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A 27-year-old man with more than 1-year history of a subcutaneous tumor in his left cheek consulted the Department of Plastic Reconstructive Surgery at our hospital. His past medical history and family history were unremarkable. Physical examination revealed an elastic hard tumor in the subcutis of his left cheek. The tumor did not adhere to the skin. There was no remarkable abnormality in his oral and nasal cavities. No lymph node swelling was observed in his head and neck. The patient exhibited no neurological deficits. Laboratory data were within normal limits. Magnetic resonance imaging (MRI) revealed a tumor 15 mm × 7 mm in diameter located anterior to the left masseter muscle, with lower intensity than the muscle on both T1- and T2-weighted images (Figure ). Imaging with a fat-suppression technique revealed slightly higher intensity in the tumor (Figure ). MRI findings were suggestive of a granuloma or a fibroma. The patient underwent local excision of the tumor under regional anesthesia. Grossly a whitish tumor was encapsulated by thin fibrous tissue, and it adhered to the masseter muscle. Postoperative investigation using MRI and positron emission tomography revealed no residual tumor or lymph node metastasis. The final histological diagnosis was ANOS of the accessory parotid gland. As the tumor was close to the surgical margin, we recommended that the patient receives adjuvant radiotherapy, but he did not wish to undergo the therapy. He is alive without recurrence of the disease 3 months after the surgery.\nThe tumor was encapsulated by fibrous tissue but extracapsular invasion was partially observed. Normal salivary gland tissue was observed adjacent to the capsule of the tumor. It was considered an accessory parotid gland. Approximately half of the tumor consisted of irregular-shaped cystic spaces of variable size containing exudates and hemorrhage (Figure ). Colloid-like material such as that observed in acinic cell carcinoma was not seen. The epithelial cells lining the cyst exhibited marked papillary proliferation with a partial cribriform structure (Figure ). The cribriform structure included true lumens but not pseudo-lumens. Thickened basement membranes were observed around the tumor cell nests in the periodic acid-Schiff (PAS) reaction (Figure ). Most tumor cells had a small, round, bland nucleus with inconspicuous small nucleoli and eosinophilic cuboidal cytoplasm. An apocrine-like appearance with apical snouts was observed in some cells (Figure ). Apocrine-like cells had cytoplasmic PAS-positive/diastase-resistant eosinophilic granules (Figure ). In the vicinity of hemorrhage, tumor cells phagocytosed the brownish pigment, hemosiderin (Figure ). Some tumor cells had foamy cytoplasm with microvacuoles similar to those of sebaceous cells (Figure ). These histological features were suggestive of LGCCA, but, in the latter half of the tumor, neoplastic epithelial cords and tubules considerably infiltrated the parenchyma with myxoid stromal reactions and sclerosis (Figure ). This invasive component lacked specific histological features of any other salivary carcinoma; therefore, the final diagnosis of ANOS was made. Neither intravascular infiltration nor perineural infiltration was observed.\nIn immunohistochemistry, some of the papillary-cystic structures exhibited rimming of the myoepithelium that was positive for smooth muscle actin (SMA), p63 (Figure ), and cytokeratin (CK) 14 (Figure ), indicating the presence of intraductal neoplastic components. Tumor cells exhibited strong positivity for CK (AE1/AE3), CK 7 and S-100 (Figure ), which was compatible with LGCCA. Epithelial membrane antigen was partially positive. Glial fibrillary acidic protein was faintly positive in the cytoplasm. Carcinoembryonic antigen was positive in the lumen of cystic spaces and tubules (Figure ). CK 20, gross cystic disease fluid protein (GCDFP)-15, CD34 (Figure ), and p53 were negative. SMA, p63, and CK 14 were negative in the neoplastic cells other than the myoepithelium rimming the cell nest. As neuroendocrine markers, synaptophysin was negative (Figure ) and a small number of CD56-positive cells were scattered (Figure ). Intracytoplasmic granules exhibited faintly non-specific staining with chromogranin A. The Ki-67 labeling index was approximately 5% (Figure ). |
A male patient, aged 13, was hospitalized in February mainly due to aggravated kinesalgia of left knee for 1 year and 9 months, which was accompanied by left lower limb lameness for two months. The pain was present when the patient was active and was relieved after rest. Fever, night sweats, nausea, vomiting, night pain and other symptoms were not concurrent. The symptoms were worsened due to strenuous exercise 2 months ago. X-ray and MRI examinations of left knee performed in other hospital revealed no abnormalities; CT and MRI examinations of bilateral hip joints revealed abnormalities. CT images showed that bilateral acetabular articular surfaces were rough with increased densities; cortical bone fractures and small free bone fragments were observed; the left femoral head was irregular with rough edges and unclear bone trabeculae; the right femoral head was basically normal. MRI examinations of bilateral hip joints showed that bilateral hip joints were in asymmetry; a small amount of hydrops were present in the left hip joint cavity; the joint space was widened, the acetabulum became shallow; the femoral head was slightly shifted upward, the femoral head and acetabular articular surface were rough and irregular; areas with abnormal signal intensity were observed in local sclerotins; T1W1 scan showed uneven and slightly lower signal intensities; T2W1 and STIR scans showed heterogeneous signal hyperintensity; the peripheral soft tissues were swollen with irregular heterogeneous signal intensity. The right hip joint space was present, the superior margin of the acetabular articular surface was rough; abnormal intensities were not observed in the right femoral head or in the upper segment of the femur and acetabulum. The imaging findings were as follows: (1) Right hip joint dysplasia; (2) Left hip joint abnormalities. Infectious disease, preferably the joint tuberculosis was considered. Aseptic necrosis of the femoral head was not excluded. The physical examinations on admission showed that the left lower limb lameness was present, tenderness of bilateral hips and inguinal region was absent, left patrick sign was positive, external rotation of left hip was obviously limited. The circumferences of the left and right knees at 15 cm above the patellas were 38 and 41 cm respectively. The distance from the anterior superior iliac spine to the medial malleolus was 82 cm for the left side, and 83 cm for the right side. Bilateral lower limbs had muscle strengths of Class V, symmetrical sensations, normal physiological reflections and negative pathological signs (−).