral hypoglycemic agents.\nThe initial electrocardiogram (ECG) showed atrial fibrillation with a ventricular response of 60-70. His echocardiography did not show any significant abnormality of the heart except bilateral atrial enlargement. On the second day of admission, he felt a globulous sensation at the mid esophagus after drinking a cold beverage in the sitting position, and then lost consciousness. His telemetry ECG monitoring at that time showed abrupt ventricular asystole that was sustained for 12 seconds (). Another episode of syncope occurred on the same day while drinking. His telemetry ECG monitoring showed ventricular asystole for 4 seconds ().\nWe evaluated the esophagus with a barium swallow; there were no abnormal findings in the esophagus. On the treadmill test, his heart rate and blood pressure increased to 176/min and 218/83 mmHg, respectively. The head-up tilt test three months before admission was negative; the head-up tilt test was repeated. During the repeated head-up tilt test, provocation with drinking a cold beverage showed no significant pause or hypotension.\nThe patient underwent permanent pacemaker implantation of a VVI type pacemaker to prevent ventricular asystole. After implantation of the permanent pacemaker, there were no further episodes of syncope after drinking a cold beverage (). |
A 37-year-old Caucasian man with a history of hypertension presented to his primary care physician with rhinorrhea, a cough and an enlarged lymph node in the left posterior neck that had been worsening over the past one to two months. Our patient was initially treated with a course of oral antibiotics, but had no improvement in his symptoms. He then developed intermittent numbness and tingling in his bilateral feet, which progressed to involve both of his legs and his fingertips. After a few more days, he developed weakness in his bilateral lower extremities and his hands. A computed tomography (CT) scan of his head revealed no significant abnormalities. A fine-needle aspiration of the neck lymph node was suggestive of, but not definitive for, HL. Excisional biopsy of a left supraclavicular lymph node was consistent with classical HL, nodular sclerosis-type.\nFurther workup with a positron emission tomography (PET)/CT scan revealed small volume left cervical, supraclavicular, axillary and mediastinal lymphadenopathy. A bone marrow biopsy demonstrated a cellular bone marrow with a slight increase in eosinophils, but no evidence of lymphoma, so our patient was given a diagnosis of stage IIB classical HL. By that time, the weakness in his arms and legs had become worse and our patient had experienced at least two episodes of falling with difficulty getting up without assistance. Our patient had right facial weakness, three out of five strength in his bilateral shoulders and hip flexors, four out of five strength in his knee flexors and decreased vibratory sensation up to the knees bilaterally. Patellar and Achilles reflexes were not present.\nA magnetic resonance imaging (MRI) scan of his brain showed no intracranial abnormality. An MRI scan of his spine showed subtle nerve root enhancement of the cauda equine, but cerebrospinal fluid was negative for any evidence of malignant cells with only one white blood cell present (CSF protein = 205). Findings from an electromyelogram and nerve conduction study were consistent with an AIDP or GBS. Our patient received two days of intravenous immune globulin (IVIG) at a dose of 1g per kg with complete resolution of his sensory complaints and improvement in his weakness. Three days later, his neurologic symptoms had worsened along with development of some mild shortness of breath. Plasma exchange was initiated for a course of five days with resolution of his shortness of breath and mild improvement of his weakness and sensory deficit.\nThe following week, chemotherapy with doxorubicin, bleomycin and dacarbazine was initiated (Table ). Vinblastine was originally omitted from the standard doxorubicin, bleomycin, vinblastine, and dacarbazine (ABVD) regimen to avoid exacerbation of our patient’s neuropathy. It was added back during cycle 2 and again held after our patient developed redness and tenderness in his fingertips following 3a. It was reintroduced at a reduced dose after cycle 4 and at full dose for cycle 5.\nBleomycin was omitted for cycle 1b and cycle 2a due to the development of cough and a 20% reduction in diffusion lung capacity for carbon monoxide (DLCO) on repeat pulmonary function tests (PFTs), but then reintroduced once our patient’s cough resolved. It was again omitted after cycle 5a due to worsening shortness of breath and further decrease in DLCO on PFTs.\nBy the start of cycle 2, our patient’s Eastern Cooperative Oncology Group (ECOG) performance status had improved from two to one and our patient no longer needed a cane for ambulation. Our patient was also treated with another dose of IVIG, which helped further improve his neurologic symptoms. After cycle 3, a PET/CT scan showed significant improvement in left neck, left axillary, left hilar and mediastinal lymph nodes. Since there was not complete resolution of the lymphadenopathy, our patient was continued on ABVD. A repeat PET/CT scan after cycle 5 showed residual disease with an increase in metabolic activity, so a biopsy was performed of a left neck lymph node and revealed persistent classical Hodgkin lymphoma.\nOur patient was then treated with ifosfamide, carboplatin, etoposide (ICE) for two cycles with persistent disease, gemcitabine and vinorelbine for two cycles with persistent disease followed by brentuximab vendotin for three cycles with progressive disease. Note that brentuximab can cause peripheral sensory neuropathy; our patient experienced numbness and tingling in the lower extremities that resolved after four days.\nSince our patient still only had localized disease at that point, intensity-modulated radiation therapy (IMRT) was given to the mediastinum, neck and axilla at a total dose of 30.6Gray (Gy) in 17 fractions with an additional 5.4Gy boost to the mediastinum and neck. He then underwent an autologous stem cell transplant with carmustine, etoposide, cytarabine and melphalan conditioning.\nOur patient went into remission for about six months, but then developed recurrent disease in his right lower lung. He is currently undergoing chemotherapy with bendamustine with plans for an allogeneic stem cell transplant. Interestingly, his neurologic symptoms had mostly resolved despite the persistence of his HL. |
This was a case of a 33 year old Gravida 5 Para 4 + 0 with 2 previous lower segment caesarean section scars, at 28 weeks of amenorrhoea using Naegele’s formula. She was a peasant farmer by occupation and her husband was a peasant farmer too. The indications for the previous caesarean sections were; cephalopelvic disproportion during the third pregnancy and then inadequate pelvis with one previous scar during the fourth pregnancy. She had two living children and these were the ones born by caesarean section after having lost the first two children during their perinatal stages. She had no family history of multiple pregnancy or hypertension or diabetes. She presented with lower abdominal pain for 11 h; the pain was vague in nature, non-radiating, relieved by lying down, exacerbated by walking, but not severe enough to affect daily activities. She had no vaginal bleeding or any vaginal discharge; she clarified that she had not had any flow of fluid from the vagina along her thighs. She had not any other gastrointestinal complaints or genitourinary complaints. We had no access to her antenatal care records because she had received antenatal care from another health unit, and she had not moved with her antenatal care documents; therefore we could not find out whether she had received medicines like NSAIDs that affect amniotic fluid volume during pregnancy. She had no pallor of mucous membranes, pulse rate was 72 bpm, blood pressure was 110/80 mmHg, had a subumbilical midline incision scar, fetal heart rate was regular at 150 bpm, cervix was thick and with a parous os. A decision to admit her due to possible preterm labour and concealed abruptio placenta was made, and she was given dexamethasone intramuscularly. Obstetric ultrasound scan was done and it revealed severe oligohydramnios with no measurable amniotic fluid pool; estimated gestation age was 29 weeks and 1 day and estimated fetal weight was 1300 g. There was not any placental abnormality. A diagnosis of severe oligohydramnios with 2 previous scars was made. A differential diagnosis of preterm premature rupture of membranes was also made as a possible explanation for the oligohydramnios. Her haemoglobin level was 11.1 g per decilitre (g/dl), white cell count was 11,010/ millilitre (ml), platelet count was 363,000/ml.\nDecision was made to deliver her by emergency ceaesaren section; she received prophylactic antibiotics and intravenous fluids. Intraoperatively, we found a gravid uterus with a transverse rupture on the lower segment anteriorly and along the previous scar, about 6 cm long with a protruding fetal arm. There was no active bleeding from the uterus. The peritoneal fluid was observed to contain some vernix caseosa. A baby boy of apgar score 10 at 5 min and weight of 1200 g, was delivered and admitted to the neonatal intensive care unit, and there was no complication of the surgery with the patient recovering well after uterine repair. There were no postoperative investigations done because the patient recovered well. Bilateral tubal ligation was not done because she had not consented to it, but she got explanation about the risk of uterine rupture in a subsequent pregnancy and was given family planning counseling. Figure showing the intraoperative findings and the timeline in Table are attached to this case report article. |
The patient was a 44-year-old man who had gone to a regional hospital with worsening dyspnea on exertion and lower extremity edema approximately 2 months prior to admission. He was referred to our hospital for diagnosis and treatment. The patient did not have an obvious history of occupational or incidental exposure to asbestos. The physical examination at admission revealed jugular distension and lower extremity edema. His blood pressure was 120/58 mm Hg and heart rate was 120 beats/min. Transthoracic echocardiography and computed tomography showed massive pericardial effusion, with a maximum diameter of 53 mm at the apex of the pericardial sac and a large tumor (95 × 99 mm in diameter) occupying the lateral to posterior pericardial space (A, B). The tumor showed heterogeneous contrast enhancement, and its border was unclear. Computed tomography showed lymphadenopathy of the mediastinal and subcarinal lymph nodes. Coronary angiography revealed that feeding vessels of the tumor extended from the circumflex artery (). Thoracentesis and pericardiocentesis were performed; however the signs/symptoms of tamponade remained. Cytological findings of the pericardial fluid specimen was grade II and was negative for malignant cells. After a preoperative discussion, our team decided upon a surgical intervention for the definitive diagnosis and to reduce the tumor mass as much as possible to improve the patient’s symptoms. A median sternotomy and pericardiotomy were performed, and the bloody pericardial effusion was removed. The pericardium was thickened. The large tumor was adherent to the epicardium and pericardium and extended from the lateral to posterior side (A). We performed a blunt dissection of the adhesions. The margins between the tumor and epicardium were unclear and most of the tumor was bluntly dissected. Some of the pericardium with adhesions to firm tumor tissue was removed, and the opened pericardium was not repaired so that pericardial fluid could drain to the thoracic space to alleviate cardiac tamponade. Only the tissue that included the feeding vessels was ligated and cut with an Endo GIA surgical stapler (Medtronic, Minneapolis, Minnesota, United States) in order to cut and ligate safely. Cardiopulmonary bypass was not needed for removal of the large tumor, which was resected as much as possible. We resected the large tumor by dividing it into sections. The weight of the resected tumor was 480 g (B). The histopathological diagnosis of the resected tumor specimen was malignant mesothelioma, sarcomatoid type (). The pericardial fluid obtained at surgery showed grade V cytology. It showed irregularly shaped nuclei and numerous mitotic divisions, and the cells were identified as malignant cells. The patient’s symptoms improved and his early postoperative course was uneventful. Postoperative computed tomography showed reduction of the tumor mass (A). We recommended adjuvant therapy for the patient because of his young age and he and his family agreed to proceed. At 3 weeks after surgery, he was administered carboplatin and pemetrexed; however, the tumor progressed. The tumor was irradiated, with a temporary reduction in size. However, 2.5 months after surgery, the tumor regrew and compressed the left ventricle (B). Heart failure developed. Chemotherapy was discontinued because of hematotoxicity. Despite maximum radiotherapy, the tumor started to increase in size. The patient was transferred to hospice care for palliative therapy and died 7 months after surgery. |
A 50-year-old male underwent a decompressive craniectomy and evacuation of intracranial hematoma because of his subdural hematoma and severe cerebral edema, caused by a motor vehicle accident. After a month, he developed hydrocephalus and required a VP shunt. His right Kocher's point was used for ventricular entry, and the peritoneal catheter was placed through a paramedian subcostal incision. He was transferred to the rehabilitation department and had been in a semicomatous state for 8 months.\nA percutaneous endoscopic gastrostomy tube was placed six months after the VP shunt by a senior gastroenterologist who had more than 10 years of endoscopic experiences. The tube was introduced into the stomach through the epigastrium with a standard endoscopic pull-through technique. As usual, the patient received prophylactic antibiotics, and the procedure was performed with special attention and using a gentle technique so as not to interrupt the intraperitoneal shunt catheter.\nAfter a few hours, the scalp covering the surgical defect of his skull, which had been made by decompressive craniectomy, began to swell up. On the next day, it became worse and we took a brain CT scan which showed severely enlarged intracranial ventricles ().\nHe had no fever or leukocytosis. Furthermore, no red blood cells or white blood cells were found in his cerebrospinal fluid. Skull X-rays were taken to confirm that the valve setting of the VP shunt had not been changed. The postprocedural abdominal film showed no gastrointestinal complications such as pneumoperitoneum or ileus, but kinking in the intraperitoneal shunt catheter was observed compared to the catheter being smooth before the procedure (). The patient underwent a distal shunt catheter revision. We made a small incision in the subcostal area and pulled out the intraperitoneal catheter. The externalized catheter was carefully observed and no obstruction or disconnection was found. After we confirmed the adequate CSF drainage of the catheter, we put it into his peritoneal cavity again. Postoperatively, the swollen scalp depressed down and the sizes of the intracranial ventricles normalized in the follow-up CT scan. After a few days of close observation, the patient was transferred to the rehabilitation department. |
A 62-year-old male with a 4-year history of chronic intermittent aching pain along the distribution of the left LON presented severe aggravation in frequency and intensity of left-sided headache with duration of 4 months. An aching pain over the left temporal area above the ear was associated with left mandibular angle areas during its initial development 4 years ago. He had been treated for dental caries. However, the treatment was not effective. He had experienced intermittent neck pain prior to the occurrence of left temporal headache. However, the pain was not severe enough to seek medical consultation. There was no precipitating event before the onset of headache. The pain was described as aching and pressure like in nature with moderate intensity (4–6 out of 10 in the numerical rating scale [NRS]). During the initial 2 years, the cluster of left temporal headache with duration of 2–3 weeks occurred 3–4 times a year. The frequency of headache cluster was increased to 5–6 times a year and its intensity was gradually increased.\nOne year prior to presentation, the pattern of occurrence and spreading of the headache became typical on every occasion. An aching pain gradually developed in his left upper temporal area over the ear and spread to posterior temporal and retroauricular occipital areas []. It then extended to the suboccipital area and lateral neck. Duration lasted up to 1 h. The headache even developed during sleep. He could not fall asleep again after awakening. He has been using three pillows on bed for more than a year to prevent the occurrence of headache. Four months prior to admission, the headache was continuously present. It occurred more than five times a day. He had been treated with various kinds of medication including triptans, propranolol, indomethacin, tramadol, and antidepressants. They were not effective at all. Repeated blocks of the GON were partially effective about 2 h. Only injection of diclofenac sodium was effective for 3 h. Magnetic resonance imaging (MRI) findings of the brain and cervical spine were nonspecific. He was finally referred for the management of refractory headache.\nThe aching and deep pressure-like headache was severe in intensity (6–7 out of 10 on NRS) at presentation. It was not aggravated with neck motion including extension. Daily activity did not influence the occurrence or frequency of headache. His medical history was unremarkable, including diabetes and gout. He denied any history of trauma to the neck. The headache was not associated with tinnitus, photophobia, lacrimation, nausea, or vomiting. Upon examination, there was no hypesthesia, allodynia, or tenderness in his left head or neck. No limitation of neck motion was observed. Neurologic examination including cranial nerve was normal. Careful reevaluation of cervical spinal MRI raised a suspicion of foraminal stenosis at the left C2/3. Therefore, myelographic computed tomography (CT) of the cervical spine was requested. A sagittal oblique three-dimensional reconstruction of myelographic CT revealed significant narrowing of the left C2/3 foramen and small osteophyte of the uncovertebral joint []. Under impression of the left C3 radiculopathy owing to foraminal stenosis, three times of selective C3 root blocks were performed with 2 ml of 1% lidocaine. The pain was completely relieved for 2 h.\nConsidering chronicity and severity of the headache, decompressive foraminotomy of the left-sided C2/3 spine was performed. The lateral edge of dural sac and proximal nerve root sheath of the left C3 were decompressed under microscopic vision [Figures and ]. Immediately after awakening from anesthesia, the pain completely disappeared. No hypesthesia or paresthesia was reported. The patient refused any medication against headache. There was no recurrence of headache or sensory deficit in his left temporal area at 1-year postoperative follow-up. |
A 67 year old Caucasian female with a past medical history significant for cutaneous malignant melanoma diagnosed 7 years ago with systemic metastasis to the brain was seen by a visiting nurse at home (Fig. ). The nurse found the patient with heart rate in the 40's without symptoms and was sent to the hospital for further management, where a 12 lead electrocardiogram demonstrated sinus tachycardia with complete heart block, junctional escape rhythm at 42 beats per minute and poor R wave progression in the precordial leads (Fig ). An echocardiogram was obtained, which demonstrated normal left ventricular systolic function with concentric left ventricular hypertrophy. On echocardiography, the septum, inferior and posterior wall were thick and there was increased echogenicity of these walls which was thought to be from hypertrophy (Fig ). Considering the history of systemic metastases of the malignant melanoma along with complete heart block, suspicion was raised to rule out cardiac metastasis. A gated cardiovascular magnetic resonance (CMR) study was performed. The steady state free precession (SSFP) cine vertical long axis and the horizontal long axis view of the left ventricle demonstrated normal left ventricular contractility with nodularity of the myocardium (Fig. ). This was representative of marked tumor infiltration of the myocardium (pan cardiac) with nodular deposits in the myocardial muscle layers with varying penetration into the endocardium and the epicardium. On SSFP cine sequences, these nodular deposits were discrete, isointense to bright compared to the normal myocardial muscle (Fig. ). The nodularity involved all myocardial walls including the membranous septum, proximal and distal anterior septum, inferior wall, anterior wall and the lateral wall (Fig. ). There were multiple mass-like deposits lining the right atrial and the left atrial wall with similar signal intensity as that of the left ventricular masses. The right ventricular free wall also demonstrated nodular deposits. On both T1 and T2-weighted images, the left and right ventricular and atrial masses were bright (Fig. ). On post-gadolinium images, these masses were enhancing due to contrast uptake and extracellular contrast retention. The cardiac valves appeared normal and there was minimal valvular disease (Fig. and ). There was a small pericardial effusion, but the pericardial layer was free of disease. The hilar and paratracheal lymphadenopathy was present along with large left and small right pleural effusion. |