\nAfter admission, the pelvic X-ray examinations showed that the left femoral epiphysis was irregular with bone defect areas on both outer and inner margins; the local bone density was increased, local bone defects were also observed on the outer margin of the femoral neck without obvious periosteal reactions. The left acetabulum was inhomogeneous in density and had multiple saccular low-density shadows. The space of the left hip joint was uneven with local stricture; peripheral spindle-shaped soft tissue density shadows were observed. The right acetabulum was thickened and whitened, and multiple mottled shadows with low signal densities were observed. The right hip joint space was not narrow. Other parts of bilateral hip joints had no obvious bone abnormalities. The findings also indicated high possibility of hip joint tuberculosis. The imaging data are shown in Figs. , , , .\nAfter admission, routine examinations were conducted. Electrocardiogram and abdominal B-type ultrasonography showed no abnormalities; the test results of hemogram, liver function, renal function and erythrocyte sedimentation rate were normal. Blood biochemistry tests showed that the serum C-reactive protein level was 7.08 mg/L (Reference value: 0–3 mg/L). In the four indices of coagulation function, the partial thromboplastin time was 38.7 s (Reference value: 24.9–36.8), and the other three indices were normal. The tuberculin test showed a strongly positive result. After admission, the patient underwent diagnostic puncture of the left hip. The result indicated the presence of uncoagulated bloody liquid. The coagulation factors were further detected in Institute of Hematology, the Peking Union Medical College Hospital. The results indicated that the value of factor VIII was 27.8 (Reference value: 50–100), both factors IX and XI were normal. Therefore, the patient was suspected as subclinical hemophilia. After the patient was infused with cryoprecipitate blood preparation, reexamination of the blood gave a normal result, and the local pain was quickly relieved. |
A 24-year-old man presented at our clinic with a four-year history of a painful nodule in his left cheek that reoccurred after two attempted excisions in the last four months by different specialists. No biopsy result or other histological information was available. Otherwise healthy, the patient complained of pain and local tenderness. Clinical examination revealed a well-defined, round, firm nodule in the middle of the left cheek, approximately 1 × 1 cm in size that appeared to be subdermic with no apparent fixation to the underlying tissues. The overlying skin was normal and the nodule was extremely painful on palpation. Examination of the oral cavity was normal and no neck lymph nodes could be palpated. Complete head and neck examination and laboratory tests were also unremarkable. Contrast enhanced CT (puffed cheek method) revealed a well-defined, contrast-enhanced, round soft-tissue mass of the left cheek over the buccinator muscle measuring 13.8 × 9 mm, with no apparent relations to the surrounding tissues (). Attached to it a very small satellite lesion was noted. In the operating room under local anesthesia, a relatively wide excision in order to remove both the masses was performed. Though we were not able to demarcate the main and satellite nodules macroscopically, no evidence of residual disease was apparent. Special care was taken regarding the protection of facial nerve branches and the cosmetic result. Histological examination of the specimen was consistent with the diagnosis of glomus tumor of the solid type, with no mention of potential residual disease (). The patient reported relief of his symptoms after surgery, but at 2-month followup he complained of gradual recurrence of local tenderness at the site of the excision. Three months later, the patient had a small palpable nodule and complete recurrence of his symptoms. Under local anesthesia, a very wide excision including the previous scar (with approximately 1 cm margin around the scar) was performed. Histological examination revealed glomus tumor with solid, angiomatoid, and angiomyomatous features (). The margins of the excision were reported as free of lesion. The patient remains without any symptoms nine months after surgery. |
A 49-year-old man presented to the ED complaining of pain and swelling of his penis secondary to an incarcerated penile constriction device placed 9 days before presentation. The device was placed by the patient himself for autoerotic motive. He later found that he was unable to remove it. He had no history of psychiatric illness and did not seek any medical attention prior to the ED visit. He complained of increasing swelling and severe pain. He was able to urinate but had a decreased force of stream. Physical exam revealed a tightly encircling metallic ring with peripheral cogs placed on the midshaft of the penis causing severe penile engorgement and edema (). The metal appeared to be a very hard alloy with thickness measuring 5–7 mm depending on the location. The penile skin under the ring was excoriated and necrotic. Due to the incarceration time, degree of necrosis, and significant distal edema, simple lubrication, compression, and manual removal were not an option for fear of amputation. Manual and electric ring cutters were used, but after several attempts, we were unable to do more than scratch the surface of the metal ring.\nAt this point, we contacted the orthopedic central parts depot and obtained a Large Orthopedic Pin Cutter. The patient was given procedural sedation and a tongue depressor was placed beneath the metal ring to provide soft tissue protection (). Using the pin cutter, enough force was generated in one attempt to snap the ring into two separate pieces (). The penis was cleansed with saline and a nonadherent antimicrobial was applied. The patient was then catheterized with a 16 Fr Foley catheter with clear urine return and was admitted for observation and care. After 24 hours, penile edema slightly improved but still persisted. The urethral catheter was removed after two days, following which, the patient voided satisfactorily. He was discharged with advice to maintain local hygiene and apply a topical antibiotic over the area. On two-week followup, penile edema had subsided completely and the overlying soft tissue was healing well by secondary intention. He had normal voiding and noticed return of nocturnal erections. The patient failed to follow up for subsequent visits. |
A 35-year-old female with a medical history significant for acute biliary pancreatitis status post laparoscopic cholecystectomy, presented to the surgery clinic as a new consult for a large cystic liver mass with increasing abdominal pain for 2 weeks. Patient reported suffering from severe pain in right chest, shoulder and diffuse abdominally that worsened with standing. Patient complained of inability to inspire due to pain and a 10-pound weight gain over 2 months. She confirmed decreased appetite especially with solids, nausea and emesis, alternating constipation and diarrhea, bruising, and pruritus at night. Computed tomography (CT) scan demonstrated a peripherally septated 10 x 7 cm cystic mass in the liver with intrahepatic biliary dilation (). The patient was referred for endoscopic retrograde cholangiopancreatography (ERCP) to establish preoperative biliary anatomy and was found to have moderate compression of the common hepatic duct managed with a right hepatic biliary endoprosthesis (); no obvious communication of the biliary tree with the cystic lesion was seen. Patient symptoms persisted despite optimizing with a protein-rich liquid diet; thus, the decision was made to proceed with the surgical plan for an open partial central hepatectomy. The patient was taken to the operative theater. After induction of general anesthetic, an upper midline incision was made. Inspection of the abdomen and liver showed no metastatic lesions grossly or with ultrasonographic imaging. The cyst was visible upon entry into the abdomen with no solid component to the mass in proximity to the cystic neoplasm. The second portion of the duodenum was adherent to the cyst with inflammatory adhesions and was quite boggy. A partial central hepatectomy was performed; a 3 mm biliary duct was found communicating to the cyst only with no drainage to the minimal liver parenchyma that was removed (). The cyst was resected en-bloc and was sent for permanent section (), which diagnosed the tumor as a low-grade mucinous cystic neoplasm measuring 8.5 × 7.2 × 6.4 cm. Microscopy revealed a smooth-walled, multiloculated cyst filled with a yellow-golden, semi-transparent and mucinous fluid (). The cyst was lined by a mucinous epithelium with ovarian-type stroma. No high-grade dysplasia or malignancy was identified. The postoperative course was uneventful, and the patient was discharged on postoperative day 6. At the 4-week postoperative visit, the patient was healing well with some incisional soreness; patient was seen for removal of her biliary endoprosthesis, and ERCP found left sided intrahepatic duct biliary dilatation including a small biliary fistula from the left system () with the appearance of torque on the extrahepatic biliary tree. A biliary endoprosthesis was left in place, and the patient returned for repeat ERCP 4 weeks later with resolution of all findings (). At present, patient remains alive with no signs of recurrence. |
A 32-year-old white woman, who was undergoing transgender body modifications and no previous medical history, presented to our emergency department (ED) with a chief complaint of right forearm pain, associated with redness and blistering. The patient was a tattoo fanatic who underwent solar branding on her right forearm approximately 2 weeks before presentation. The patient first noticed the blistering of the branded area 3 days after her body modification. Over the next few days, the entire branded area became very warm, swollen, painful to touch, and developed cellulitis with weeping blisters. Given her dire situation, the patient finally sought medical care in the local urgent care center. The patient was diagnosed with a superficial infection of her right forearm full thickness burn and prescribed oral clindamycin. The patient continued to develop painful blisters for the next week and did not notice any improvements in her cellulitis despite the antibiotic treatment, prompting her to come to our ED for a second medical opinion.\nDuring her physical examination, the patient was noted to have painful blisters, redness, and swelling of the right forearm and hand (Figure ). She did have a full range of motion of the hand; however, there was mild pain noted on the flexion of the hand. There were no signs of sepsis, and her vital signs and her laboratory panel which included white blood cell count were all within normal limits. The rest of the physical examination was also unremarkable. The patient was admitted to the Burn Surgery Service for intravenous (IV) antibiotics and possible operative interventions.\nThe patient was started on IV clindamycin 600 mg every 8 hours, and once adequate pain control was achieved, a bedside debridement was performed in the Burn Unit. The following day, the patient was taken to the operating room for tangential excision of her wound and split-thickness skin autograft (STSG) placement onto her right forearm. The left anterolateral thigh was chosen as the donor site. The grafted site was dressed with xeroform, followed by a layer of bacitracin ointment and then wrapped with Kerlex and Coban dressings. A right forearm elbow splint was fitted and placed by the occupational therapy service for further graft protection. Postoperatively, the patient was transferred back to the floors without complications, and antibiotics were stopped. The right forearm dressing was taken down on postoperative day 4 to evaluate the STSG. The entire graft was viable and taken. Subsequently, the graft staples were removed on postoperative day 5.\nThe patient recovered exceptionally well and was discharged home a few days later. Two weeks later, the patient returned to the burn clinic for a follow-up appointment. The skin graft and the donor site healed remarkably well (Figure ). The patient reported no pain and had a full range of motion of the hand and wrist. The patient was discharged from the burn service and advised to use over-the-counter moisturizer or lotion as needed. |
A 36-year-old female leucoderma patient sought treatment complaining of a lesion located between teeth 21 and 22, painless and compromising the aesthetics of her smile (). Intraoral physical examination showed an injury inserted in the interproximal gum, measuring 1.2 × 0.9 × 0.5 cm on the facial surface and 0.7 × 0.5 × 0.3 cm in the palatal face, exophytic and nodular. The radiographic examination showed no related changes ().\nSurgical techniques were performed as described below: after local anesthesia with 2% lidocaine with epinephrine at a concentration of 1 : 100,000, the excision of the lesion was proceeded with a 15C scalpel blade (Figures and ), removing all the gingival and periodontal tissue involved, followed by scaling and root planing of the same teeth ().\nAfter excision of the lesion, the removal of a free gingival graft from the palate was performed, which was placed in the exposed conjunctive tissue area to recreate the band of keratinized tissue lost as a result of the lesion itself and its excision. The graft was taken from the palate and its format was similar to the open area of the receiving tissue (). The apical and coronal dimension and thickness were measured so that it could be suitable and uniform. The graft was sutured along its entire length (). Digital pressure was performed with saline moistened gauze to remove any blood clot and maintain the graft in intimate contact with the recipient bed.\nThe material obtained from excisional biopsy was sent for pathological analysis. Histologically, the lesion showed an intact squamous epithelium and in the lamina propria a highly cellular component of fibroblasts was observed with central area of calcification, setting the diagnosis for peripheral ossifying fibroma ().