A 39-year-old man with hoarseness and dysphasia was referred to our department for evaluation of left vocal cord palsy and a tracheal tumor that was revealed on computed tomography (CT). He had a previously resected left upper eyelid tumor that was diagnosed as schwannoma. However, he did not have other problems in his medical or family history. Laryngoscopy demonstrated a fixed left vocal cord. CT showed a tumor involving 3.5 cm of the left tracheal wall and protruding inside and outside the tracheal wall (Fig. ). Positron emission tomography (PET) presented a maximum standard uptake value of 3.09 on the tumor. There was no accumulation on other parts of the body. Bronchoscopy verified a submucosal tumor affecting four tracheal cartilaginous rings (Fig. ). Endobronchial ultrasonographically guided transbronchial needle biopsy and aspiration did not detect malignant tissue and cells.\nAlthough a preoperative diagnosis could not be made, the tumor was suspected to be malignant. If the tumor were to become larger, tracheal resection for complete removal would become impossible due to associated risks. Therefore, we performed median sternotomy to explore and diagnose the tumor with a subsequent planned tracheal resection if indicated and possible. Informed consent from the patient and his family was obtained. Surgical exploration revealed a yellowish, soft, multinodular tumor stretching along the left recurrent nerve and infiltrating the tracheal and esophageal walls (Fig. ). The left recurrent nerve involved with the tumor was transected, and an incisional biopsy was performed. The intraoperative pathological diagnosis was schwannoma without malignancy. This tumor was much more widespread than expected. The tumor invaded outer tracheal wall over four tracheal rings. However, submucosal infiltration seemed to be developed more. We confirmed small nodules on the cranial side of tracheal wall apart from the main tumor, which were also diagnosed as schwannoma (Fig. ). The invasion to esophageal wall could not be anticipated from CT findings. As it was impossible to achieve complete resection, we determined to preserve the trachea and resect the tumor nodules to the greatest extent possible (Fig. ). Additional movie files can show this operation in more detail [see Additional files and ].\nThe pathological features were identical to those of typical schwannomas, including being composed solely of Schwann cells frequently disposed in a compact, palisaded fashion (Antoni A) (Fig. b) and occasionally in a loose texture (Antoni B). This tumor did not have cellular atypia, hyperchromasia either. Cellularity was not high. There was no remarkable pleomorphism. We could confirm few mitotic activities. Immunohistological staining for S-100 protein showed diffuse positivity, while that for neurofilament showed weak positivity only in peripheral areas, which is atypical for a neurofibroma. The patient’s postoperative course was uneventful. A lump on the trachea remained, but no further growth was observed in an 8-month postoperative follow-up period.\nTaking the findings for this tumor into consideration, we can postulate two hypotheses about how this plexiform schwannoma progressed. First, it is possible that there were two individual tumors—one arising from the left recurrent laryngeal nerve and the other from the trachea itself. Second, the plexiform schwannoma may have originated from the recurrent nerve and infiltrated the tracheal wall. Whichever postulation is true, such an occurrence is very rare.\nPlexiform schwannomas have been reported to represent 4.3 % of all schwannomas, and they are often seen in the head and neck region []. Only one case of tracheal plexiform schwannoma was described in a case series reporting uncommon primary tracheal tumors [], and it was not presented in detail. In a literature search using PubMed, we could not find any description of a plexiform schwannoma originating from the recurrent laryngeal nerve or of a plexiform schwannoma infiltrating the tracheal wall. In one report, the association of plexiform schwannoma with neurofibromatosis type 2 and with schwannomatosis was 5 % each []. Our patient did not have a history of bilateral vestibular schwannomas or of tumors arising from the central nervous system, such as meningiomas. He also did not have a family history suggesting neurofibromatosis. Schwannomatosis has recently been recognized as the third major form of neurofibromatosis that causes multiple schwannomas without a diagnosis of neurofibromatosis type 2 []. Some researchers have reported genetic alterations in this disorder []. However, the diagnostic criteria have not yet been established. Our patient had a history of left eyelid schwannoma, and he may be diagnosed with schwannomatosis in the future.\nIf repeated needle biopsies or resection of the intraluminal bulge under rigid bronchoscopy had proven schwannoma before surgery, we might avoid trying the risky tracheal resection and observe the tumor unless tumor growing or tracheal stenosis occurred. However, at that time, it was not expected that the tumor was a benign tumor, such as schwannoma, and a repeat biopsy was not done. We therefore determined that we could make an accurate diagnosis and adequate treatment decision through surgery. After the intraoperative diagnosis, we determined to preserve the trachea and resect as much of the tumor as possible. Wright and coworkers argued that tracheal resection by more than 4 cm was a significant risk factor for anastomotic complications, although there were some measures to loosen the tension []. In the present case, even if we had attempted the longer tracheal resection, there would be no guarantee that we would not leave a tumor at the tracheal stump. Residual plexiform schwannoma can recur. Recurrent plexiform schwannomas were reported even with such a high-risk surgery [, ]. Moreover, as the tumor had infiltrated a part of the esophageal wall, we considered that it would be impossible to achieve curative resection. Plexiform schwannoma can become enlarged, but it occurs slowly. Debulking is supposed to extend significantly the progression-free period. Periodic follow-up must continue in order to check for regrowth. If this tumor grows up intraluminally, we will resect it under rigid bronchoscopy.\nPathological diagnosis of this tumor was typical schwannoma without malignant findings. Some of plexiform schwannoma are difficult to be differentiated from malignant peripheral nerve sheath tumor []. With detailed pathological findings about cellularity and mitotic activity, this tumor could clearly be distinguished from malignancy. Therefore, the prognosis of this case is expected to be good though local recurrence may happen. A few malignant transformations were reported in familial schwannomatosis []. It is unknown whether the plexiform schwannoma in this patient may transform to the malignancy. We should suspect of malignant transformation when we see the rapid enlargement. |
A 64-year old man underwent Oxford medial UKR in September 2004. He had previously had an arthroscopy of the same knee for a complex tear of the medial meniscus. The patient’s medical history included coronary artery bypass graft, atrial fibrillation, and right mid-foot fusion. He had been on a number of medications including Warfarin.\nAt the index procedure a medium-sized femoral component, 50 × 32 mm tibial tray, and a size 7 meniscal bearing provided a stable articulation. The knee remained relatively pain-free for 5 years until he sustained a twisting injury to his knee. Following this he developed severe pain and swelling in the knee and was unable to weight bear. He attended the A&E Department on the same day and was reviewed in the fracture clinic the following day. Radiographs demonstrated a posterior dislocation of the mobile bearing.\nThe patient consented to an exchange of bearing or revision total knee replacement. The knee was explored through the previous mini anteromedial incision. Both femoral and tibial components were found to be well-fixed with no macroscopic evidence of burnishing, abrasions or scratching over the metal surfaces. The anterior cruciate and collateral ligaments were intact. Intraoperatively, despite an extensive search, the dislocated bearing could not be retrieved. A trial reduction with a size 9 medium meniscus provided a stable articulation with no impingement, no varus or valgus instability, and no lift off of the trial meniscus. It was decided not to explore the knee through a posterior approach to retrieve the meniscus in order to avoid the added morbidity of a more invasive procedure. A definitive bearing was inserted and the wound was closed in layers. Total surgical time for the procedure was 1 h and no additional antibiotics were administered. The postoperative recovery was uneventful and the patient was mobilised, fully weight-bearing, without any difficulty. The patient's status was reviewed regularly in the outpatient clinic. Clinically he remained relatively pain-free and continued to enjoy a relatively normal function of the left knee for 2 years following insertion of the replacement bearing.\nIn order to ensure that the dislocated bearing was not migrating and endangering the neurovascular bundle in the popliteal fossa, we performed serial ultrasound scans. These scans demonstrated that the meniscus was located posteromedially approximately 2 cm below the joint line and some distance from the neurovascular bundle (Fig. ). Its position remained unchanged. The patient did not wish to have any further surgery to retrieve the dislocated meniscal component, which would have required a posterior approach. |
A 48-year-old Korean female patient presented with an abnormal mass lesion that was detected by abdominal computed tomography in a visit to our hospital. She had experienced intermittent abdominal pain for several months. She had no other specific past medical history and no history of trauma. The patient’s vital signs were stable and laboratory tests were normal. Chest X-ray showed no abnormal findings and the computed tomography of her chest showed a 4-cm-sized round mass with areas of calcification in the left hemidiaphragmatic area (Figure \n). After a review of the diagnostic imaging, we were still unable to localize the mass, but we concluded that the lesion was most likely located in the left pleural space based on its proximity to the diaphragm. We decided to remove the mass. The patient was taken to the operating room for thoracoscopic surgery. General anesthesia with double lumen endotracheal tube intubation and one lung ventilation was done. Two 5-mm ports and one 10-mm port were placed in the left chest (fifth intercostal space in the midclavicular line, sixth intercostal space in the anterior axillary line, and eighth intercostal space in the posterior axillary line) for the thoracoscopic approach. No mass was visualized in the pleural space, but a bulge was visualized in the diaphragm consistent with the location of the lesion noted on chest computed tomography. The diaphragm was opened with electrocautery around the mass lesion. Then we identified the mass in the diaphragm (Figure \n). The mass was adhered to the crucial fibers of the diaphragm but was relatively well marginated. We dissected carefully, and a small feeding vessel was noted and clipped. The dissection was relatively easy and the mass was removed. The diaphragm defect was closed with interrupted polyester sutures and one chest tube was placed. The postoperative course was uneventful. The chest tube was removed on the third postoperative day and the patient was discharged the following day. The specimen measured about 4 cm in diameter, 9.4 gram in weight, and was well-defined and reddish. Cut sections of the mass showed sponge-like appearance with cartilage and yellow-colored mucoid materials. Histologic evaluation of the specimen was consistent with the diagnosis of an extralobar sequestration (Figure \n).\nPulmonary sequestration was first defined by Pryce in 1949\n[] as characterized by a non-functional lung without communication with the bronchial tree and the presence of an aberrant blood supply. On the basis of morphological patterns, they are divided into two types: intralobar and extralobar. An intralobar sequestration shares the same pleura with normal lung, but an extralobar sequestration has a separate pleura. Intralobar sequestrations are more common (75-85% of cases), while only 25% are extralobar sequestrations\n[,]. Extralobar sequestrations are most commonly found in the thorax, usually on the left side\n[]. Only 10-15% of extralobar sequestrations are located in the abdomen\n[,]. Usually, extrathoracic extralobar pulmonary sequestrations are infradiaphragmatic, masquerading as suprarenal masses\n[,-]. Intradiaphragmatic extralobar pulmonary sequestration is rare and there have been very few reported cases until now\n[-]. The location of extralobar pulmonary sequestrations in the diaphragm sheds light on the relationship between the embryology of sequestration, diaphragm, and lung. The pleuroperitoneal folds form and coalesce the primordial diaphragm from the body wall during the 9th to 12th weeks of gestation; therefore, a bronchopulmonary sequestration that arises during this period may have a higher chance of forming within the diaphragm\n[]. True intradiaphragmatic pulmonary sequestrations are rare and all reported cases have been younger than two year old. In the present case, the patient was a 48-year-old female. This is the first case found in an adult. Pulmonary sequestration can usually be identified by diagnostic imaging as a soft tissue mass with an aberrant blood supply\n[]. In our case, computed tomography of the patient showed a soft tissue mass but did not reveal an aberrant blood supply. The imaging diagnosis of intradiaphragmatic pulmonary sequestration is not easy. In 2009, Meier et al.\n[] described the “split hemidiaphragm sign” as a radiologic finding of two leaflets of diaphragmatic muscle surrounding a soft tissue mass on computed tomography. This is helpful for preoperative diagnosis of this rare disease. However, in our case, we could not identify such findings on our patient’s computed tomographic scan. The appropriate management of extrathoracic extralobar pulmonary sequestration remains controversial. Some authors advocate expectant management without resection\n[,]. Other authors recommend embolization of the systemic artery as a treatment option\n[]. However, most authors recommend surgical removal, especially for extrathoracic lesions, due to concern for infection, malignant degeneration, and difficult differentiation from another neoplasm\n[,]. In this report we could not diagnose the extrathoracic pulmonary sequestration. We chose surgical removal to allow differentiation from another neoplasm such as teratoma or certain types of malignancy. For surgical removal of intradiaphragmatic extralobar pulmonary sequestration, thoracoscopy is recommended. McAteer et al.\n[] described that thoracoscopy provides excellent visualization of intradiaphragmatic masses and easy access for surgical resection. They also noted that the thoracoscopic approach allows careful dissection of the mass away from the diaphragm and primary repair of the resulting defect. In our case, we performed the operation via thoracoscopy and dissection of the mass from diaphragm and primary repair of the defect were not difficult. |
A 43-year-old man with a history of leukoplakia underwent biopsy of his oral mucosa in 2010; it revealed moderate to severe dysplasia. He remained asymptomatic until 2014 when he felt a mass in his tongue. A computed tomography (CT) scan of the head and neck showed a density in the right tongue with no cervical lymphadenopathy. Biopsy of the tongue revealed moderately differentiated squamous cell carcinoma (SCC). A staging positron emission tomography (PET) scan demonstrated evidence of ipsilateral cervical lymph node involvement. He underwent tracheostomy, right neck dissection, right tongue cancer resection, and reconstruction with a free flap graft from his right forearm. Pathology revealed a 3 cm, invasive, well-differentiated SCC of the keratinizing subtype. The patient received 2 months of chemotherapy with cisplatin and radiation. A PET scan was done in May 2015 that showed complete remission.\nA surveillance CT scan done 1 year later, in May 2016, showed left lung lesions suspicious for metastatic disease, and bronchoscopy confirmed SCC of these lung lesions. He then underwent chemotherapy with 2 cycles of paclitaxel, carboplatin, and radiation. A repeat PET scan in September 2016 showed complete response, and the patient decided to proceed with observation. Another surveillance CT scan in December 2016 showed a cardiac lesion in the left ventricular (LV) apex (). The patient was referred to our specialized cardiomyopathy clinic. In the clinic, the patient's physical examination was unremarkable but his electrocardiogram (ECG) showed ST elevations in the anterior and lateral leads suggestive of myocardial injury (). He underwent a comprehensive transthoracic echocardiogram, which showed a 4.6 × 2.8 cm mass infiltrating the apical anteroseptal and anterolateral wall segments (). The mass had an abnormal texture with less echodensity than the adjacent LV myocardium and was highly suspicious for metastatic disease. The patient underwent a cardiac magnetic resonance imaging (MRI) scan, which demonstrated a 3.3 × 4.2 cm infiltrating lesion within the apex of the LV without early or delayed enhancement (). The patient was referred for a right ventricular echocardiogram-guided myocardial biopsy. The pathology immunohistochemical stains (p40 and CK5/6) were consistent with myocardial involvement by metastatic SCC (). The patient was started on palliative immunotherapy treatment with pembrolizumab. A follow-up cardiac MRI done 2 months later showed a substantial increase in the size of the mass as well as extension into the right ventricular apex (). A repeat PET scan done in March 2017 showed widespread metastasis (). The patient's treatment was switched to palliative combination chemotherapy with 5 fluorouracil, carboplatin, and cetuximab, to which he had a very good partial response. A repeat cardiac MRI done in July 2017 showed some improvement in the overall size of the cardiac mass. However, a cardiac MRI done in December 2017 showed interval progression of the infiltrative tumor mass involving the LV myocardium, with features suggesting central necrosis (). A follow-up PET scan showed multiple new metastatic lesions (). The patient was admitted to the hospital in February 2018 with worsening dyspnea and acute hypoxic respiratory failure. An echocardiogram showed interval progression of metastasis to the left and right ventricular cavities (). |
A healthy 27 year-old immunocompetent male was referred for orthopaedic oncology evaluation of a possible neoplasm involving the inferior pole of his left patella. The man described a four month history of progressive anterior knee pain and swelling. Suspecting a knee infection, his orthopaedic surgeon performed two separate aspirations for culture, both of which were negative for any microorganisms including fungus. Radiographs demonstrated a lucent lesion in the inferior pole of the patella, and a CT and MRI of the knee were ordered. The initial radiologist’s interpretation was suggestive of a primary bony neoplasm, and orthopaedic oncologic consultation was obtained.\nThe patient presented to our clinic with left anterior knee pain and limited range of motion. He reported an episode of “Valley Fever” two years previously when he worked in the Central Valley of California. The isolated pulmonary infection was ultimately confirmed to be Coccidioides spp. by sputum culture. He was treated with a 6-month course of oral fluconazole with complete resolution of all symptoms. Notably, he never had any musculoskeletal complaints during that episode. On review of systems he described a 10 to 15 pound weight loss over the past month with intermittent nocturnal fevers and sweats.\nOn physical examination, the patient walked with an antalgic gait. There was a left knee effusion and warmth. He had localized tenderness along the inferior pole of his patella. His passive range of motion was limited from 5 to 95 degree. There was no palpable mass about the affected knee.\nRadiographs of his left knee demonstrated a joint effusion and lucency involving the inferior pole of patella, which was visualized best on the lateral view (Figure ). On CT performed at an outside hospital, a low attenuation lytic lesion was more clearly defined without associated soft tissue calcification (Figure ). The outside MRI demonstrated knee joint effusion with extensive synovitis extending into Hoffa’s fat and continuity with the low attenuation area within the inferior pole of the patella along with extensive marrow edema (Figure ) of the proximal tibia and patella.\nLaboratory data at our institute revealed a mild elevation in the C-reactive protein level of 3.0 (normal range: 0 ~ 0.8) and the Westergren erythrocyte sedimentation rate of 22 (normal value: 0 ~ 10). He had a normal white blood cell count (6.93 × 103) with a normal polymorphonuclear cell differential (66%) and a mildly elevated monocytic differential (9%, normal monocyte differential: 2% ~ 8%). His coccidioides antibody serology tests were positive by complement fixation (CF) method (1:1024, normal value: <1:2) as well as by the enzyme immunoassay method to IgG (IgG antibody: 9.6, normal value: ≤0.9) and IgM (IgM antibody: 1.2, normal value ≤ 0.9) antibodies.\nKnee infection related to the previous coccidiomycosis exposure was highly suspected despite the two prior negative cultures on aspiration. He therefore underwent arthroscopic examination and biopsy of the left knee which revealed inflamed synovium throughout the knee with soft necrotic bone at the inferior pole of the patella. Frozen section analysis of the patellar bone revealed abundant chronic inflammatory cells without evidence of malignancy. Extensive open debridement over the inferior patella was performed. Postoperatively, his pain was immediately better, and his fevers and night sweats resolved.\nFinal pathologic examination revealed large thick walled spherules filled with endospores establishing the final diagnosis of extrapulmonary coccidioidomycosis (Figure ). The patient was treated with an oral antifungal regimen (oral fluconazole 800 mg as loading dose at first day, followed by 400 mg daily for at least one year) per infectious disease specialist recommendations. At six week follow-up, he had complete resolution of symptoms and normal knee range of motion.\nAn active 78 year old immunocompetent male was referred for orthopaedic oncology evaluation of a left patellar bone lesion. The patient had experienced three months of increasing left anterior knee pain without any history of antecedent trauma. He had no history of fevers, chills, malaise, or weight loss. His only medical issues were cardiac arrhythmia requiring pacemaker placement and benign prostatic hypertrophy. The referring physician had attempted conservative treatment with a corticosteroid injection without improvement of the knee pain. The patient lives in the central valley of California, but had no known history of coccidioidomycosis.\nPhysical examination was notable for exquisite tenderness of the superolateral border of the patella. Gait, alignment, and knee range of motion were normal. No inguinal or popliteal lymph nodes were palpable. There was no knee effusion.\nRadiographs revealed a lucent lesion of the superolateral patella (Figure ). Bone scan demonstrated increased uptake of the lesion without any other areas of abnormal skeletal uptake (Figure ). Computed tomography further demonstrated the low attenuation lesion without associated soft tissue mass or knee effusion (Figure ). Notably, the imaging reports stated that there was a high suspicion for primary bone malignancy or metastatic disease.\nOpen biopsy was performed, and gross examination of tissue from the lesion was most consistent with infection. Frozen section analysis was non-diagnostic. Limited curettage of the lesion through the open biopsy tract was therefore performed. Final pathology revealed spherules filled with endospores, classic for coccidioidomycosis (Figure ). Culture of the tissue confirmed Coccidioides spp. infection. Chest radiographs showed mild right hilar fullness. His coccidioides antibody serology tests were positive by complement fixation (CF) method (1:32, normal value: <1:2) as well as qualitatively by the enzyme immunoassay method to IgG but not to IgM.\nThe patient noted immediate improvement of his knee symptoms post-operatively. One month follow-up revealed resolution of the patellar tenderness and normal knee function. The patient was seen by an infectious disease specialist and initiated on a 6 month course of oral fluconazole. |
A 50-year-old female patient was referred to our hospital for further evaluation and management of an incidentally reported right atrial mass. The patient had a history of ankylosing spondylitis under treatment with methylprednisolone and methotrexate, and factor V-Leiden mutation that had lead to several peripheral venous and arterial thrombosis during the last 5 years. The patient also experienced multiple episodes of pulmonary embolism that had been treated with anticoagulants. Although under warfarin treatment with a good time in therapeutic range of international normalized ratio, the patient developed recurrent pulmonary embolism which was later attributed to the presence of a newly diagnosed right atrial mass. The patient was suffering from heart failure symptoms with New York Heart Association class III–IV status due to chronic thromboembolism resulting in significant pulmonary hypertension (systolic pulmonary pressure = 55 mmHg). A new transoesophageal examination was performed at admission and it revealed two pedicled, mobile, ‘myxoma-like’ masses in the right atrium, one larger (21 × 20 mm) attached to the free wall and one smaller (12 × 4 mm) attached to the mid portion of interatrial septum (Figure , B, D). These masses did not cause any extra symptoms and despite partial occlusion of the tricuspid valve by the larger mass, no functional stenosis was observed with a mean gradient of less than 1 mmHg on the valve. In order to avoid any further mass-originated embolic events and any further mass-related complications, surgical removal was suggested and the operation was scheduled within the same admission.\nAt her admission, the physical examination revealed normal first (S1) and second heart sound (S2), with no murmur; normal findings from the auscultation of the lungs and bilateral pedal oedema.\nThe operation was performed under general anaesthesia with a median sternotomy and cannulation of the ascending aorta, inferior and superior vena cava. The right atrium was vertically opened and surprisingly revealed the presence of only the smaller mass attached to the interatrial septum, which was removed along with its pedicle. Despite careful and meticulous visual and manual search of the right atrium, right ventricle (RV) and main PA, the larger mass could not be identified. A repeated intraoperative transoesophageal echocardiography (TOE) examination confirmed the absence of the second larger mass and as it was considered to have embolized into the pulmonary arteries. Persistent suction was applied into the right ventricle between the trabeculae and into the main PA in an attempt to capture and remove the mass. Lastly, the right atrium was closed and cardiopulmonary bypass was discontinued for clinical and hemodynamic evaluation of the patient. Since the patient was stable with satisfied oxygenation without any need of inotropic support, she was transferred to the intensive care unit (ICU). Unfortunately, two hours later she developed cardiogenic shock, with severe hypoxaemia and a bedside transthoracic echocardiography demonstrated signs of acute right ventricular failure and increased systolic PA pressure. As these findings were in keeping with acute pulmonary embolism, an emergent pulmonary computed tomography (CT) angiogram was performed and it revealed a large mass occluding the left PA (Figure ). The patient was retransferred to the operation room under massive inotropic support for emergent surgical embolectomy. After CBP, the main PA was incised longitudinally across its entire length and a 21 × 20 mm mass was removed from the initial part of the left PA using LeGardin graspers and suctioning. Remarkably, the RV showed a significant, dramatic recovery and the patient demonstrated immediate hemodynamic improvement allowing the surgeons to transfer her back to the ICU. After this intervention, the patient had an uneventful further hospitalization period without extra oxygen and inotropic support need, improvement of her symptoms and the pedal oedema by the physical examination. Histopathologic examination of these two masses confirmed the diagnosis of multiple cardiac myxomas (Figure ). |
A 54-year-old female with no significant past medical history presented to emergency room with complaints of nausea and dizziness. As per the family, the patient was in a normal state of health and had full functional ability 5 months prior. Thereafter, she gradually began to lose her balance and started developing short-term memory loss along with occasional episodes of urinary incontinence. The memory loss became progressively worse over the course of a few months to the point that she would rely on family members to answer questions and remind her of previous events. During questioning, it was evident that the patient would attempt to think or elaborate her thought process but was unable to recall and explain recent events. As per her family, the patient had also been increasingly losing her balance which had resulted in a progressively increased number of falls and injuries over the past 5 months. She was initially evaluated at different faculties at the initial onset of symptoms but was discharged after neurologic workup was otherwise unremarkable. The patient however continued to experience these symptoms which resulted in her losing her job and was unable to care for herself. Upon this presentation, the family elaborated that for the past 1 day she had been experiencing dizziness and episodes of vomiting.\nOn examination, her vital signs revealed a blood pressure of 143/80 mm Hg, pulse of 87/min, respiratory rate of 18/min, and temperature of 97.8 °F. On general examination, the patient was alert and oriented. Neurological examination showed a short-term memory loss (unable to repeat three simple words), lower extremity muscle wasting, and unsteady gait; however, there were normal power and normal reflexes, plantar responses were flexor bilaterally, and cranial nerves two to twelve were grossly intact. As mentioned, the patient had a wide based gait and relied on a cane for ambulation. Hematological, biochemical parameters, and chest radiography were within normal limits.\nAs the patient had clinical picture of NPH, a head CT () was done which demonstrated an increase in the size of ventricles with a possibility of either atrophy or non-obstructing hydrocephalus. Further investigations of infectious causes and etiologies were ruled out. Laboratory results revealed a positive HIV type 1 test, with CD4 count of 74 and viral load of 453,310. Once the patient and her family members were informed regarding her HIV status, it became clear that the patient was in fact diagnosed with HIV in the past. Although she was informed in the past, she had remained in a denial state and had refused therapy for several years. Brain MRI () was conducted and demonstrated findings of ventriculomegaly and multiple supratentorial and infratentorial foci of abnormal T2 white matter hyperintensity. A lumbar puncture was done with an opening CSF pressure of 12 cm H2O. No white cell or red cells were present. Protein and glucose concentrations were 30 and 62 mg/dL, respectively. Syphilis serology in both blood and CSF was also negative. Molecular analysis (PCR) for infectious etiologies in CSF showed an absence of cytomegalovirus, varicella-zoster, herpes simplex type 1 and type 2, Epstein-Barr, human herpes 6 and JC viruses, cryptococcus and tuberculosis. The patient’s gait was reassessed 72 h after the lumbar puncture and showed mild improvement. She was assessed by an HIV specialist and decision was made by the patient’s caregivers to initiate highly active retroviral therapy (HARRT therapy). At this time, patient was stable for discharge with further workup and management as outpatient by the HIV specialist and neurologist. |
A 56-year-old woman presented to our oncology department in May 2018 with a painful erythematous rash with multiple blisters covering the upper part of her back toward the neck. In January 2018 she had been diagnosed with breast cancer and was in the middle of her adjuvant chemotherapy after partial mastectomy. She was on day 8 after the first course of docetaxel treatment (90 mg/m2 in a three-week regimen), which was given after three courses of prior treatments with epirubicin and cyclophosphamide. Earlier on day 3, she started to feel irritation and pain over her upper back and neck. An extensive rash over the area was noticed on day 5 with increasing pain. She visited the emergency room on day 6 and was found neutropenic. Blisters appeared over the erythematous area later that day. The patient suffered from an intensive pain despite maximal dosages of paracetamol and ibruprofen day 8, she showed up in our clinic and presented a confluent, elevated, burning erythema distributed on previously sunburned areas, the neck and the upper back, strictly excluding areas previously covered with clothes and hair. The appearance reminded of an acute sunburn. However, she had not been exposed to the sun for the last weeks. The patient was admitted to the infectious diseases ward for assessment because herpes infection was suspected. One of the blisters was punctured and PCR tests for herpes simplex virus type I (HSV I), herpes simplex virus type II (HSV II) and varicella zoster virus (VZV) were performed. The patient had fever up to 38.3 degree Celsius and the treatment with valaciclovir was initiated. However, the clinical picture was atypical since the erythema was covering several dermatomes while distinctly sparing the parts under her bra straps, rising suspicion of photo toxicity. The patient had not been sun exposed during the period of chemotherapy, but she had undertaken a trip to Vietnam for two weeks 5 months before the docetaxel treatment. Her upper back and neck was sunburned at that time, but to a much lighter degree without skin bullae. The rash progressed with a vesicular erythema and some large blisters followed by shallow erosions. At day 9, the patient was sent for dermatological consultation. A skin biopsy from the back was taken and it revealed a blistering dermatitis with interface changes including basal vacuolar degeneration, apoptotic keratinocytes at all levels, engagement of a sweat gland duct, intraepithelial vesiculation and partial epidermal necrosis. A subepithelial lymphocytic infiltrate with few eosinophils and neutrophils was seen. These findings were similar to erythema multiforme. Immunohistochemistry with antibodies against HSV1/2 and VZV were negative, which were also confirmed by the negative PCR results by serology. Thus, we diagnosed a severe docetaxel-induced photo toxicity which was recalled 5 months after the initial sunburn. The medication of valaciclovir was stopped after two days of administration.\nThe recall eruption was much more intense than the initial sunburn. Both the clinical (Fig. ) and pathological (Fig. ) presentations showed similarities to HSV/VZV infections. Although the erythema was clinically assessed as grade 4, it resolved relatively quickly after 3 days of application with topical corticosteroids (betametasone cream once a day). The erythema faded with a superficial desquamation leaving a hypo- and hyperpigmentation on day 11. Due to this severe skin toxicity, the patient was not continued with docetaxel treatment. Instead, weekly paclitaxel was administrated. Six doses of paclitaxel were given successfully, and no more skin toxicity was observed. The adjuvant chemotherapy was thus completed according to the planed time schedule, without any delay.\nPhoto-recall phenomenon is a phototoxic eruption occurring on areas of previous ultraviolet-induced solar erythema following a systemic administration of a chemotherapeutic drug. It has been mostly described with methotrexate but remains rare with other antineoplastic drugs. Few cases have been documented with gemcitabine, etoposide, and cyclophosphamide [, , ], but even more rarely cases have been reported with taxane based regimens [, , ]. Droitcourt et al described a case of docetaxel-induced photo-recall skin rash on a woman treated for a non-small-cell lung cancer []. He et al. reported a case induced by paclitaxel [].\nApparently, docetaxel seems to be more potent to recall the photo toxicity than other cytotoxic drugs the patient has received. Prior to docetaxel, our patient had already received three repeated courses of epirubicin in combination with cyclophosphamide relatively shortly after the initial sunburn, but no skin toxicity was developed. In addition, she has also received 6 courses of paclitaxel treatment after the docetaxel course, without any occurrence of skin toxicity. Furthermore, the recall occurred 5 months after the initial sun damage, which was far longer than that in other reports (1–8 days), but within the time period of 2–7 days after administration of the triggering drug.\nThe physiopathology of photo-recall phenomenon is still unclear. One hypothesis was that after a sub-erythemal exposure to UV light, the UV-dependent pro-inflammatory factors might be upregulated by the triggering drug to reach the erythemal threshold. However, this theory can hardly explain the recall reported in our patient which occurred 5 months after the initial sun damage. Another theory is that some skin-resident cells remember previous damage, and memory T cells that were primed during the original inflammation may persist at high levels during the asymptomatic phase and evolve into long-lived memory cells that can cross-react with unrelated drug []. This theory may explain our case. However, these hypotheses need to be further confirmed in future research.\nTo our knowledge, this is the first reported case of a breast cancer patient with a rare but severe photo toxicity recalled by an adjuvant docetaxel treatment 5 month after the initial sunburn. We believe this rare and peculiar type of skin eruption induced by docetaxel is worth being recognized by the oncologists as well as dermatologists and pathologists. Paclitaxel can be the drug of option after docetaxel recalled photo toxicity. |