\nThree months after the procedure (), a second surgical procedure was performed in order to cover the exposed root of tooth 22. The biomechanical preparation of the surface of the root was accomplished with scaling and root planing () and application of EDTA 24% neutral pH (Pref-Gel®, Straumann). The coronally advanced flap technique, described by de Sanctis and Zucchelli (2007) [], was the selected technique: two horizontal beveled incisions were performed, mesial and distal to the recession, located at one end of the anatomical papillae and equal to the height of the recession plus 1 mm; two oblique incisions, slightly divergent, starting at the end of the two horizontal incisions and extending to the alveolar mucosa (). The coronal portion of the flap is partially divided, while the portion apical to the recession is a full thickness flap, exposing 3-4 mm of bone (). The relaxing vertical incisions are elevated in partial thickness. Apical bone exposure is held in the partial thickness flap, ending where it is possible to passively move the flap in coronal direction and coronally in the cementum-enamel junction. At this time simple sutures are performed throughout the flap ().\nAfter the initial results were achieved, root coverage of 100% was obtained after 6 months (), and suitable gingival contour was promoted which prevented the development of a mucogingival defect or root exposure with its functional and aesthetic consequences. After five years () the margin remained at its initial position, with no relapse in the exhibition of the cementum-enamel junction; and tissues were stable and characterized by color harmony, demonstrating the success of the chosen techniques. |
A 76-year-old woman with a 1-month history of epigastric discomfort was referred to the Department of Gastroenterology. She was being treated as an inpatient in the department of neurology after a diagnosis of Parkinson disease. Twenty days previously, she was found to have a gastric ulcer after having undergone upper gastrointestinal endoscopy in a local clinic. She continued to complain of abdominal discomfort, although she had been taking ulcer medications. On admission, her physical examination findings and laboratory data were unremarkable. A small amount of left pleural effusion was noted on a chest radiograph, and an abdominal radiograph showed no abnormality. Because she had upper respiratory symptoms, including cough, on admission, she had received antibiotics for suspected pneumonia. During her hospital course, a few days after starting the antibiotics, she showed improvement in respiratory symptoms. Symptoms associated with Parkinson disease and other upper respiratory symptoms had been well controlled with an oral antiparkinson drug and oral cough syrups followed by 3-day intravenous antibiotics. She did not report any upper respiratory symptoms, so the pleural effusion was expected to disappear soon.\nUpper gastrointestinal endoscopy was performed to evaluate her symptoms. Endoscopic examination demonstrated a slightly elevated lesion (4 cm) with multiple shallow erosions, absence of a definite ulcer scar, and bridging or converging folds over the anterior wall of the antrum (type IIa) (). The endoscopic ultrasonographic (EUS) image revealed that the diffuse thickened hypoechoic lesion was limited to the mucosal layer, with no irregular narrowing or budding sign (). Histopathological examination of gastric biopsies showed a moderately well-differentiated adenocarcinoma. Abdominal computed tomography (CT) revealed no evidence of gastric wall thickening, enlargement of abdominal lymph nodes, or distant metastasis. The lesion was diagnosed as mucosal gastric cancer. She was transferred to our department for ESD, which was performed according to the expanded ESD criteria.\nOn the day after ESD, the patient reported fever and frequent cough. A follow-up chest radiograph showed an increased amount of left pleural effusion (). Aspiration pneumonia was suspected, and a diagnostic thoracostomy was performed along with initiation of intravenous antibiotic therapy.\nThe ESD specimen revealed a round, brownish, elevated lesion measuring 6×4×0.4 cm, with no surface change. Microscopic examination showed that the tumor was limited to the submucosal layer, with no involvement of the deep resection margin. The tumor showed clusters, or nests, of moderately well-differentiated adenocarcinoma, mostly confined to the large, dilated lymphatic channels with adjacent foci of stromal invasion (). The mucosal layer also showed multifocal lymphatic tumor emboli without stromal invasion.\nAnalysis of the left pleural fluid revealed the following findings: red blood cells, 32,000/mm3; white blood cells, 1,120/mm3; neutrophils, 4%; lymphocytes, 38%; adenosine deaminase activity, 15.4; and pH, 7.36. The ratio of pleural fluid protein to serum protein was 0.98. The ratio of pleural fluid lactate dehydrogenase (LDH) to serum LDH was 0.66. Metastatic adenocarcinoma cells were unexpectedly detected by cytological examination of the pleural effusion ().\nTo determine the primary site of metastatic adenocarcinoma, immunohistochemical (IHC) staining tests and positron emission tomography (PET)-CT were performed. EGC metastasis to the pleural space is rarely observed, but the ESD specimen from this patient showed this unusual histopathological feature.\nThe results of IHC staining tests were as follows: D2-40, positive; cytokeratin 7 (CK7), positive; CK20, negative; CD56, negative; synaptophysin, negative; human epidermal growth factor receptor-2, negative; and thyroid transcription factor-1, negative. The test results were negative for mucin (MUC) 1 and MUC2, and positive for MUC5A and MUC6.\nPET-CT showed a focal abnormal hypermetabolic lesion in the aortopulmonary window, most likely representing malignant uptake; the standard uptake value (SUV) was 8.76. The SUV was elevated in the left lower lobe of the lung, with pleural effusion; the SUV of 4.43 indicated probable benign uptake.\nAfter all analyses, instead of gastric metastasis, we concluded that the proper diagnosis was primary EGC metastasis to the pleura, with accompanying unusual histological findings. We planned to start chemotherapy, but she refused further treatment and wanted to undergo conservative management. She died 1 year after the diagnosis. |