A man aged 35, works as a laborer, admitted because of problems with urination as a feeling of pressure and pain during micturition, which persist for the last few months. In the regional medical institution treated as acute prostatitis. Good general condition, and the physical examination findings orderly. The laboratory findings of elevated CRP: 145mg / l, while the other parameters were of the reference values. On the ultrasound examination of the urinary tract, in the bladder was revealed tumor change the largest diameter to 70mm, which covers the front wall of the bladder, and whose origin cannot be determined. Computed tomography revealed that in the pelvic there was edge well vascularised formation that covers the front wall of the bladder, with visible signs of infiltration of the musculature of the anterior abdominal wall measuring 75x80mm, which was seen as a change of bladder tumor (, , ). On uretrocistscopia the capacity of bladder easy decreased and on the front wall of the urinary bladder more over the left side of the suspicious bullous edema and external signs of infiltratation of the wall of the bladder. Colonoscopically, except for internal hemorrhoidal plexus other findings were normal.\nEndoscopic transurethral revealed on the front wall of the bladder solid tumor diameter 40x50x10mm entering the lumen of the bladder, and mucosa above the change is largely intact and marginally more toward the back wall bullous and cerebriform. It was taken biopsy of change which as microscopic showed the elements of proliferative lesion binders most by type of fibromatosis.\nThe patient underwent resection of pelvic tumors with partial cystectomy and Right Hand iliac lymphadenectomy. Intraoperatively, dilated small bowel diameter twice large compared to the usual size was seen and identified tumor of pelvic diameter 70x80mm, which invaded the anterior wall of the bladder, m.rectus abdominis from the right and parietal peritoneum anterior lateral abdominal wall, rest on the iliac vessels on the right hand and right ureter, but did not infiltrate them. Tumor engage omentum and convolutions of the ileum without involvement of the small and large intestines. Right iliac lymph nodes in ex tempore PH survey pointed to inflammation and necrosis. The operation lasted 120 min. It was accessed to tumor tissue with lower median laparotomy. In the postoperative period there came to subocclusion which was resolved with nasogastric suction 5 days with stimulation peristalsis neostigmine. Postoperatively, until the patient’s discharge from the hospital, the patient was on parenteral antibiotic therapy as well as on the therapy with low molecular heparin. The patient was discharged on the twelfth postoperative day in good general condition.\nA definitive histological diagnosis was retroperitoneal fibromatosis with purulent inflammation of local adipose tissue and chronic lymphadenitis.\nFive months after the operative procedure the patient feels well and has no problems. In laboratory findings CRP: 2.85 mg / l, MRI of the abdomen and pelvis with no signs of recurrence of the underlying disease (). |
A 64-year-old woman presented with nipple discharge from right breast for 3 months. On physical examination, a palpable mass was noted in the right breast without other remarkable findings. She had no remarkable medical history or familial history. Diagnostic mammogram revealed a 5.4-cm-sized mass with microcalcification in the palpable area in the right upper medial portion of the breast. In magnification view, the parenchymal distortion measured about 6.3 cm in maximal diameter including grouped coarse heterogeneous calcification. In diagnostic ultrasound, a 3-cm-sized heterogeneous area including calcification in the inner part was observed 3 cm from the nipple in the right upper medial 2 o’clock direction and five core needle biopsies were performed. The pathologic diagnosis of biopsy was ductal carcinoma in situ (DCIS) with a suspicious area of invasion showing mucinous differentiation. Breast magnetic resonance imaging showed right nipple retraction without pathologic lymph node, and skeletal metastasis was not observed in whole body bone scan. The patient underwent total mastectomy and sentinel lymph node dissection of the right breast.\nThe surgical specimen was sent to the Department of Pathology. On gross examination, the cut surface revealed a gelatinous gray white mass (2.2×2.0 cm). On histologic examination, the tumor with expanding margin was observed in the low-power view (). The tumor cell clusters were floating in the mucin pool and the cell density was higher in the periphery than in the center (). In the high-power view, the tumor cell cluster floating in the mucin pool showed nuclear atypia suitable for nuclear grade 3 and the mitotic count was 14 in 10 high power fields. Many tumor cells were seen as signet ring cells with the tumor nucleus pushed into a corner by abundant intracellular mucin (). DCIS was observed in the periphery of the expanding invasive nodule, comprising 60% of invasive tumor area (). The DCIS component showed a significantly high nuclear grade and signet ring cell differentiation, but extracellular mucin was not observed (). Serial immunohistochemical staining results showed that tumor cells were negative for ER () and PR () and positive for HER-2 (3+) (), with a Ki-67 labeling index of about 30%. In addition, tumor cells were positive for mammaglobin (), gross cystic disease fluid protein-15 (GCDFP-15) (), E-cadherin, and MUC-1, and tumor mucin was positive for Alcian blue and mucicarmine. A total of 12 axillary lymph nodes were evaluated, but no metastasis was noted. The patient has been followed on an outpatient basis after surgery and to date, there is no evidence of recurrence or metastasis. |
A 55-year-old gentleman presented to the surgical emergency with complaints of progressively increasing pain and swelling on the left flank, high grade fever and decreased urine output for the last 15 days. He also had a history of extreme weakness and feeling of being unwell for 1 month. He was a known diabetic on oral hypoglycemic agents. On examination the patient was a thin built, sick looking individual, pale, febrile with tachycardia of 110/min. He was not icteric and had no lymphadenopathy. Abdominal examination showed a large area of abscess over the left flank and back covering an area of around 15 × 10 cm, which showed inflammatory changes and evidence of tenderness and crepitus on palpation. The investigations showed a markedly deranged blood urea (150 mg/dl) and serum creatinine (4 mg/dl) levels. The blood sugar was 300 mg/dl but there was no evidence of ketonuria at time of admission. An ultrasound (USG) of the abdomen was performed and the left kidney showed evidence of air in the parenchyma, which was extending up to the parietes, suggestive of emphysematous pyelonephritis. Computer tomography (CT) scan was done which showed the left-sided kidney to be enlarged with multiple air lucencies extending up to the skin and subcutaneous tissue [Figures and ]. Since the inflammatory material had already spread to the parietes, and due to poor general condition of the patient, a decision for incision and drainage was taken under local anesthesia. The wound was left open after draining the gas and the purulent material that were reaching up to the level of the left kidney. The patient was started on broad spectrum antibiotics and the pus sent for both aerobic and anaerobic culture. The anaerobic culture was negative. The gram stain showed gram negative cocci. The pus came out positive for Klebsiella, which was sensitive to the antibiotics already being used. The patient was started on human insulin on sliding scale to control the deranged sugar profile. The patient started responding to this treatment within the next 24 h with fever subsiding and there was improvement in the general well being of the patient. The patient's blood urea and creatinine levels progressively decreased to stabilize at urea (60 mg/dl) and creatinine (2 mg/dl). The patient's urine output was adequate at all. times during the admission period. The patient was subsequently discharged after a period of 3 weeks with minimal discharge from the surgical site and is doing well after 6 months of follow-up. |
The case was a 53-year-old single male who had a hemorrhagic right hemisphere stroke 6 months prior to the study. He lived in an apartment on the third-floor with his wife and son, and was a driver in a private company. Prior to onset of stroke, he participated in various recreational activities, such as cycling and football. He was hypertensive and used to smoke. He had a relatively supportive family and was transferred to the hospital immediately after the stroke and received preliminary medical care. He stayed in the intensive care unit (ICU) for 3 days and in the neurology ward for 1 week. Then, he was discharged and regularly received physiotherapy services at home. However, he had weakness and imbalance in his left limbs. He could walk short distances with a quad cane; he depended on others for some ADL, such as toileting and dressing. His range of motion was limited due to spasticity and pain and he almost had no functional use in his upper limbs. He participated in this study after completing the written consent form. The current report was conducted 6 months following a comprehensive assessment of his functional status.\nThe comprehensive ICF Core Set of stroke was considered as the main reference in this case study (). Since there are different categories in stroke ICF Core Set that are relevant to the services of all health care providers, such as physicians and nurses, and considering the results of a study by Glässel et al. in 2010, only the categories attributed to the occupational therapy were selected and applied for the patient (). By the documentation template suggested by Escorpize, all the tests, assessments, and interventions, which could be performed by an occupational therapist, were recorded in the ICF Core Set categories for stroke. The occupational therapist identified some difficulties in the patient’s body functions, such as reduction of proprioception function (b260) and mobility of joint functions (b710) in upper and lower joints of the affected body side. Also, the patient developed an increase of muscle tone function (b735) in one side of the body. Imbalance and incoordination were observed and recorded by the evaluation of inventory movement function (b755) and control of voluntary movement functions (b760). The occupational therapist diagnosed some limitations in most mobility domain categories, such as hand and arm use (d455), walking (d450), and moving around under different circumstances (d460). In addition, the patient had some problems in the self-care domain, such as toileting (d530), washing (d510), and dressing (d540). Evaluation of the code d850 remunerative employment revealed that the patient could not drive (d4751: Driving motorized vehicles) and go back to work as a driver at that time. He was capable of performing some leisure activities, such as crosswords and watching TV; however, his capability in performing other activities, such as cycling, was completely limited.\nThe results of stroke ICF Core Set categories showed different limitations in mobility and self-care of the patient. These limitations were mainly associated with neurological damages (motor-sensory dysfunction) caused by the stroke and the inability to functionally use the affected side of the body that limited the participation in leisure and work activities.\nAs soon as all ICF Core Set categories were graded based on the qualifiers, a comprehensive definition as ICF categorical profile was provided regarding the functional status of the patient (). The patient’s family (e310) and the therapist (e355) were supportive and facilitators. Since the patient used a quad cane, the code e120 was also added as an environmental facilitator. The patient was motivated to take part in the occupational therapy program, while he had accepted his current conditions.\nThe ICF Assessment Sheet () used in this stage relies on the clinical reasoning of the therapist. The sheet, which provides a comprehensive definition about the functional conditions of the patient, constitutes 2 parts: The upper part indicates complications experienced by the patient and is completed via a routine interview with the patient. In the lower part of the sheet, which reflexes the viewpoint of the therapist, the reasons and causes of the experienced complications are transferred into the ICF categories. Hence, finding an association between patient’s complications and the causing factors is facilitated; for example, the factors that caused the patient’s mobility complications (the upper part of the sheet) can be identified via the ICF lists completed by the therapist based on his/her examinations and assessment of the patient (for example, tone of muscle (b735) and mobility of joint function (b710). The relationship is then shown by the connecting lines ().\nThe prognosis was favorable, comparing the improvement observed in neurological conditions (stroke recovery) of the patient and other studies with hemorrhagic stroke ().\nEvaluation (6 months following stroke)\nThe goals of the therapeutic program were established by the participation of the patient and by considering different components of ICF. The patient’s goal (PG) was to regain his independence. The long-term goal (LTG) of the patient was to regain his capabilities to drive and go back to work. Short-term goals (STG) are achieved within a short-term cycle by rehabilitation practitioners (). Two short-term goals (STG) were identified for the patient. First, mobility improvement, particularly the ability to walk without a cane and going up and down the stairs; second, improvement in the patient’s capabilities to achieve maximum independence in activities of daily living (ADL), such as toileting and dressing. The goals were entered into the ICF Categorical Profile and ICF Assessment Sheet. The interventional targets were selected according to the goals that were set by talking to the patient. For example, according to the first short-term goal, the selected interventional targets were increased muscle tone, involuntary motor reactions, and reduced muscle strength in the affected side of patient’s body. Such activities as change and maintenance of different body positions, including walking and moving under different locations, were also considered as intervention targets. In addition, to facilitate the patient’s mobility, use of assistive devices, such as splint and insole, was also considered as a facilitating environmental factor. A goal value was set for each of the first and second short-term goals expected to be met by the end of the 1- month intervention.\nSome of the interventions provided by the occupational therapist are shown in the documentation template (). A set of occupational therapy interventions is provided for each ICF category. According to the set short-term goals, various interventions were used to improve mobility and ADL. The occupational therapist used occupation-based therapeutic and neurophysiological approaches towards improvement in designated goals. Accordingly, some therapeutic techniques, such as using thermal modalities, weight bearing, active, passive, and stretching movements, muscle strengthening, and different balance exercises were performed to achieve the goals. Some environmental adaptations were also conducted to induce more independence in the patient regarding his daily activities; for example, embedding a handhold in the bathroom to facilitate sitting and standing, using bath chair, and removing the barriers to bath entrance.\nThe patient’s functional conditions were reassessed by the end of the 1 month- occupational therapy program, and the results were transferred into the documentation template (). Based on the first short-term goal, t spasticity reduced in knee extensors and ankle plantar flexors, and muscle strength increased in some muscles, such as knee flexors. In addition, the balance reactions were improved in the patient. Such changes improved the patient’s balance and mobility. He can now walk without a cane, but he still uses it when walking far distances and going to unfamiliar places. He can also go up and down the stairs without help. The patient can now wear upper and lower clothes independently by learning the adaptive and compensatory (one-handed technique) methods although he still has problems in some activities, such as fastening a belt, which is due to motor problems in the upper limb. His independence and balance improved following the installation of some devices, such as embedding a handhold in the toilet and using bath chair and non-slippery mats in the toilet. After the patient completed the occupational therapy program, he was reassessed for the interventional ICF categories; the results are demonstrated in the ICF evaluation display (). |
An eighty-two-year old female presented to the urology outpatient clinic with a short history of fecaluria, pneumaturia, and passage of urine per rectum. She had an indwelling urethral catheter inserted, because of the total urinary incontinence, for a period of over eight years. This catheter was last changed twelve weeks prior to her visit to the urology clinic by her district nurse. The patient had positive MSUs in the year prior to her admission demonstrating mixed growth of enteric organisms, but no prior febrile urinary tract infections were reported.\nShe received radical radiotherapy alone for a muscle-invasive bladder tumor (stage T2b) in 1991 and underwent abdominal exploration for drainage of an appendicular abscess when she was in her early twenties. The patient had a background history of cerebrovascular disease, hypertension and ischaemic heart disease. She was felt to be an unsuitable candidate for radical surgery for her invasive bladder cancer due to these comorbidities. The decision to manage her incontinence with an indwelling urethral catheter was made having discussed available treatment options with the patient. A long-term indwelling catheter was felt to be the most appropriate option for her given her other extensive comorbidities. She did not have urodynamic testing prior to urethral catheterization. The exchanges of the catheter were being carried out every three months in the community and there had been no reported difficulty with her previous catheter changes. The patient was not undergoing routine cystoscopic surveillance of her bladder.\nOn physical examination she was generally unwell with low-grade fever and pallor. Her abdomen was soft, mildly tender with no palpable masses and no signs of peritonitis. Her urethral catheter drained feculent material mixed with urine.\nLaboratory investigations showed a low hemoglobin of 7.8 gm/dL, white cell count of 17 109/L with a C reactive protein of 233. Magnetic resonance images (MRIs) of the abdomen and pelvis were performed urgently and were reviewed by a senior consultant radiologist and his impression was that those images confirmed the presence of the inflated balloon of the indwelling urethral catheter in the lumen of a bowel segment (Figures and ).\nIn enterovesical fistulas treatment is undertaken depending on the aetiology, clinical status and general condition of the patient. Enterovesical fistulae seldom close spontaneously []. Given the nature of the pathology in this case and the presence of a foreign body in the fistulous tract (the urethral catheter), it was felt that the only way forward was to perform an urgent laparotomy and removal of the foreign body with excision of the fistulous tract. The situation was explained to the patient and she was consented for laparotomy, with all the risks and potential complications of surgery fully explained including both urinary and faecal diversion. On laparotomy there were severe adhesions, and the catheter balloon was found to have penetrated through the bladder wall and was lying in a terminal ileal segment adherent to bladder wall. Excision of this segment and urinary diversion by fashioning an ileal loop conduit was performed. A Hartmann's procedure was carried out due to the high clinical suspicion of presence of another separate colovesical fistula. The patient was transferred postoperatively to the intensive care unit and was moved afterward to the urology ward for 10 days. She had a smooth postoperative course with no recorded complications and is currently on regular urology outpatient clinic followup. Histology from the resected segment of terminal ileum demonstrated chronic inflammation only with no evidence of tumor recurrence. |
A 35-year-old woman was referred to the outpatient’s clinic with a long-standing nonunion, persistent pain on a functionless hand and wrist, and a severe angular deformity of her left forearm. Eight years ago, the patient fell from the horse back and sustained a closed bifocal fracture of the radius (radial shaft and distal radius) and a fracture of the ulnar shaft (), initially treated with open reduction and internal fixation (plates and screws for the forearm diaphyseal fractures and a palmar plate for the distal radius fracture) (). According to past medical records, 3 months after the initial procedure, she presented with persistent forearm pain and radiological signs of hardware failure and loosening. She underwent surgical debridement and revision of the osteosynthesis applying new plates and screws enhanced by bone grafting. Intraoperative sample cultures were obtained and one of them proved positive for Escherichia coli. The patient was subsequently given intravenous antibiotics for 3 weeks.\nOne year later, due to nonunion and presence of a bone gap, she underwent reoperation with “enhancement of the union potential” use of eptotermin alfa, bone morphogenetic proteins, and autologous iliac bone grafting, which were proved effective for the radial shaft fracture. Five months later, the ulnar nonunion and bone gap were treated with distraction osteogenesis with the use of an external fixator. Enhancements with two more surgical interventions with corticocancellous bone grafting were done with 4 months interval. The external fixation remained for 3 weeks after the last procedure, until clinical and radiological signs of union. The patient experienced a period of 4 years free of symptoms with no significant functional impairment. However, eventually she gradually established pain, swelling and severe deformity of her left forearm with magnetic resonance imaging and Ga 67 bone scan raising suspicion for infection. She was admitted for surgical debridement, hardware removal, and intravenous and then oral antibiotic treatment (ciprofloxacin and rifampicin) while intraoperative culture samples were negative. The patient’s medical and family history is clear, while she referred smoking from her social history.\nAt her presentation in our department, she had a significant deformity with angulation of the forearm, lack of motion with pronation and supination <10°, and severe functional loss, obliged to use a splint to use her hand. Elbow flexion was restricted to 20° and wrist range of motion was 20–0–20° (). Lack of swelling, erythema, and increased local temperature, or any other clinical sign of infection was detected. Laboratory tests were negative for infection (white blood cell: 6800, C-reactive protein: 0.2, and erythrocyte sedimentation rate: 7 mm).\nAt the operating theater, after an extensive and demanding approach at the site of the non-union arduous approach of the nonunion, debridement was done along with excision of all non-viable bone segments, and shortening of about 2.5 cm so as to allow reduction without excessive stretching of the nerves and vessels. Internal fixation with reconstruction plates was carried out, augmented with posterior iliac crest autograft. Intraoperative cultures of soft tissues and bone were obtained to rule out infection (). The patient had a short hospital stay, the culture results were negative and she was discharged wearing a long arm splint for 6 weeks. The patient was followed up at 1, 2, 4, and 7 months and 1 year postoperatively with clinical evaluation and radiological examination (, ). At last follow-up supination and pronation of the forearm were approximately 100°. Elbow flexion was significantly improved at more than 90° and range of motion of the wrist was 50–0–50. |
The patient is a 40-year-old female who presented to the emergency department with a complaint of left knee pain and swelling from falling down the stairs. She had no significant past medical or surgical history, nor a history of similar trauma. Clinical examination showed swelling of the knee and tenderness over the lateral aspect of the proximal tibia. Knee ligaments were stable and neurovascular status was intact. X-ray images showed a Schatzker Type III tibial plateau fracture. Computed tomography and magnetic resonance imaging better defined the fracture pattern and were used to rule out any concomitant knee pathologies (Figure ). Surgery was performed after signed consent. The patient was informed that data concerning her case would be submitted for publication. Her consent was obtained.\nSurgical technique\nFollowing a careful evaluation of the pre-operative CT scan, we planned to achieve a combined intraoperative arthroscopic- and fluoroscopic-guided reduction of the articular depression through a lateral cortical window. The fracture was fixed using a minimally invasive fixation technique. The patient was supine on a Jackson table under general anesthesia. She received 2 g of intravenous cefazolin for infection prophylaxis. Non-sterile tourniquet was applied. The right knee was prepared, draped, and flexed to 90 degrees using foot support and a lateral thigh kidney shape support. A standard lateral and medial anterior arthroscopic portals were made. Diagnostic knee arthroscopy was conducted and the fracture was assessed. No other knee pathologies were identified. The inflow pressure was kept to a minimum throughout the case to decrease the risk of fluid extravasation and reduce the subsequent potential for increased compartment pressure. An anterior cruciate ligament (ACL) guide was used to place a drill-tipped guide pin in the center of the depressed fragment through a small incision in the proximal anterolateral aspect of the tibia (Figure ).\nIntraoperative anterior-posterior (AP) and lateral fluoroscopy were used to confirm that the guidewire was drilled into the desired fragment with the appropriate trajectory depending on the direction chosen on pre-operative CT. A size 8 coring reamer was used to circumferentially open the tibial cortex while as little bone as possible was removed. A bone impactor was used to elevate the depressed fragment. The anatomical reduction was obtained and confirmed by arthroscopy and fluoroscopy (Figure ).\nThe resulting metaphyseal defect was grafted using bone allograft and a size 10 bioabsorbable screw was then introduced through the reamed bone tunnel to support the impacted bone below the reduced articular surface. In addition, two percutaneous 3.5 mm cortical screws were introduced subchondral from lateral to medial. Final fluoroscopic images confirmed the anatomic reduction of the fracture. The skin was closed and a dry dressing applied. The patient was allowed to have an immediate range of motion as tolerated. Partial weight-bearing was recommended initially and full weight-bearing was allowed six weeks after surgery. The postoperative course was uneventful. The patient had recovered full range of motion and the wounds were barely visible. One-year X-ray showed healed fracture without any evidence of displacement or subsidence (Figure ). |
Written informed consent was obtained to report the case. A 34-year-old G2P1 pregnant woman diagnosed with AS presented at the obstetric outpatient clinic at 18 weeks of gestation. She experienced back pain when she was 25 years old; these symptoms made walking difficult during her first pregnancy at 31 years old. Following her first vaginal delivery, she was able to walk although pain persisted to a lesser degree than during pregnancy. Radiography revealed osteosclerosis of the posterior surface of cervical vertebrae and osteoarthritis of the right hip with joint space narrowing (Figure ). The initial diagnosis of AS was made at 33 years of age.\nPain was managed by the administration of nonsteroidal anti-inflammatory drugs (NSAIDs) and acetaminophen. After the present pregnancy was diagnosed, only acetaminophen continued to be administered, but pain increased during the present pregnancy. She complained of right hip pain, with a visual analog scale (VAS) score of 7/10. A dosage of 5 mg of oral prednisolone was administered daily from 18 weeks of gestation, and thereafter, the symptoms temporarily improved to 0/10 on the VAS. Unfortunately, the symptoms relapsed at 31 weeks of gestation. At 32 weeks of gestation, she complained of restricted neck mobility with difficulty gargling, restricted lumbar mobility that caused difficulties in bending her back, and restricted right hip joint mobility with a limitation of 10º of abduction, which also affected internal and external hip rotation. Peripartum management was discussed and planned by obstetricians and anesthesiologists. Given that only hip abduction and rotation, but not flexion, were limited, it was considered that an attempt of vaginal delivery would be possible when spontaneous labor occurred. However, if an emergency cesarean section (CS) was necessary during labor owing to other obstetric complications such as nonreassuring fetal status, special anesthesia management may be required because of the possible failure of spinal anesthesia due to calcified spinal ligaments and difficult tracheal intubation and airway management for general anesthesia. In such cases, the preparation of awake fiberoptic intubation or supraglottic airway device insertion should be considered.\nAt 38 weeks of gestation, the patient was admitted to our hospital because of membrane rupture. Labor analgesia was not provided. Oxytocin administration was required due to prolonged second stage of labor. Vacuum delivery with episiotomy left of the midline, which was opposite to the restricted right hip joint, was performed due to fetal bradycardia at birth. A healthy baby with a weight of 3358 g was successfully delivered. Both mother and neonate had a good postpartum course. Prednisolone and NSAIDs were started after pregnancy. |
A 43-year-old male was brought to the emergency department due to inattention and transient episodic memory loss. He was relatively healthy, except for taking medications for a headache that developed a year ago. For 2 weeks prior to admission, he showed excessive daytime sleepiness at work and an indifferent mood noticed by his colleague during a conference. One day before admission, the patient got lost going back home from work. It took more than 2 hours to get home, which normally took 30 minutes. On the day of admission, the patient showed transient episodic memory loss of that day. He had intact remote memory. Neurologic examination demonstrated no other lateralizing of focal deficit. His initial Korean mini mental status exam (K-MMSE) score was 27 out of 30, showing a mild impairment in attention. On the following day, K-MMSE score had fallen to 19 out of 30, showing a worsening of attention and mild impairment in memory recall. The electroencephalogram was normal. A cerebral spinal fluid study showed high opening pressure of 330 mmH2O, but was acellular showing a nonspecific finding. Brain MRI showed a DAVF in right lateral sinus with a bilateral thalamic venous infarction (). The following cerebral angiography confirmed a right transverse sigmoid DAVF with a feeding artery of the right occipital artery and left posterior meningeal artery (). Endovascular embolization was performed three times due to a highly stenotic, compartmentalized sinus and small, tortuous vessels (). After the third intervention, there was a decrease in extent of venous congestion in the bilateral thalami. Follow up Brain MRI taken one month later after embolization showed a remaining DAVF, though with a decreased signal change in the bilateral thalami. At the time of discharge, the follow-up K-MMSE score showed improvement with a score of 27 out of 30 with improvement in attention and memory recall.\nThe fourth embolization was performed 5 months after initial endovascular therapy. Since there were some thrombus at the sagittal sinus and straight sinus, we decided to maintain warfarin anticoagulation. At the time of discharge, his mental status and mental function was well preserved and the patient had begun his normal daily life. Follow up cerebral angiography performed 1 year later showed no evidence of a residual AVF. |
A 32-year-old, previously healthy, African American man presented to an emergency department 45 minutes after the acute onset of left facial droop and right-sided weakness (Fig. ). A thorough history confirmed an episode 1-week prior, during which he developed sudden onset of dizziness associated with nausea and vomiting that resolved within hours. He denied any past medical or surgical history and was taking no medications. He has no family history of tumors. In the emergency room, his vital signs were within normal limits. His physical examination was significant for a left facial droop and right hemiparesis. Auscultation of his chest revealed a regular rate and rhythm with no appreciable murmur. No additional significant findings were noted. Stroke protocol was initiated. A chest X-ray was normal and an electrocardiogram showed normal sinus rhythm. A head computed tomography (CT) scan was negative for signs of intracranial hemorrhage. He was subsequently started on tissue plasminogen activator (tPA) therapy. Magnetic resonance imaging (MRI) of his brain demonstrated a right basal ganglia infarct and an old left cerebral infarct. A carotid ultrasound was negative. TTE demonstrated a 1 cm by 1 cm mass on the posterior leaflet of the mitral valve with a moderate mitral regurgitation In addition, TTE revealed a questionable mass on the left coronary cusp of the aortic valve. These findings were confirmed with TEE (Fig. ), which verified no sign of endocarditis and no atrial septal defect. A complete hypercoagulable workup was negative. Stroke protocol continued with the working diagnosis of cerebrovascular accident secondary to emboli from the mitral valve mass. Within 24 hours, he regained function of the right side of his body and had complete resolution of symptoms. He was diagnosed as having transient ischemic attack (TIA) and discussion was undertaken regarding surgical excision of his mitral valve mass.\nA median sternotomy was performed and cardiopulmonary bypass was employed via aortic and bicaval cannulation with full anticoagulation. His aorta was cross-clamped and his heart arrested with retrograde cardioplegia. The aortic valve was examined through an ascending aortotomy and all three valve leaflets appeared normal. A left atriotomy was made and the mass was easily identified on the posterior mitral valve leaflet adjacent to the mitral valve annulus (Fig. ). The mass was excised and a frozen section confirmed globular myxoma cells with abundant eosinophilic cytoplasm consistent with myxoma. The valve leaflet was reconstructed with an autologous pericardium patch and the annulus was supported using a running DeVega-type suture. The valve appeared normal and was tested; no regurgitation was noted. His left atrium and aorta were closed. His aorta was unclamped, after aggressive venting and de-airing maneuvers, and his heart returned to normal sinus rhythm with successful weaning from cardiopulmonary bypass. Anticoagulation was reversed with protamine and his chest was closed after placement of drains and pacing wires. At the conclusion of the operation, TEE confirmed appropriate mitral valve function and normal aortic valve with no evidence of a mass and no regurgitation at either location. |
A 3-year-old boy was referred to the King Faisal Specialist Hospital and Research Center (KFSH&RC) from the regional hospital in Al-Ahsa region in the eastern province of Saudi Arabia. The patients experienced recurrent chest infections associated with a persistent wet cough which resulted in four hospitalizations over the last year. The child was born uneventfully after stable pregnancy and had no remarkable background history in the first 2 years of his life apart from infrequent mild upper respiratory tract infections without symptoms, suggestive of a lower respiratory tract infection. Shortly after his second birthday, the boy had the first onset of lower respiratory tract infections and presented to the local hospital with fever, tachypnea, and respiratory distress, significant enough to require ICU admission. He responded partially to the initial regimen with oxygen therapy, systemic antibiotics, and systemic steroid and nebulized bronchodilators during the hospitalization. His cough persisted after he was discharged from the hospital and he soon suffered from additional attacks during which his symptoms worsened. His symptoms included respiratory distress, an aggravated cough, and hypoxia, and required frequent emergency visits and a total of 4 hospitalizations over the course of 1 year. The patient’s parents are non-consanguineous, and there was no history of respiratory infections or symptoms in the family. Upon examination of the patient’s immune system, test results showed normal immunoglobulin levels and normal lymphocyte markers. A computed tomography (CT) scan of the chest (Fig. ) indicated multiple cystic changes within the posterior segment of left lower lobe, which most likely represented bronchiectasis changes. FB ingestion was suspected after radiology; however, congenital lung malformation could not be ruled out. At this point, the clinical and radiological presentation were suggestive of a foreign body had been ingested by the child and was overlooked. Consequently, the child was referred to our facility where we performed flexible bronchoscopy after the boy had been ill for 1 year.\nClinical examination performed at our facility demonstrated a normal oxygen saturation on room air. There was a decrease in air entry in the left lower zone with no adventitious sounds. Initial chest x-ray (Fig. ) showed a significant left lower lobe consolidation with Bronchiectasis. Flexible bronchoscopy was performed under general anesthesia, and the patient was intubated by an endotracheal tube in the operating room. A sphere-shaped foreign body (Fig. ) was identified in the left lower lobe posterior segment. The patient’s parents were certain that the foreign body represented an Orbeez ball.\nAn attempt was made to remove the foreign body using a retrieval basket and a flexible scope. Unfortunately, the foreign body was squashed and fragmented into smaller pieces, which required the removal of each piece using the retrieval basket. Eventually, all fragments were removed successfully. We observed that granulation tissue was causing a significant obstruction of the left lower lobe posterior segment airway, to the extent that we could not pass the scope through it. After reconstructing the pieces of the foreign body, the patient’s parent indicated that it appeared much larger than its original size. The fragmented pieces were reviewed by a pathologist who confirmed the inorganic nature of the foreign body. At an evaluation after 6 months following the removal of the foreign body the child’s symptoms were minimal, with no additional hospitalizations recorded. His chest X-ray demonstrated a persistent bronchiectasis in left lower lobe with interval improvement in the previously noted consolidation (Fig. ). |
A 58-year-old obese middle-aged housewife had a history of insidious pain in her right hip with difficulty in walking for the past three years. She went to a surgical centre for management in her native town and was diagnosed with a right femoral neck fracture. Further workup revealed osteomalacia and vitamin D deficiency. She underwent reduction and fixation with three cannulated cancellous screws for her initial management. Her medical management with calcium supplements and vitamin D was initiated. Restricted weight bearing with the use of crutches was allowed after six weeks and progressed to full weight-bearing within three months on the advice of her local practitioner. Though she was unable to ambulate without support since surgery, the ambulatory status further deteriorated in two months with a progressive left hip pain. She was no longer able to walk for daily routine activities. She presented to us with the relevant concern for treatment.\nShe complained of severe pain in both hips (left more than right) and marked difficulty in doing her routine daily activities including unrestricted utilization of toilet for the last two months. No history of trauma is reported. She was a postmenopausal obese female weighing 106 kilograms for her height (170 cm). Her body mass index (BMI) was 36.6 kg/m2. There was tenderness over bilateral hip joints (left more than right), external rotation deformity, and shortening of the left lower limb. Movement of the left hip joint was extremely painful and was not encouraged. There was pain at extremes of motion of the right hip joint. The patient was able to do straight leg raise on the right side but not on the left side. There was no distal neurovascular deficit in both lower limbs.\nSequential radiographs were available and first radiograph showed fmoral neck fracture in the right lower limb with an initial in-situ fixation by three partially threaded screws (Figure ).\nThe second follow up radiograph at two years of right hip fracture fixation demonstrated no collapse or avascular necrosis of right hip fracture but with varus angulation of both hips with sclerosis along left femoral neck region concentrated along medial aspect with no fracture (Figure ).\nThe follow-up radiograph at three years showed persisting sclerosis along inferomedial aspect neck femur with a neck shaft angle of 115° on the right side and 117° on the left side (Figure ).\nThe radiograph of hip joints at the time of presentation showed displaced femoral neck fracture left side (Figure ) with right sided varus fixed neck femur fracture with screws in situ and radiolucent line along neck femur (Figure ).\nWhole spine radiographs revealed no abnormalities. The erythrocyte sedimentation rate (ESR) was measured 12 mm/hour and C-reactive protein (CRP) was qualitatively negative. Preoperative investigations included normal calcium, phosphorus, alkaline phosphatase, parathyroid hormone and 25-hydroxyvitamin D levels. Informed consent has been obtained from the patient to publish the data.\nShe underwent surgery in September 2018. The options of fixation and arthroplasty were discussed with the patient and planned for a bilateral THA. The goal was fracture management simultaneous with deformity correction (neck-shaft angle). The option of sequential, simultaneous or unilateral THA were discussed with the anaesthesia team and the patient. The patient was American Society of Anaesthesiologist grade 3 and considered for a simultaneous bilateral THA. Preoperative medications included intravenous antibiotic prophylaxis with injection cefuroxime and prophylactic subcutaneous enoxaparin. It was a single-stage bilateral total hip replacement. The surgery was done using combined spinal and epidural anaesthesia. Patient’s left hip operated first in a lateral position with a posterolateral approach. An uncemented total hip replacement was done with Depuy Johnson and Johnson implant (Pinnacle cup with poly-liner and Corail stem with large size 36 mm ceramic femoral head). The duration of surgery was 74 minutes. Right hip procedure followed in the same sitting with position change and re-draping. Upon removal of screws, fibrous union was detected with frank preoperative mobility at the fracture site conforming to the non-union of fracture. A hybrid THA was done with Depuy Johnson and Johnson implant (Pinnacle uncemented cup with poly-liner and cemented Corail stem with large size 36 mm ceramic femoral head). Duration of second surgery was 85 minutes. A preoperative haemoglobin level of 13.4 gm/dl fell to postoperative level of 10.4 gm/dl on day one postoperatively. Two units of packed red blood cells were transfused. Patient was ambulated with walker support from the second postoperative day. Postoperatively, she was given therapeutic doses of vitamin D and calcium supplements. She was discharged on the fifth day of the surgery. There were no immediate soft tissue or postoperative complications.\nPatient at three months follow-up was clinically pain-free and was able to mobilize without support. She had no limb length discrepancy or gait abnormality. Patient at two years follow-up was pain-free and able to walk full weight-bearing without support and carry out her daily activities comfortably. Radiologically, well-positioned replacement components with no evidence of loosening or failure (Figures -) |