A 45-year-old male was transferred to our facility from an outside institution with the diagnosis of TEF and the presence of a possible foreign body seen on CT scan of his chest. The patient gave a history of recurrent pneumonia and cough in the last few months. Prior to presentation to the hospital, the patient had finished a course of antibiotics. Despite this, he complained of increasing cough over the prior week but no history of dysphagia, hemoptysis, hematemesis, or chest pain. The physical exam was unremarkable except for inspiratory crackles and diminished breath sounds heard at the lung bases. Axial CT images of the chest both with and without intravenous contrast were obtained. A small, linear, walled air collection was noted just above the carina tracking towards the esophagus, a finding suspicious for a tracheoesophageal fistula ().\nUnder sedation and spontaneous ventilation, bronchoscopic and endoscopic examinations were performed. A hard, disc-shaped foreign body was impacted in the esophagus and was also protruding into the lumen of the distal trachea (see ). Gentle attempts to remove the FB were unsuccessful and aborted. Two days later the patient underwent a thoracotomy for foreign body removal and TEF repair. Mask ventilation was easy with low positive inspiratory pressures. A left double lumen tube was placed via direct laryngoscopy. With fiberoptic bronchoscope (FOB) allowing for direct vision, the bronchial lumen was advanced into the left main stem bronchus with subsequent deflation of the right lung. With the double lumen ETT in position, ventilation of only the left lung avoided any air insufflations through the fistula into the esophagus (due to the anatomical location of the fistula 1 cm above the carina). The patient was placed in the left lateral decubitus position and the ETT placement again confirmed with the FOB. Surgical approach was via right thoracotomy, with removal of the foreign body and primary repair of both the trachea and the esophagus. After the surgery the patient resumed spontaneous ventilation and was extubated prior to transfer to the ICU. Pathology confirmed that the foreign body was a dental plate. Later, the patient revealed that after an appendectomy 5 years ago, he could not find his partial dental plate. |
A 27-year-old Hispanic man with no significant prior medical history presented to the emergency room with shortness of breath and a cough for three weeks. He also complained of mild precordial chest pain and generalized fatigue. His family history was contained type 2 diabetes mellitus in both parents but reported no history of cancer or genetic diseases. His social history revealed that he worked as a gardener, was married with two kids, was a non-smoker, occasionally consumed alcohol and had no history of drug abuse. He gave no history of any psychological illnesses. A physical exam revealed sinus tachycardia, distant heart sounds and elevated jugular venous pressure and a normal blood pressure. Chest X-ray showed an impressive mediastinal enlargement. Transthoracic echocardiogram (TTE) showed a large pericardial effusion (\n) and collapse of the right atrium during diastole along with respirophasic changes consistent with cardiac tamponade physiology. The patient underwent an emergent therapeutic pericardiocentesis that yielded hemorrhagic fluid. Cytological analysis of the fluid revealed no malignant cells. Computed tomography (CT) of the chest showed persistent pericardial effusion and a left anterior mediastinal mass (\n). Work-up for possible metastases performed using Gallium scintigraphy showed no evidence of involvement of any other organs. Left anterior thoracotomy, pericardial window and left anterior mediastinotomy were done, revealing a well-encapsulated gelatinous tumor originating from the pericardium. The tumor grew from the inner surface of the pericardium near the origin of the superior vena cava and ascending aorta (\n). A portion of the pericardium where the tumor was attached was resected and the tumor was biopsied. Complete resection was not attempted due to the proximity and attachment to the great vessels.\nReview of pathology on routine hematoxylin-eosin stain showed that the tumor was monophasic and was composed of numerous spindle to oval cells in a fascicular growth pattern. The cells showed scant cytoplasm, oval to spindle nuclei with granular chromatin and with inconspicuous nucleoli (\n). The tumor was mitotically active and showed 3–4 mitosis per high power field (\n). Some areas showed myxoid background (\n) and others showed large areas of necrosis (\n). Immunohistochemistry showed that the tumor cells were strongly positive for BCL-2 (\n), CD 99 (\n), Vimentin (\n), FLI1 (\n) and neuron-specific enolase (NSE) (\n). Stain for epithelial membrane antigen (EMA) showed focal scattered positive cells (\n). Muscle markers (smooth muscle actin, desmin and myogenin), neural and melanoma marker (S-100), epithelial markers (AE1/AE3), CD57, CK5/6 and p63 were negative. Based on morphology and immunostaining results, a diagnosis of monophasic synovial sarcoma was made. Fluorescent\nin-situ hybridization (FISH) was done to confirm the diagnosis and to exclude Ewing sarcoma. It showed the presence of SS18 (SYT) gene rearrangement on chromosome 18q11, substantiating the diagnosis. Also the FISH assay for rearrangement or dosage abnormalities of the EWSR1 oncogene at Ewing sarcoma locus 22q12 was negative.\nThe patent was treated neoadjuvantly with high dose ifosfamide (2000 mg/m\n2/day intravenous over 3 hours for 4 days) and mesna (1200 mg/m\n2) repeated every 21 days between July and December 2012. On repeat CT imaging prior to planned surgical excision, the tumor had substantially decreased in size (> 50%) and the patient was noted to have elevation of left hemi-diaphragm indicating likely involvement of the left phrenic nerve.\nA median sternotomy was performed and access to the heart and great vessels was obtained. The pericardium was opened and the tumor was seen abutting the bifurcation of the pulmonary artery and distal aortic arch, and involving a portion of the pericardium including the left phrenic nerve. The portion of the mass that was attached to the distal part of the pulmonary trunk and a lateral portion of the pericardium were resected. The tumor appeared to encase the left phrenic nerve consistent with the finding of elevation of left hemi-diaphragm on CT scan. This nerve was transected and its edges were controlled with hemoclips. The hemoclips also served as markers for possible future radiation treatment. The entire outline of aortic arch and the pulmonary artery were visible after removal of the thymus and the tumor, with no evidence of direct invasion. Once the tumor was mobilized laterally towards the left lung, it showed possible involvement of the wedge of the left upper lobe. The lung parenchyma was divided and a portion of the lung was transected from the left upper lobe. The entire tumor was removed en bloc with the surrounding tissue. The level 5 (subaortic) lymph node was also resected and sent for pathology.\nThe resected mediastinal mass (6.5 × 4 × 3 cm) was confirmed to be a monophasic synovial sarcoma on histology and immunohistochemistry. The adjacent lung was not involved by sarcoma. The three lymph nodes resected along with the mass were benign with non-necrotizing granulomas. The level 5 lymph node also showed non-necrotizing granulomas and was negative for malignancy. One of the resection margins was positive for malignancy. The patient recovered well after the repeat thoracotomy and EBRT was initiated 1-month post surgery. He had no recurrence of tumor on CT imaging at the first and second three-month intervals post surgery. Chest CT repeated at 9 months (third three-month interval) showed local recurrence. The patient was noted to have an anterior mediastinal mass extending to the left upper lobe of the lung and a left pleural effusion. There was no evidence for any distant metastases. The patient underwent resection of the mass and left upper lobectomy, left video-assisted thoracoscopy, drainage of pleural effusion and total decortication of left lung. Pathology confirmed the recurrence but the resection margins were free of malignancy. The pleural fluid was also negative for malignant cells. Chemotherapy was reinitiated with docetaxel (100mg/m\n2) every 21 days along with steroid premedication with dexamethasone (4mg) for 5 days prior to treatment. This resulted in minor improvement on repeat CT imaging, but more importantly there was no further symptomatic worsening. The patient is clinically stable 18 months after diagnosis. |