A 21 years old male underwent uncemented modular total hip arthroplasty in 2002 for secondary arthritis of the left hip. The implants used were AML A PLUS 11.0 mm femoral stem, 28 mm COCR head and DURALOC acetabular liner and shell 50 mm (Depuy Orthopaedics, Warsaw, USA). He had good outcome in terms of pain relief and restoration of function. He lateral started working as a Policeman.\nIn January 2012, at the age of 31 years, he presented with acute onset pain and inability to move left hip while he was turning in bed. Plain radiograph of the hip confirmed dissociation of the components at the head and neck taper junction without dislocation ( and ). Due to anticipated difficulties in closed reduction, open reduction was performed. It was noted that there was wear of the acetabular liner and wear at the head and neck taper. Implants were reduced and patient was advised revision of total hip prosthesis.\nAs the facility for revision surgery was not available at our centre, he was referred to a higher centre for revision hip surgery. In April 2012, the revision surgery was performed using trochanteric osteotomy when it was noted that the acetabular shell and femoral stem were well fixed but there was failure of taper as seen by wear of the trunnion (), which did not engage the femoral head taper, and there was wear of the acetabular liner (). Similar changes of wear were noted in the dissociated head component. No abnormal reaction or changes in the peri-prosthetic tissues were noted.\nIntra-operatively, attempts at attaching the new femoral head component with 2 different trunion changes failed, hence the full coated femoral stem was removed using proximal femur split. Femoral component was revised in full and acetabular liner was changed as the shell was well fixed.\nTwo years after revision surgery, patient reported no pain and was ambulating without support. The extended femoral osteotomy had healed well. There was evidence of heterotopic ossification resulting in some restriction of movements (). |
A 4 year-old right-hand dominant girl presented to fracture clinic with a painful right thumb. She had initially injured it 10 days previously while on holiday in Spain. A rugby ball had been accidentally kicked at her and she sustained a hyper-extension injury to her right thumb. She was taken to a local hospital where she was assessed and relevant radiographs taken. She was told she had ‘sprained’ her thumb and was prescribed analgesia and advised to be reviewed at a local clinic on her return to the UK. On review in our fracture clinic, the MCPJ of her right thumb was swollen and bruised on examination. The metacarpal head of her thumb was prominent dorsally compared with the left and she was in substantial pain on any attempt to move her thumb. Radiographs were repeated and showed a dorsally dislocated MCPJ of the right thumb ( and ). Due to her age she would not have tolerated a digital ring block and attempted closed reduction in clinic. Therefore she was admitted for manipulation under anaesthesia +/- open reduction. In theatre closed reduction was attempted and although the joint could be relocated it was unstable and was re-dislocating immediately. Open reduction was undertaken. A dorsal approach over the MCPJ was made through the skin and subcutaneous tissues using a longitudinal incision over the centre of the joint down onto the extensor apparatus ( and ). The extensor pollicis longus tendon was dissected and retracted medially. A transverse incision was made in the middle of the joint capsule to expose the joint. The volar plate was found interposed within the joint, preventing successful reduction. This was released from the joint and the MCPJ reduced successfully. Both the radial and ulnar collateral ligaments were intact. The thumb was stable on further passive manipulation, which was confirmed with image intensifier. The joint capsule was closed with sutures, as was the incision. A plaster of Paris thumb spica was applied for 4 weeks. Radiographs confirmed that the joint remained reduced out of plaster ( and ). After this the patient was advised to start actively moving the joint and by eight weeks post-op a good range of movement had been regained and she was discharged at 12 weeks. |
A 74-year-old man presented with epigastric pain for 2 months and was diagnosed with pancreatic body cancer through diagnostic imaging studies. Computed tomography (CT), endoscopic ultrasonography (EUS), and magnetic resonance imaging (MRI) revealed a 3-cm-sized mass located on the body of the pancreas with suspicious perivascular infiltration around the celiac axis and superior mesenteric artery with distal pancreatic duct dilatation. Positron emission tomography (PET)-CT revealed no distant metastatic lesion. Several endoscopic trials had failed to obtain tissue samples for the pathologic conformation. Finally, exploratory laparotomy was performed for tissue diagnosis. No peritoneal metastasis was confirmed after the opening of peritoneum through the midline incision. In opening the lesser sac to evaluate for a pancreatic mass and celiac or SMA invasion, a hard pancreatic mass consistent with malignancy was observed on the body of the pancreas. Dissection of soft tissue around the celiac trunk was performed and the resected tissue was sent to a pathologic laboratory for prompt frozen section biopsy. This soft tissue sample was revealed to free of carcinoma. Anterior radical antegrade modular pancreatosplenectomy was performed without complication or event during operation.\nThe patient's postoperative recovery was uneventful and there was no remarkable postoperative complication. A routine postoperative follow-up imaging using abdomen CT scan was performed on postoperative day 7, in which there was free-fluid collection of 5.5×2.2 cm in size () around the pancreatic resection margin. The pathologic examination confirmed pancreatic ductal adenocarcinoma with lymph node metastasis in 7 out of 19 lymph nodes. Both lymphovascular and perineural invasion were reported. The resection margin was free from carcinoma with a 2.5-cm safety margin, but the tangential margin near the superior mesenteric artery was very close to the malignant cells. The patient discharged on postoperative day 11 without any noticeable complication.\nOne month after discharge, a follow-up abdomen CT scan was performed as baseline study for adjuvant chemoradiation therapy, and revealed that a large amount of fluid (18 cm in diameter was collected in the right subhepatic space (). Therefore, percutaneous drainage for this fluid collection in the subhepatic area was performed through a transhepatic approach. Amylase and lipase levels of the drained fluids were reportedly 3,921 U/L and 6,057 U/L, respectively. The amount of drained fluid and the levels of amylase/lipase decreased significantly 1 week after percutaneous drainage. The percutaneous drainage catheter was removed, as usual; however, after removal of the catheter, the patient presented with severe abdominal pain around the catheter removal site and systolic blood pressure fell to below 60 mmHg. After managing the shock, an emergency CT scan was performed, in which a large, newly developed hematoma with active extravasation of contrast material and severe tearing of the right liver parenchyma was noted, and total collapse of the intrahepatic inferior vena cava due to subcapsular hematoma (). Emergency hepatic angiography was performed to identify the bleeding focus and reveled active extravasations of contrast from branches of the right hepatic artery, however, there was no evidence of pseudo-aneurysm formation at the hepatic artery or other major arteries. Arterial embolization was performed promptly to stop hepatic bleeding. Liver enzymes had dramatically increased to more than 10,000 IU/L after embolization of the hepatic artery. Despite of vigorous supportive management in the intensive care unit, he died due to liver failure and cardiovascular complications at 4 days after the bleeding event. |
A 64 years old man was referred to our oncology clinic, with a large ulcerated lesion (12 cm in diameter) in his vertex. He had the lesion for 15 years. The patient had already received two cycles of superficial X-ray radiotherapy in childhood due to Tinea Capitis infection. In the 50s and 60s, superficial X-ray radiotherapy was considered as a standard treatment for fungal infections of the head that were resistant to conventional medications and was used in the treatment of this common infection till the evolution in the medical treatment of fungal infections in the mid-20th century. Our patient suffered from a scaling patch in the vertex with pruritus and dryness sensation during his adulthood and later in life. The patch became ulcerative with an infected appearance in the last 15 years. He mentioned that the ulcer has got larger and he has been experiencing frequent bleeding episodes from the lesion. In the last 10 years, he received routine treatments for BCC of the skin including topical 5FU, three extensive surgeries with the use of skin flap in two out of three surgeries and two cycles of radiotherapy with linear accelerating (LINAC) device. The first cycle of radiotherapy was performed using 8 Mega electron volt (MeV) with a total dose of 50 Grays in 20 fractions, and the second cycle was performed after four years from the first cycle in 20 sessions using X-ray 6 MeV with the final dose of 60 Grays. He also underwent 6 courses of Cisplatin-based chemotherapy including Cisplatin 45 mg day (1-3) and 5FU 900 mg day (1-3) in the past year. Chemotherapy resulted in a slight initial response but had no long-term response. The patient was referred to our oncology clinic for further treatment. We decided to administer cetuximab (700 mg loading dose based on body surface area calculation and weekly 430 mg). There was not any complication in treatment period within two months after initiation of chemotherapy, resolution of suppurative discharge and bleeding from the lesion was observed and after continuation of chemotherapy for 6 months, the inflammation and ulcer subsided and the primary lesion turned into a somewhat dry and almost asymptomatic patch. The patient was referred to a plastic surgeon to perform curative surgery for lesion excision and reconstruction of the lesion site. Figure represents the case. |
An active 48-year-old man presented at our Orthopaedic Department with a history of 10 months of pain and swelling in the left ankle. His symptoms were exacerbated by weight bearing and athletic activity. His medical record showed no injuries.\nOn clinical examination, the ankle joint presented with swelling and pain that was exacerbated by motion. He had a limited range of motion with dorsiflexion of 0 to 20° and plantar flexion of 5°, whereas muscle strength was normal.\nSerial radiographs of the ankle joint showed lesions in the anterior and posterior compartments with an increase in number and dimension (). Moreover, MRI showed multiple lesions in all compartments and synovial hyperplasia ().\nThe patient underwent ankle arthroscopy under spinal anaesthesia and the use of tourniquet. The patient was treated by standard, first anterior (medial and lateral) and then posterior (medial and lateral) arthroscopic approaches. The arthroscopic examination showed multiple loose bodies with some cartilage fragments still attached to a hypertrophic synovial membrane. The treatment consisted of removing all loose bodies and partial synovectomy. In supine position with the use of distraction, our first step was loose bodies removal from all compartments through standard anterior ankle arthroscopy portals; then with patient in lateral position we proceed to remove the posterior remaining few larges fragments via posterior portals.\nPost-operatively the ankle joint was passively and actively mobilized and partial weight bearing was allowed as tolerated; the patient was able to return to daily activity after one month. Histopathology showed multiple calcified and ossified loose bodies, with lobules of cartilage in the synovial membrane and confirmed the diagnosis of synovial chondromatosis. At clinical follow-ups after one, four and 12 months and again at 10 years, the ankle joint presented a full range of motion without pain or swelling. Post-operative radiographs at one month and at 10 years () showed an absence of lesions, i.e., no recurrence. After 10 years, the radiographs indicate signs of mild degenerative changes. The patient was able to return to dancing. |
A 16-year old right-handed girl with paraesthesia in the left arm, initially occurring periodically and later more frequent, since the age of 12 was referred to our hospital due to these symptoms. X-ray showed a cervical rib on the left side and a minor one on the right side (no symptoms on right side; Fig. ). She had similar symptoms as in Case One, such as paraesthesia and numbness in the three ulnar fingers of the left hand when carrying things in the hand, when a pressure was applied supraclavicularly (e.g. carrying a backpack) or when working with the hands above the plane of the shoulder. Percussion of the area of the palpable cervical rib on the left side elicited symptoms in the three ulnar fingers and "hands up tests" exaggerated the symptoms in the same fingers. The radial pulse was normal in all positions of the arm. She had good strength in all muscles of the upper extremity and a normal sensibility in the hand. Isometric test and endurance of grip showed 32% and 62%, respectively and weakness in the left hand compared to the right side (BTE Primus work simulator). Isometric test of the flexion in the left shoulder and endurance showed 16% and 54%, respectively lower values, compared to the right side. Electrophysiological examination showed no abnormalities. MRI showed a 6 cm long cervical rib from C7 on the left side, which articulated against a cranially oriented bony process from the first rib where the articulation was bulky (Fig. ). The left brachial plexus was slightly lifted up by the skeletal abnormality. On the asymptomatic right side a 2.5 cm long cervical rib was found, which had no contact with the brachial plexus.\nThe brachial plexus and the cervical rib of the patient were explored when the girl was 17 years. The brachial plexus was distorted at and adhered to the ventral edge of the cervical rib and the bony process from the first rib (Fig. ). The main part of the cervical rib including the bone process from the first rib was resected after the lower trunk was lifted up (Fig. ). The subclavian artery was not impinged by the bone formation. The direct postoperative events were without problems, but later she was investigated at the Department of Infectious Diseases due to fever of unknown origin. No cause of the fever was found and later she recovered completely. She was followed regularly as with Case One.\nAt one year follow-up she had no symptoms in the hand. The preoperative symptoms had disappeared although she still experienced a feeling of impaired strength in the left arm. She had full range of motion and it was not possible to provoke any paraesthesia. Tests of fine motor activity in the hand (Crawford pins and sleeve and Minnesota picking test) showed improved values. Tests in the work simulator showed improvement [isometric test 5% weakness (preoperatively 32%), endurance 54% weaker (preoperatively 62%), isometric test of extension with elevated arm 4% weaker than the right side (preoperatively 16%), endurance of flexion/extension with elevated arm similar value on the right side (preoperative 54% weaker)]. MRI follow-up 11 months after surgery revealed no occurrence of the resected cervical rib. There were no differences compared to the two CT-scans done at three and six months after surgery (done for other reasons; fever investigation and a fall from a horse). The patient was pleased with the surgery. She continued with her previous studies and leisure activities without restriction. |
A 23-year-old man presented with sudden onset severe headache to a peripheral hospital followed by reduction of the level of consciousness. There was no history of trauma. His history indicated a minor psychological problem that was under medical treatment. Because of progressive reduction of the level of consciousness, he has been referred to our hospital for further investigation and treatment. When he reached our emergency ward, he was in a deep coma (decerebrate rigidity). The right pupil was dilated, and the left one was myotic. The patient had central neurogenic hyperventilation. He underwent brain computed tomography (CT) scan that disclose a high density hemispheric subdural hematoma (SDH) on the right side with SAH [].\nThere was a filling defect between hematoma and cortex in the center of hematoma. There was marked mass effect with displacement of ventricular structures to the left side. The laboratory data including coagulopathy screen were normal. Because of the patient's critical situation, we decided on emergency craniotomy and removal of hematoma without performing angiography or CT angiography. In the operating room a large right side craniotomy has been performed. The dura was tense. After opening the dura, a thick subdural clot was removed. After hematoma removal, at the center of the field, there was a thick SAH and a vascular lesion at the center of SAH. By dissecting the arachnoid, the lesion was an aneurysm connecting to precentral artery by a neck []. Aneurysm was pulsatile with thin membrane and a small clot adherent to it indicating of recent hemorrhage. The neck has been ligated and aneurysm coagulated and removed. Dura has been closed with a pericranial patch. The bone removed because of the brain edema. Postoperative CT scan has shown reduction of mass effect without additional bleeding []. Postoperative CT angiography of cerebral vessels showed no vascular abnormality [].\nAfter 35 days of operation, the patient discharged with mild left side hemiparesis and dysphasia. Then he just has mild hemiparesis several months after admission. |