A 72-year-old female presented with exertional chest pain for one month. Chest pain was retrosternal and occasionally radiated to left arm. Patient had history of significant coronary artery disease with a critical lesion of the left main coronary artery ostium, posterior descending artery (PDA), first diagonal artery, and therefore underwent coronary artery bypass surgery three years ago prior to current presentation. She had left internal mammary artery (LIMA) graft from left subclavian to distal left anterior descending (LAD) artery, which bypassed the proximal LAD lesion. She also had two aortocoronary saphenous vein bypass grafts on to the PDA artery and another to the diagonal. A prior catheterization for abnormal stress test (moderate inferior ischemia) revealed an atretic LIMA graft, however patent SVG graft to diagonal artery maintained TIMI-3 flow to LAD via the diagonal artery. The SVG graft to PDA was occluded with significant disease in right coronary artery. This correlated with the area of ischemia on the nuclear stress test and was thus treated with percutaneous coronary angioplasty. On this presentation her vitals were stable and remaining physical examination was grossly unremarkable. EKG showed normal sinus rhythm without evidence of ischemia. Cardiac enzymes were also normal. Given the nature of pain and her prior history of significant CAD, further risk stratification was done by performance of a vasodilatory nuclear stress test. The myocardial perfusion study revealed uniform perfusion, however transient ischemic dilation (1.39) was observed which was indicative of possible balanced ischemia due to presence of multi-vessel disease. Therefore the patient underwent cardiac catheterization which showed an eccentric 70% lesion at ostium of the left main. The first vein graft to right coronary artery was totally occluded with patent stents in the RCA. The second venous graft to the diagonal vessel was widely patent and was providing TIMI-3 flow to the LAD as well. In the proximal aspect of this vein graft severe spasm was noted in multiple fluoroscopic projections and was associated with chest pain (). The patient’s symptoms and spasm resolved after administration of intracoronary (IC) nitroglycerine (). The angina symptoms in our patient were therefore attributed to spasm of the saphenous vein graft. Patient was subsequently started on amlodipine and isosorbide mononitrate which resulted in long-term resolution of her symptoms. |
The patient is a 15-year-old Caucasian female who was diagnosed with CRMO in 2007 at 5 years in the context of right thigh pain. provides a timeline of the patient's symptoms and course of disease. Magnetic resonance imaging (MRI) showed multifocal abnormal bone marrow signal in the right femur, the left femoral neck, and the proximal epiphysis and metaphysis of the right tibia, which was associated with osteolysis and callus formation (). Radio-nucleotide bone scan found increased uptake involving the left sacrum, left proximal femur, and femoral neck region as well as the midshaft of the right femur and the proximal right tibia. Bone biopsy of the lytic lesion involving the midshaft of the right femur was not consistent with malignancy and showed red blood cells and scattered neutrophils and lymphocytes. Over the next several years (2007–2015), she was followed by orthopedic surgery and was treated only with intermittent ibuprofen as needed for pain. She was noted to have a leg length discrepancy at the age of 7 years, and in 2014, at the age of 12 years, she had surgery to fuse the growth plate to prevent right leg growth (right leg was 4.5 cm longer than her left leg at that time).\nShe established Rheumatologic care in March 2015 at age 13, and right quadriceps muscle atrophy and failure to thrive with a weight and height under the third percentile were noted at this time. Bone scan revealed increased uptake in the right femur and asymmetry of activity in the growth plates of the knees and ankles with decreased activity in the right side compared to left. She was treated with naproxen 250 mg twice daily (8.7 mg/kg BID) and prednisone 20 mg per day (0.7 mg/kg), tapered by 5 mg weekly. Two months later, prednisone was discontinued, and she continued to have good control of her leg pain on NSAID monotherapy. After a few months, she developed new diarrhea with vomiting and weight loss. Her blood pressure was elevated, and a renal ultrasound and electrocardiogram were unremarkable.\nShe was referred for Gastroenterology evaluation and was found to have positive stool occult blood along with a perirectal skin tag. Laboratory studies revealed anemia with a hemoglobin of 7.1 g/dL (nl 12–16 g/dL), thrombocytosis with platelets of 744 k (nl 150–450 k), erythrocyte sedimentation rate (ESR) of 69 mm/hr (nl 0–10 mm/hr), and C-reactive protein (CRP) of 129 mg/L (nl 0–3 mg/L). There was no family history of autoimmune disease, and tuberculin testing was negative. Colonoscopy revealed pancolitis with crypt inflammation and crypt abscesses with no granuloma consistent with ulcerative colitis (). NSAID therapy was discontinued, and treatment for UC with prednisone 1 mg/kg/day (40 mg) tapering by 5 mg weekly along with sulfasalazine was initiated. Infliximab 3 mg/kg infusions were added the following month.\nAt the visit for her second infliximab infusion, she presented with tachycardia, a blood pressure of 230/190, and headache and was admitted to the pediatric intensive care unit for hypertensive emergency requiring nicardipine infusion. Her examination was notable for right-sided Horner's syndrome, decreased right leg pulse pressure, and an abdominal bruit. Echocardiogram showed a small pericardial effusion with reduced left ventricular ejection fraction. Laboratory studies revealed negative ANA and ANCA screens, normal C3 and C4, and normal von Willebrand factor antigen. CT angiogram (CTA) of the abdomen and pelvis showed narrowing of the mid-aorta, proximal renal arteries, celiac artery, and superior mesenteric artery (Figures –). CTA of the chest showed marked descending thoracic and abdominal aortic wall thickening with progressive luminal narrowing and wall thickening of the visualized portion of the right common carotid artery and celiac trunk, enlarged left atrium, left ventricular hypertrophy, and a small pericardial effusion (). MRI/MRA brain showed anterior and posterior circulations of the brain were without occlusion or aneurysm with the patent carotid and vertebral arteries of the neck (Figures and ). Given these findings, she was diagnosed with TA complicated by middle aortic syndrome. She was started on metoprolol 50 mg daily (1.5 mg/kg daily), amlodipine 5 mg daily, famotidine 20 mg twice daily, aspirin 81 mg daily, and increased dose and frequency of infliximab from 3 mg/kg every 8 weeks to 5 mg/kg IV every 4 weeks along with prednisone 20 mg daily (0.6 mg/kg daily) and mesalamine 1 g BID. The patient continued infliximab every 8 weeks rather than every 4 weeks as recommended.\nMagnetic resonance (MR) angiography of the chest, abdomen, and pelvis and Cardiac MR three months later showed luminal narrowing of the distal thoracic and upper abdominal aorta similar to previous CT studies, stenosis of origin of celiac axis, stenosis of proximal superior mesenteric artery (SMA), and moderate stenosis of bilateral proximal renal arteries. Echocardiogram noted concentric LVH with mildly reduced function, measuring 45%, and normal coronary arteries.\nAt this point, the patient transferred her care to our rheumatology clinic, and despite treatment with infliximab 5 mg/kg every 8 weeks, she continued to report right thigh pain and developed new inflammatory arthritis of the left ankle and increased inflammatory markers with an ESR of 55 mm/hr (nl 0–20 mm/hr) and an elevated CRP of 73 mg/L (nl < 3 mg/L). Additionally, interval imaging found new wall thickening around the right common carotid artery. This was concerning for uncontrolled TA and CRMO activity. There was an unfortunate delay in treatment escalation due to social circumstances. Two months later, she started treatment with parenteral methylprednisolone 1 gram weekly for 8 weeks, and her dose of infliximab was increased from 5 mg/kg to 10 mg/kg every 4 weeks. Based on adult data demonstrating a positive response to higher doses of infliximab, the decision was made to increase the dose of infliximab instead of trying another TNF inhibitor.\nMultidisciplinary evaluation at Boston Children's Hospital, Center for Middle Aortic Syndrome by neurosurgery, nephrology, and rheumatology, led to recommendations of a prednisone dose increase and the addition of methotrexate 15 mg/m2 weekly to infliximab 10 mg/kg every 4 weeks. At this time, her echocardiogram revealed moderate left ventricle dilation and mildly depressed left ventricular systolic function. One month later, repeat head and neck CTA showed progression of her right carotid artery stenosis to 80%. Her ESR had normalized, and CRP decreased to 7.8 mg/L at this time. Given her worsening carotid artery stenosis, infliximab was increased from 10 to 15 mg/kg every 4 weeks. On a combination of moderate dose prednisone, weekly methotrexate, and infliximab, she denied joint pain, swelling, abdominal pain, diarrhea, or blood in stool. Follow-up brain MRI/MRA three months later was normal. Repeat chest and abdomen MRA showed all of the areas of stenosis appeared to be stable and inflammatory markers had normalized.\nCurrently, her clinical course is stable without further anatomic progression, and she has normal inflammatory markers. Thus, she has continued on her current regimen with infliximab 15 mg/kg every 4 weeks and methotrexate 15 mg/m2 once weekly. Prednisone decreased gradually and discontinued. She has not developed any infectious sequelae on this regimen. Her blood pressure is stable on carvedilol alone. We continue to assess blood work monthly. Given her use of prednisone, vitamin D level was followed and found to be low, and she has started on a vitamin D supplement. Dilated ophthalmologic exam was normal. Repeat MRI of the lower extremities and bone scan show that her CRMO lesions are inactive. Repeat MRI/MRA of the brain, chest, abdomen, and pelvis in June of 2018 show stable changes without the need for stenting. Given the predisposition for autoinflammatory conditions in this patient, the authors are considering genetic testing in search of a monogenic cause that may support a unifying diagnosis. |
A 76-year-old female with a history of dementia presented to her local physician complaining of a 1-month history of right nipple pruritus. Topical steroids resulted in minimal improvement, and the patient consequently consulted the Department of Dermatology at our hospital. Upon examination, there was no evidence of a palpable breast mass or a swelling of the axillary lymph nodes. The right nipple appeared swollen and inflamed with occasional bleeding, but the right mammary areola was normal (Fig. ). Mammography findings were normal, but ultrasonography revealed a swelling of the right nipple, an internal punctiform hyperechoic area, and rich blood flow.\nBased on these clinical findings, inflammatory changes in the right nipple and Paget's disease of the breast were considered as differential diagnoses, and a punch biopsy of the right nipple skin was performed. Histopathology revealed the presence of proliferating, highly atypical squamous cells accompanied by occasional mitotic figures. We did not detect Paget cells or an invasion of atypical cells beyond the basement membrane into the ruptured interstitium. Therefore, the patient was diagnosed with Bowen's disease.\nThe patient desired a simple resection, and a surgical plan to resect and plicate the right nipple under local anesthesia was developed. A 5-mm horizontal margin was established beginning at the right nipple, which was resected down to the level of the areolar subcutaneous fat proximal to the lactiferous duct. Histopathological analysis of the resected tissue revealed tumor cells within the ductal epithelium, and the deep margin was positive for malignancy (Fig. , ). The patient was consequently referred to the Department of Breast Surgery of our hospital for additional resection.\nMRI of the breast showed a relatively limited area of contrast in the operated site; however, we were unable to establish whether this signal resulted from postoperative changes or the presence of residual lesions. Nevertheless, it was highly likely that a progression of the lesion within the lactiferous duct was limited to the vicinity of the nipple, and a partial mastectomy was indicated. A fusiform skin incision was made around the areola and the previous surgical scar, and the mammary gland was dissected to the level of the pectoralis major. Histopathology revealed that intraductal tumor cells spanned 8 mm of the tissue, but all resection margins were negative for cancer (Fig. ).\nThe patient refused radiation therapy of the right residual breast because of treatment-related anxiety and her history of dementia. One year postoperatively, the patient shows no signs of recurrence and is undergoing follow-up on an outpatient basis. |