A 14-year-old boy with morbid obesity and no known prior psychiatric history underwent sleeve gastrectomy. Prior to the surgery, he weighed 167 kilograms with a body mass index (BMI) of 54.5. Within a few months postoperatively, he weighed 70 kilograms with a BMI of 22.8. The patient’s substance use disorder started at the age of 15, one year after the bariatric surgery. Of note, the patient's parents were separated and he lived with his mother and siblings. None of his family members or relatives had a history of substance use disorder. He initially started using fenethylline (marketed under the brand name Captagon), as it was a common substance used by his peers at school. He started with two tablets daily and increased his use gradually up to 15 tablets daily. He started smoking cannabis a year later, at the age of 16, starting with one cigarette per day and increasing his use gradually until reaching a peak of 20 cigarettes per day. The patient started drinking alcohol occasionally at the age of 16 as well, and it soon became an issue of excessive use on a daily basis. The patient drank different types of alcoholic beverages. He reported incidents of fainting in relation to alcohol use but had never experienced withdrawal. He mentioned that he started using alcohol as a way to reduce his use of other substances. Two years later, at the age of 18, the patient started using methamphetamine, which caused him to develop paranoid ideation, auditory hallucinations, severe insomnia, and aggressive behavior. The patient was admitted to an inpatient psychiatric unit for a few days and was started on haloperidol 3 mg orally twice daily, benztropine 2 mg orally twice daily, and quetiapine 50 mg orally as needed for insomnia. His psychotic disorder improved with the cessation of substance use and the treatments initiated on the inpatient side. After his discharge, he unfortunately relapsed and continued to use the aforementioned substances.\nAfter arranging for close follow-up, the patient voluntarily presented to the rehabilitation center, motivated to stop using all substances, as he was legally and financially burdened by this disorder. He was incarcerated twice for substance use-related criminal charges. He was also motivated to start a new life and to enroll again in higher education, as he dropped out of school previously due to his polysubstance use disorder. The patient has thereafter been involved in a rehabilitation and relapse prevention program, which included inpatient admissions as needed to the rehabilitation center, involvement in individual and group therapy, occupational therapy, and addiction counseling. |
Patient 2 is a 78-year-old male who was experiencing persistent lower urinary tract symptoms despite combined medical therapy with alpha blockade and 5-alpha reductase inhibitors. His medical history included coronary artery disease status post-coronary artery bypass grafting and percutaneous coronary intervention, and a history of pneumonia. Preoperative cystoscopy revealed enlarged median and lateral lobes, as well as severe trabeculations of the bladder with a TRUS measuring a 41 cc prostate. HoLEP was carried out utilizing a two-incision technique. Upon completion of morcellation, it was noted that the patient's abdomen was distended, but his peak airway pressures were normal, the abdomen was soft, and the catheter drainage was noted to be clear. In addition, there was no suspicion for a significant mismatch between irrigation used and fluid output collected in the drainage system.\nGiven the previous similar presentation in Case 1 with no suspicion of bladder injury, we suspected that the patient had extraperitoneal extravasation of the saline irrigation through a capsular perforation as occurred in Case 1. The decision was made for the patient to be awakened, extubated, and transferred to the recovery room where he was further monitored. A stat noncontrast abdominal CT scan was performed that revealed a moderate amount of free fluid in the pelvis and upper abdomen; the fluid in the pelvis and lower abdomen was distributed in the extraperitoneal region with no evidence of hematoma (). The patient remained hemodynamically stable and was transferred to the floor with continuous bladder irrigation. The patient was given a 40 mg dose of Lasix ∼8 hours after the operation was completed. Overnight, there were no acute events. On POD 1, the patient's abdomen was soft and significantly less distended. The Foley catheter drained 3950 cc of urine overnight without evidence of hematuria. The patient was discharged with a catheter on POD 1. The patient had his catheter removed on POD 9. A postoperative CT cystogram revealed no evidence of leak with resolution of the pelvic and perivesical fluid (). Thirty grams of benign prostate tissue was removed on final pathology analysis. The patient was noted to have a bladder neck contracture seen on cystoscopy 4 months after his procedure for which he underwent cystourethroscopy and laser incision of bladder neck contracture. |
A 60-year-old woman presented to an outpatient colorectal clinic with a 2-month history of rectal bleeding. Her past medical history included a colpopexy which had been performed via a Pfannenstiel incision 20 years earlier. On examination a large villous adenoma was palpable within the rectum. She was referred for a diagnostic colonoscopy. The colonoscopy was performed without immediate complication using 25 μg of fentanyl and 2 mg of midazolam. Bowel preparation was performed to good effect with 2 litres of polyethylene glycol solution. The exclusion value of the examination was reported as excellent. Caecal intubation was confirmed by visualisation of the ileocaecal valve. The terminal ileum was not intubated. Two 4 mm sessile polyps were located within the middle third of the rectum and these were removed using hot biopsy. The lower third of the rectum contained a large sessile villous adenoma which occupied 50% of the rectal circumference at this level (7 cm diameter). The lesion was sampled by plain biopsy. Histological analysis revealed the 4 mm polyps to be tubulovillous adenomas and the larger sessile polyp to be a villous adenoma.\nEight hours after colonoscopy the patient developed gradual onset of abdominal pain associated with nausea and vomiting. She was admitted under the acute surgical service the next day. On examination she was afebrile with a heart rate of 130 beats per minute. Abdominal examination revealed diffuse tenderness with fullness and peritonism in the right iliac fossa. Peripheral blood analysis demonstrated a white cell count of 16.9 × 109/litre. Plain abdominal and chest radiography demonstrated several loops of dilated small bowel in the left upper quadrant but no evidence of free abdominal gas (Figure ). The patient was assumed to have a post-colonoscopic perforation with a resulting ileus. At laparotomy the mid-ileum was found to be strangulated within a congenital band adhesion. The adhesive band was localised to the right iliac fossa and was well removed from the site of her previous pelvic surgery. The 30 cm herniated intestinal segment was non-viable and was resected. Continuity was restored with a primary end-to-end anastomosis. The patient made an unremarkable recovery and was discharged home 6 days later. |
A 22-year-old Indian male, painter by occupation, presented to us with a painless swelling along the ulnar aspect of his right distal forearm since the last four months. To begin with, it was the size of a peanut but had gradually increased to its present size. There was no history of any other swelling in the body, fever, loss of weight or appetite, or history of similar complaints in the past. The family, occupational, recreational and drug histories were not significant. The general physical and systemic examinations were within normal limits.\nOn examination, there was an oval swelling 6 × 4 cm, occupying the distal third of ulna. The overlying skin was of normal colour and temperature. There was no overlying scar, sinus or prominent veins. The swelling was diffusely tender and homogenously firm in consistency. It was free from the overlying skin but adherent to the underlying bone. The range of motion of wrist was normal and painless. The distal neurovascular status was normal and grasping power equal in both hands.\nSerum biochemistry studies were within normal limits. Plain radiographs of the right ulna showed an expansile, multiloculated lytic lesion at its lower end with absence of periosteal reaction (). Plain chest radiographs were within normal limits.\nMagnetic Resonance Imaging (MRI) study was planned to delineate the extent of soft tissue involvement, but the patient was very poor and chose not to have the investigation.\nA clinical diagnosis of GCT was made, which was confirmed post operatively by histopathological examination. The condition, its prognosis and various treatment modalities were discussed at length with the patient.On the basis of clinical and radiographic evaluation, the lesion was graded as Stage 3 (aggressive) as per the Enneking Staging system for benign bone tumors []. As per the standard recommendations for Stage 3 lesions, a wide resection was planned after obtaining informed and written consent from the patient. The resection margins were calculated keeping in view the radiological extent of the lesion. Keeping in view the patient’s high functional demands, we also decided to stabilize the ulnar stump using the extensor carpi ulnaris tenodesis technique described by Kayias & Drosos [].\nThe tumor resection was extra-periosteal with 3 cm margin of the normal bone proximal to the tumor. This included approximately half (twelve centimetres) of the distal end of ulna, the triangular fibro cartilage complex, the ulnar border of the pronator quadratus and a part of the distal radio-ulnar joint capsule (, ). The extensor carpi ulnaris (ECU) tendon was dissected free from the tumor mass by blunt dissection and longitudinally split to a point 1 cm proximal to the cut end of the ulna. The tendon was passed through a 3.2 mm drill hole, 5 mm above the end of the ulnar stump in a dorsal to volar direction with the forearm held in supination. The tendon was then directed to the ulnar side and sutured back on itself (). This manoeuvre resulted in a cuff of the ECU tendon, which effectively stabilized the ulnar stump. The remnant of distal radio ulnar joint capsule was sutured with the ECU tendon in order to prevent ulnar subluxations of the carpus. The wound was closed in layers over a drain. Postoperatively, the forearm was immobilised in supination using an above elbow splint for two weeks, following which physiotherapy was commenced. Thereafter, the forearm splint was used only at night for another eight weeks and the patient was gradually advised full range of motion at the wrist and elbow.\nFollow-up was carried once every three months for the first two years and six monthly thereafter. Functional evaluation was done using the criteria described by Ferracini []. The patient had a normal function and muscle strength, no pain and no ulnar instability. He had restriction of forearm pronation - supination of approximately 15 degrees. However, the patient did not seem to be bothered by this and could carry out his professional activity as a painter quite efficiently. This patient therefore scored 16 out of a total of 18 points on the Ferracini scale which is consistent with an excellent functional outcome. |
A 10-year-old male came to the clinic at the Faculty of Dentistry in King Abdulaziz University complaining of irregular teeth and unpleasant smile [Figures -].\nOn examination, the patient had the following:\nDental and skeletal Class I relationship Normal overbite and overjet Severe crowding in the upper and lower arch - more than 10 mm in each arch Congenitally missing lower second premolars.\nPhase I\nextraction treatment in the upper arch and a modified serial extraction in the lower arch. The time duration for this phase was 2 years.\nThe age of the patient was 10 years when serial extraction treatment started and finished when all his permanent teeth erupted by the age of 12 years. The extractions were done in a serially ordered manner as follows:\nIn the upper and lower arches Extractions of all deciduous canines were done to have better alignment of the permanent lateral incisors and to provide space for them. After 5 months, the extractions of all deciduous first molars were done to stimulate eruption of all permanent first premolars. 6 months later, the extraction of the upper deciduous second molars was performed to stimulate the eruption of the upper permanent second premolars in the upper arch. In addition, the extraction of the decidous lower second molar was done, although the patient has congenitally missing second premolars; the space of the missing second premolar was utilized to resolve the anterior crowding along with the spontaneous closure of the extra spaces by the physiologic movement of permanent mandibular teeth.[] After 7 months and following the eruption of all the permanent first premolars; extractions of the upper first permanent molars were done to facilitate and allow space for the eruption of the upper permanent canines. However, no extractions of the first premolars in the lower arch were done due to the congenitally missing second premolars. 6 months later, all the patient permanent teeth had erupted and his age was 12 years, this is the end of Phase I.\nIn summary, a total of 14 teeth (12 deciduous teeth and 2 permanent teeth) were serially extracted during Phase I of the treatment as shown in Figures and .\nPhase II\nFollowing the completion of Phase I, and for a period of 6 months, fixed appliance treatment was performed to align the teeth and to close all the spaces as shown in .\nThe treatment overall time duration of both phases was 2 years and 6 months. Following the completion of the treatment, retainers were placed, Hawley appliance in the upper arch and fixed 3-3 in the lower arch as shown in Figures and .\nThis case was treated successfully with a modified serial extraction plan in the lower arch because the patient had congenitally missing lower second premolars with severe crowding. The treatment consisted of selective removal of the deciduous mandibular teeth only (C, D, and E) and no permanent premolar removal. The space of the missing second premolar was utilized to resolve the anterior crowding along with spontaneous closure of the extra spaces by physiologic movement of the permanent mandibular teeth. Whereas in the upper arch conventional serial extraction was performed. |
A 45-year-old female had no remarkable findings in her medical or family history. She started to have the symptoms of sudden headache and nausea. At the time of visit to our hospital, she presented with right dominant gaze rotatory nystagmus and right cerebellar hemisphere symptoms. Head computed tomography (CT) scan and magnetic resonance imaging (MRI) showed a highly contrasted cystic tumor with a mural nodule in the right cerebellar hemisphere []. The cerebral angiography showed an intense tumor stain from the anteroinferior cerebellar artery and draining into the petrosal vein []. The blood biochemistry findings showed no abnormalities such as polycythemia. No abnormal finding was found by a whole body scan including the retina. No VHL gene mutations were found by direct sequence of DNA 3p25 region using whole blood. Total tumor resection by craniotomy was conducted, and a pathological diagnosis of hemangioblastoma was made []. The symptoms disappeared and she was discharged on foot. Thereafter, recurrence of a nodular tumor developed at the site of the initial tumor 38 months after the initial surgery []. Since she did not wish to receive surgery, local stereotactic radiosurgery (SRS) at 20 Gy was performed. As a result, the tumor gradually shrank over a period of 2 years. ]. However, paraplegia developed in the legs 91 months later. MRI showed the presence of three spinal cord tumors at the levels of C7, Th7, and L2 []. The tumor at Th7 was resected and diagnosed as hemangioblastoma. The tumor at C7 was treated with cyberknife at 17 Gy in five fractions. The tumor at L2 was also resected 107 months later during follow up because the tumor enlarged. During this surgery, microvascular proliferation was observed in the arachnoid around the tumor, for which the presence of hemangioblastoma cells was confirmed by pathological examination. The general condition of the patient rapidly deteriorated thereafter, and MRI revealed disseminated foci in the subarachnoid space in the cranium as well as over the spinal cord. Ventriculo-peritoneal shunt (VP shunt) and irradiation at 36 Gy to the whole brain and whole spine were performed, but no improvement was obtained [Figure ,]. The patient died due to respiratory failure after the course of 120 months. Autopsy findings showed the foci restricted in the cerebrospinal region. There was multicentric nodular dissemination of hemangioblastoma in the cerebrospinal subarachnoid space, and compression of the medulla oblongata was considered to be the cause of death [Figure –]. The initial tumor treated by SRS became fibrosis foci accompanied by hyalinization with a maximum diameter of 15 mm, and no viable tumor tissues were observed []. |
The second case is a 75-year-old Irish woman with a psychiatric history of bipolar affective disorder, stable for several years on olanzapine and valproate, enabling her to lead an independent lifestyle. There was no history of cognitive impairment. She suffered from multiple medical conditions including: atrial fibrillation, type 2 diabetes mellitus, obstructive sleep apnea, and a recent mitral valve repair complicated by postoperative delirium.\nShe was admitted medically to a rural Irish hospital in November 2015 for management of a raised INR. During the admission she developed sudden onset left-sided weakness and altered levels of consciousness, as well as rigidity and one isolated temperature spike. The concern was raised that she may be or might have been suffering from neuroleptic malignant syndrome and her neuroleptics were stopped as a precaution (Table ). She was transferred to the intensive care unit (ICU) in the MMUH in Dublin with a suspicion of neuroleptic malignant syndrome or encephalopathy. Computed tomography (CT) brain imaging was normal at the time. As neuroleptic malignant syndrome was suspected, olanzapine was stopped. However, her creatinine kinase levels were normal as was her body temperature. Hence, neuroleptic malignant syndrome was deemed to be unlikely. An electroencephalogram during admission showed changes suspicious of encephalopathy and MRI imaging showed no acute abnormality. A working diagnosis of metabolic encephalopathy was established but extensive investigations yielded no cause for the encephalopathy.\nDue to prolonged altered levels of consciousness and unexplained altered mental state, the Liaison Psychiatry service was consulted in January 2016.\nOn examination, she responded with a mouthed single word greeting, but made no other attempt at verbal interactions. She inconsistently followed the examiner with her gaze, but stared out of the window for most of the examination. On physical examination she presented with waxy resistance to passive movement and psychomotor retardation. The impression was that these features were most likely related to a catatonic exacerbation of her bipolar affective disorder, in the absence of an organic explanation. She was diagnosed as having bipolar I disorder with catatonia as per DSM-5 (Table ).\nDelirium was raised as a differential diagnosis (Table ), but she had been reviewed in September 2015 by the Liaison service, when she was delirious after her valve replacement and her presentation was distinctly different on that occasion.\nShe was initially treated with intravenously administered lorazepam, but became drowsy, with a significant drop in Glasgow Coma Scale (GCS). As such the treatment was abandoned. Instead, olanzapine was cautiously reintroduced, which led to a significant improvement in her mental state within days. On follow-up review, she was mildly confused but engaged well at interview, and was euthymic with no evidence of thought disorder or movement disturbance. Subsequently she was discharged back to her own home. She was not reviewed at 6-month follow-up as she was living in a rural area and was followed up in her local service.\nOf note, in 2017, the same patient was readmitted to the MMUH ICU, from the same peripheral hospital, in a very similar state to the presentation in November 2015. Again her neuroleptics had been stopped when she was acutely unwell and she developed typical traits of acute catatonia. She was trialled on lorazepam, which she did not tolerate and reinstitution of her neuroleptics brought no improvement. The therapy was then escalated to electroconvulsive therapy (ECT), to which she had a dramatic response and significant improvement of her mental state. |
A 57-year-old female visited a respiratory internal physician due to suspected lung cancer (based on a mass screening chest X-ray examination). She did not have any symptoms. The chest X-ray showed a tumor shadow in the upper-middle field of the right lung with pleural effusion and a tumor shadow in the upper field of the left lung (). Computed tomography (CT) of the neck and chest revealed that the tumor shadows had been caused by a substernal goiter connected to the thyroid gland in the neck. According to the patient, she had been diagnosed with a goiter about 23 years ago, and it was followed up, but the follow-up process had been discontinued several times. After about 20 years, she visited our hospital for surgical treatment.\nIn a physical examination, the palpable thyroid gland was found to be diffusely swollen and soft and exhibited poor mobility. The lower pole of the thyroid was not palpable.\nA blood examination revealed normal thyroid function, a thyroglobulin level of 352 ng/ml, and negativity for the thyroglobulin antibody.\nUltrasound showed that the cervical thyroid gland was diffusely enlarged and exhibited multiple regions of cystic degeneration, but no obvious malignant findings were observed.\nCT of the neck and chest () showed the diffusely swollen thyroid gland and a substernal goiter, which extended to both sides of the thorax. Specifically, it extended to the bifurcation of the trachea on the dorsal side of the superior vena cava, the innominate vein, the aortic arch, and the ventral side of the trachea. The width of the goiter at the mediastinum was 145 mm (length: 80 mm, thickness: 80 mm). The right side of the substernal goiter was bigger than its left side. The interior of the lesion was heterogeneous, and calcification was seen in part of it. The goiter had compressed the trachea in the mediastinum, and the lumen of the trachea measured 6 mm in diameter at its narrowest point. Pleural effusion was noted in the right thorax. We performed 18F-fluorodeoxy glucose positron emission tomography to determine the malignancy of the substernal goiter, but no radiotracer accumulation was observed.\nWe also conducted a pathological examination. Fine-needle aspiration cytology of the cervical thyroid gland resulted in the lesion being classified as of “indeterminate significance,” and a pathological examination of a needle biopsy sample from the same site led to the lesion being diagnosed as a follicular neoplasm. Fine-needle aspiration cytology of the right pleural effusion demonstrated that it was benign.\nThe patient underwent total thyroidectomy using a transcervical and full sternotomy approach. The anesthesiologist intubated the patient with a bronchoscope. Although tracheal stenosis was observed, intubation was performed smoothly. Later, the tracheal tube was replaced with an NIM™ EMG endotracheal tube so that intraoperative nerve monitoring could be performed. The patient was placed in a supine position with her neck well extended. A cervical skin incision was made, and a median chest midline incision and full sternotomy were performed. First, we identified the bilateral vagal nerves and confirmed the absence of paralysis with the NIM™. As a preparation for the resection of the substernal goiter, the major blood vessels, including the innominate vein, brachiocephalic trunk, superior vena cava, and left subclavian artery, were carefully separated from the substernal goiter, and then thyroidectomy was performed ().\nThe right superior thyroid pedicle and right middle thyroid vein were ligated and dissected to allow the right thyroid lobe to be rotated to gain a view of the recurrent laryngeal nerve (RLN) from the lateral aspect of the thyroid gland, but the goiter prevented the right thyroid lobe from being rotated. It was difficult to identify the right RLN, so we decided to try to exteriorize the left thyroid lobe, which was smaller than the right thyroid lobe. The left superior thyroid pedicle and the left middle thyroid vein were ligated and dissected. The left thyroid lobe was more mobile than the right thyroid lobe, and the left RLN could be identified by rotating the left thyroid lobe in the medial direction. The NIM™ was effective at identifying the RLN. After identifying the left RLN, the left lower thyroid artery was ligated and dissected. The left RLN was carefully separated from the dorsal side of the left thyroid lobe and the substernal goiter so as not to cause any damage. The substernal goiter, which was connected to the left thyroid lobe, was pulled in the cranial direction, and the part adhering to the surrounding tissue, particularly the tissue between the goiter and the innominate vein, was dissected by ligation and coagulation with an energy device. Subsequently, the left thyroid lobe was also separated from the trachea. The exteriorization of the left thyroid lobe improved the mobility of the right thyroid lobe, and the right RLN was identified by dislocating the right upper pole to the caudal side. We carefully separated the right RLN from the goiter and ligated and dissected the right lower thyroid artery. We pulled the substernal portion of the right thyroid lobe gradually; separated the tissue connected to the goiter, including the left thyroid lobe; and succeeded in moving the substernal goiter in the cranial direction. The remaining attachments between the right thyroid lobe and trachea were broken, and a total thyroidectomy was conducted. We found three parathyroid glands had adhered to the resected thyroid gland, so we performed autotransplantation using the sternocleidomastoid muscle. The wound closed after drains were inserted in the neck and mediastinum. After the surgery, the patient was extubated immediately because no respiratory tract problems (e.g., tracheomalacia) were noted. The total duration of the operation was 9 h and 22 min, and the total amount of intraoperative blood loss was 3298 ml. The resected thyroid weighed 614 g ().\nPostoperative transient hypoparathyroidism was observed. Routine treatment with calcium (3 g daily orally) and 1 alpha-hydroxyvitamin D3 (2 μg daily orally) was administered. The patient was discharged home on the 9th postoperative day on levothyroxine (100 μg daily orally). A histopathological examination did not reveal any signs of malignancy, and so the lesion was diagnosed as an adenomatous goiter. |
A 44-year-old man without notable previous medical history presented with severe headache, nausea, and vomiting that had persisted for a few days. He was admitted to a local hospital, and computed tomography (CT) and magnetic resonance imaging revealed a large tumor with intratumoral hemorrhage in the right frontal lobe. For the management of intracranial hypertension, osmotic diuretics were administrated, leading to dehydration. The third day after admission, he was transferred to our hospital due to neurological deterioration including progressive disturbance of consciousness and left hemiparesis.\nMagnetic resonance imaging performed at our hospital showed a rapid increase in mass effect, resulting in subfalcial and transtentorial herniation (). The commonly measured coagulation parameters, including prothrombin time, activated partial thromboplastin time, international normalized ratio, and thrombocyte count, were within the normal range. The patient underwent an emergency right frontal craniotomy performed in the supine position without excessive head rotation. The tumor was removed en bloc using the navigation-guided fence-post procedure without opening the ventricle or cistern. Accordingly, excessive CSF leakage did not occur during the operation. In addition, postoperative CSF loss through closed subgaleal drain did not occur.\nPostoperatively, the level of consciousness remained unchanged from the preoperative level and no new neurological deficits were detected. A routine postoperative CT scan on the day after surgery revealed a bilateral cerebellar hemorrhage along with the cerebellar sulci facing the tentorium (). Magnetic resonance imaging showed bilateral anterior cerebral artery infarction due to subfalcial herniation and right posterior cerebral artery infarction due to transtentorial herniation that reflected the preoperative intracranial hypertension, and no evidence of residual tumor at the surgical site was found (Figures –). The cerebellar hemorrhage was carefully treated conservatively with osmotic diuretics. A follow-up CT scan demonstrated no further progression of hemorrhage and no occurrence of obstructive hydrocephalus due to compression of the fourth ventricle by the associated edema (). The level of consciousness recovered gradually and no neurological deterioration occurred during the course. The histological diagnosis was anaplastic oligoastrocytoma. Conventional radiotherapy of 60 Gy concurrent with PAV combination chemotherapy of procarbazine, nimustine hydrochloride (ACNU), and vincristine was performed. The patient did not have any cerebellar deficits and was transferred to a rehabilitation center for rehabilitation of paraparesis that occurred due to anterior cerebral artery infarction. |
A 52-year-old adult female who is a known patient of fibromyalgia and spondylolisthesis at the lumbar vertebral levels L2, 3, 4 for the last 3 years presented with a 3-day history of acute onset very severe epigastric pain increasing in severity with time since onset. At presentation, the patient reported a score of 10 on a pain rating scale of 10. The quality of the pain was sharp, and it was radiating to the back with mild relief on bending forward. There were no known aggravating factors. The pain was associated with nausea and non-bilious vomiting with no traces of blood. The patient denied any similar episodes in the past and has no history of alcohol consumption. She has a history of cholecystectomy done 2 years back for abdominal pain, but the gallbladder was apparently devoid of any gallstones.\nAn ultrasound and a computed tomography of the abdomen and blood workup were performed to determine the cause of pain. At admission, the serum lipase levels were elevated to > 1,600 IU/L (laboratory reference level 10 - 53 IU/L). An ultrasound of the abdomen revealed a normal common bile duct with its diameter of 5.4 mm (). The abdomen CT findings were also consistent with the diagnosis of acute pancreatitis wherein there was stranding in the retroperitoneum and the abdominal mesentery with trace fluid in the perihepatic and pelvic space. There was edema of the head and body of the pancreas with peripancreatic edema but without any pancreatic pseudocyst. The remaining findings were unremarkable with no intrahepatic or extrahepatic biliary dilatation, patent mesenteric blood vessels and normal spleen and adrenal glands (). Further workup was done to look for other less frequent causes of pancreatitis. A magnetic resonance cholangiopancreatography (MRCP) was done which did not reveal any gallstones or sludge in the common bile duct (). IgG4 levels were normal thus ruling out autoimmune pancreatitis. Triglycerides were within the normal limits, and viral hepatitis panel was negative. At this stage, a possibility of drug-induced acute pancreatitis was considered. The patient was on oxycodone 5 mg twice daily for fibromyalgia and gabapentin 600 mg three times daily for neuropathic pain for the last 3 years. She also gave a recent history of being prescribed doxycycline 100 mg twice daily by her dermatologist for worsening acne. The patient had been taking doxycycline regularly for the past 7 days. Since the patient was doing all right with oxycodone and gabapentin for a longer time, it was considered that doxycycline would be the most probable cause for the patient’s condition. However, all three drugs were stopped immediately. By then, the serum lipase levels began to fall. It was 1,140 IU/L the second day, 347 IU/L the third day and 111 IU/L on day 4.\nThe patient was started on enteral feeding when her nausea subsided. However, her abdominal pain persisted. Due to her constant and severe abdominal pain, a repeat abdomen CT was done on day 6 which showed a little necrosis of 6 mm in the head of the pancreas. The patient was managed conservatively with IV fluids and parenteral morphine. After the repeat abdomen CT was done, the patient was rechallenged with oxycodone and gabapentin, and the lipase levels were found to be normal. As the patient’s condition improved, oral feeding was started and was subsequently discharged. |
After suffering gross hematuria for 2 years, a 76-year-old man was referred to the hospital. He had been diagnosed with a complete duplication of the right renal pelvis and ureter in childhood, but had no symptoms. He was taking an antiplatelet drug due to a history of angina. There were no abnormal laboratory findings in blood samples other than a slightly high value of creatinine at 1.04 mg/dL. Abdominal ultrasonography revealed a grade 3 hydronephrosis in the upper right kidney. No tumor was suspected and prostate volume was approximately 34 mL. Flexible cystoscopy did not detect any bladder tumor, renal hematuria, or ectopic orifice. However, an enhanced tumor 15 mm in diameter in the pelvis of the right kidney was detected by computed tomography (CT) scan. The ureter from the upper kidney was not open to the bladder and an ectopic opening to the prostate was suspected. This tumor site was mild diffusion reduction in magnetic resonance imaging (MRI) scan. Based on MRI scan shown in Figure , it was clearly suspected that the ureter downstream of the tumor was expanding with ectopic opening into the prostate. All urine cytology was negative (class 1 or 2).\nUreteroscopy (URS) under general anesthesia revealed the ectopic ureter opening beside the verumontanum (seminal colliculus), and the findings are shown in Figure . A rigid ureteroscope could be inserted smoothly via the ectopic ureter through a hydrophilic guidewire. The lower ureter was expanding but the scope could not be inserted because of meandering. A 10/12-Fr ureter access sheath was able to be inserted without difficulty and the flexible ureteroscope detected a hemorrhagic papillary tumor corresponding to the imaging findings of the renal pelvis. There was no tumor in the ureter. Although renal pelvic urine cytology was negative (class 2), the tumor was suspected to be malignant by findings of URS and imaging, and so laparoscopic radical nephroureterectomy was performed. Because there was no obvious tumor around the ectopic ureter and lower ureter, the lower ureter was blinded and the prostate was preserved. A pedunculated papillary tumor 18 mm in diameter shown in Figure was detected in the renal pelvis of the right upper renal unit. Histopathological examination of the tumor demonstrated urothelial carcinoma, high grade, pTa. The blind end of the ureter was detected by cystography performed 6 days after surgery, shown in Figure . |
A 61-year-old male presented with a past history of TEVAR (GORE TAG thoracic endoprosthesis; 37 mm×20 cm, 40 mm×20 cm; W. L. Gore and Associates) for a true aneurysm of the descending aorta 4 years prior. In addition, he had a past history of tuberculosis and right nephrectomy for renal cell carcinoma. The diameter of the native aorta at the distal end of the stent graft was 38 mm at the time. Subsequently, the diameter of the aorta at the end of the thoracic stent graft gradually increased, and the diameter of the native aortic wall exceeded the size of the stent graft 2 years later. A type Ib endoleak and descending aneurysm enlargement were not observed because of thrombosis between the aorta and the stent graft. The patient refused surgical intervention, and a careful follow-up was continued. However, a type Ib endoleak occurred 4 years later because of enlargement of the aorta at the diaphragm (from 38 mm to 58 mm; ) associated with an enlargement of the middle descending aorta and onset of consumptive disseminated intravascular coagulation (DIC). His DIC score was 6, as defined by the International Society on Thrombosis and Hemostasis. The distance between the distal edge of the stent graft and the celiac artery was less than 17 mm (). Moreover, the ostium of the celiac artery and superior mesenteric artery was very close, and the aortic diameter below the celiac artery was approximately 30 mm. We considered TEVAR with a fenestrated stent graft or celiac artery coverage as treatment options; however, long-term results remained unclear with technical difficulties. Therefore, we planned graft replacement between just above the celiac artery and the distal edge of the stent graft.\nGraft replacement of the entire descending aorta was not chosen because severe adhesion in the upper left pleural cavity was predicted because of the past history of tuberculosis. The patient was placed in the right lateral decubitus position, and a left thoracoabdominal incision was made through the seventh intercostal space. The abdominal section was approached via the retroperitoneal space, and the aorta was exposed at the level of the superior mesenteric artery. A femoral–femoral extracorporeal bypass was established. Heparin (300 U/kg) was administered, and the activated coagulation time was maintained above 400 s. The pump flow was 1.5 L/min, and the systolic upper body blood pressure was maintained above 100 mmHg. The proximal aorta was 7 cm in diameter and was clamped using two large arcuate forceps at the distal descending aorta. Distal clamping was performed between the celiac artery and the superior mesenteric artery (). The proximal anastomosis was as follows: 1) the flared stent of the distal end of the stent graft that appeared to make the proximal anastomosis difficult was cut first and 2) an invaginated 28 mm J graft (Japan Lifeline, Tokyo, Japan) was inserted into the previously deployed stent graft (). There was a size discrepancy between the stent graft (40 mm in diameter) and the native thickening of the aortic wall (58 mm in diameter). Anastomosis was performed using an over-and-over suture with 3-0 PROLENE® (Ethicon. Inc., Somerville, NJ, USA), reinforced by a Teflon™ felt strip, adjusting the size discrepancy. This procedure could be easily performed even with the limited and distant surgical view. Some additional suturing was performed to secure hemostasis. The distal end of the inserted graft was proximally extracted, and the cut was adjusted for distal anastomosis. Distal anastomosis between the native aortic wall and the new prosthetic graft was performed using an over-and-over suture with 4-0 PROLENE® (Ethicon. Inc.) (). There was no bleeding at the proximal anastomotic site. The postoperative computed tomography (CT) image is presented in . |
An 11-year-old boy had presented to the orthopedic department with the complaint of head tilt to the left along with facial deformity in the form of a small left side of the face and skull. He had a full-term normal vaginal delivery in hospital and was apparently normal at birth. The parents noticed a head tilt to the left when the boy was six months of age but they did not seek any medical help at that time. The condition progressed mildly over the years, however, no physical discomfort was experienced by the patient. The child had adjusted well to this tilt. There was no history of similar complaints in other family members.\nOn inspection, we noticed an asymmetry in the contour of the neck, the shoulder, and the bilateral scapula. Anterior neck muscles were prominent on the left with the the head turned to the left side (Figure ). The left shoulder was at a higher level than the right, the left scapula was elevated and there was mild scoliosis to the left (Figure ). The patient had no restriction in flexion and extension movements in the anteroposterior (AP) direction or tilting the neck to the left side, however, there was a significant restriction in tilting the head to the right side. In view of the above findings, a clinical diagnosis of contracture of left SCM was made and the patient was referred for imaging.\nDue to asymmetry in the shoulder position, a radiograph of the neck (Figure ) was acquired to look for any bony anomalies. The radiograph of the neck (Figure ) and the chest (Figure ) were largely unremarkable and all the vertebrae and bilateral shoulder joints were normal. However, mild scoliosis was seen. Ultrasound (US) of the neck region was subsequently performed (Figure ). On examining the patient with a linear probe, the right sternocleidomastoid muscle was not visualized and was replaced by a fibrotic hypoechoic structure seen extending from the mastoid process to the clavicle. The left SCM, however, was normal in size and echotexture. No focal lesion was seen within it. A provisional diagnosis of atretic or aplastic sternocleidomastoid was made. Subsequent evaluation with contrast-enhanced MRI of the neck demonstrated the complete absence of the right SCM muscle along with the absence of the ipsilateral trapezius muscle (Figure -). The left sternocleidomastoid was normal. The sections of the posterior fossa revealed hypoplasia of the right cerebellar hemisphere (Figure ). The spine, however, was normal. The right lung was normal in position and no herniation into the neck was seen. At present, the patient had marked torticollis as well as cosmetic deformity and was thus advised release. |
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