Mr P was a married man in his 40s with one child. He had served in the UK army and was deployed to the Bosnian conflict in the mid-1990s. During his deployment he was stationed at a morgue that had been set up to aid the identification of bodies recovered from a mass burial site. He described how the morgue contained a large number of bodies in various states of decomposition, with varying degrees of physical trauma. He reported re-experiencing fragmented intrusive images of the morgue and daily emotionally distressing nightmares of which he could not recall the content. He experienced high levels of anxiety that triggered his dissociative symptoms. These appeared to function as a coping strategy to avoid distressing emotions connected to his memory of the morgue. Other avoidance symptoms included feeling detached from people around him and feeling emotionally numb. Mr P avoided family occasions and busy places which could trigger his hyper-arousal symptoms and had never spoken to anyone about his experiences. He reported coping historically through the use of alcohol to block out his emotions. His avoidance behaviours had maintained his PTSD symptoms.\nAt the start of therapy sessions, Mr P was reluctant to talk about his past experiences stating he only felt able to discuss vague details about his intrusive memories. Mr P attempted to use physical objects such as stones and leaves, or his e-cigarette, as grounding strategies in the clinic room. However, he also reported feeling unable to tolerate any emotions connected to the events and found it difficult to identify peri-traumatic cognitions. Mr P appeared to experience intrusions of one particular fragmented image and could not recall any other contextual information from before or after this moment.\nIn both cases Mr C and Mr P experienced difficulties in engaging with traditional methods of imaginal reliving and prolonged exposure, due to high levels of avoidance and low thresholds for dissociation, and subsequently had difficulties accessing their cognitions for updating. For both clients the prior use of multiple grounding methods in the therapy room were unsuccessful. They experienced their traumatic memories as fragmented images with a sense of ‘nowness’ (), which indicated a distinct lack of contextualization.\nAn adaptive approach was then trialled to address these hindering factors. This involved three stages: (1) walking the client through the imaginal scene outdoors to address dissociation, (2) viewing the imaginal scene from multiple perspectives to facilitate contextualization of the memory and (3) identifying and reappraising the cognitions with frequently used approaches in trauma-focused therapy. |
An 11-year-old boy was struck in his right eye by a barbed fish hook while fishing with friends. The father cut off the fishing line and tried to remove the hook but failed. The child was brought to the hospital 12 hours after the injury. On examination, he had the best-corrected visual acuity (BCVA) of perception of light and projection of rays in all four quadrants in right eye. The fish hook had penetrated the cornea obliquely, 3 mm away from limbus at 9'0 clock []. Slit-lamp examination of the RE revealed, a shallow irregular anterior chamber filled with exudates due to which tip of fish hook was not clearly visible []. Fundus examination showed no fundal glow. B scan was not done as patient was highly unco-operative for examination. It was penetrating type of open globe injury of grade 4 involving zone 1 in right eye. The left eye was normal.\nThe patient was given inj.Tetanus Toxoid 0.5 cc intramuscular Surgical Technique and started on oral antibiotics and advised for corneal FB removal and corneal tear repair under general anaesthesia. A written consent was obtained from the parents for removal of the fish-hook under general anaesthesia (GA). Under GA, first we tried to remove barbed fish hook with help of forceps through same entry wound but the attempt was failed because of lack of visibility of tip due to exudates filled in irregular anterior chamber and incarceration of iris tissue in the tip. Then we made side port incision at 2 O clock at limbus and anterior chamber tap was taken, then anterior chamber was washed with help of saline and viscoelastic injected []. The iris tissue plugged in the tip was separated with the help of iris repositor []. The entry wound was extended with help of 15 number blade to facilitate smooth removal of tip of barbed fish hook [].[] The configuration of the wound after enlarging it with the blade was curvilinear. After filling anterior chamber with viscoelastic material, fish hook was removed from same wound by oblique movement (cut-it out technique)[] without damaging adjacent structures [], the wound sutured with six interrupted10-0 nylon sutures [] and side port hydrated. Bandage contact lens (BCL) was placed at the end as there was epithelial damage during removal of fish hook []. On first postoperative day, the best-corrected visual acuity (BCVA) of perception of light and projection of rays in all four quadrants in right eye. The corneal sutures were intact and the anterior chamber was showing presence of exudative membrane with superior one third filled with air bubble []. B scan of right eye was normal with attached retina. The patient was started on topical antibiotic (Moxifloxacin 0.5% eye drops 6 times a day) and cycloplegic (Atropine 1% eye drops three times a day) and oral antibiotics were continued for 1 week duration. After confirming micro reports of exudative material after 3 days (i.e., no evidence of infection), the patient was started on topical steroids (prednisolone acetate 1% eye drops 6 times a day).\nAt 1 week of follow-up, the best-corrected visual acuity (BCVA) was perception of light and projection of rays in all four quadrants in right eye. The intraocular pressure was digitally normal. The BCL was removed. The eye showed resolving exudative membrane in anterior chamber with damage to the iris at the pupillary margin and a traumatic cataract was visible []. The parents were counselled for the need of a cataract extraction in the future. |
Our patient is a 16-year-old boy who experienced a sting at the back of his right hand from P. physalis at the end of a dive close to the coast of a Spanish Mediterranean island. The animal was identified by one of the dive instructors and had also occurred before on the coast nearby, which was reported by a popular newspaper []. He had no prior medical history and no history of atopy or any known allergies. The family history is negative for dermatological conditions or allergies except for a mild seasonal allergic rhinitis of the boy's father. During the dive, the patient was protected on his head and body by a diving suit. The sting occurred after reaching the water surface while pulling off his equipment, having his hands unprotected. Immediately after the sting, the skin was carefully dried and a class II (referring to WHO classification) topical corticosteroid ointment was applied to the affected area. The hand was also cooled over 2 h with cooling packs, and contact with freshwater was avoided. 1.5 h after contact with P. physalis the affected hand was also immersed in vinegar and immobilised. Shown in Figure is the affected hand 2 h after primary contact with P. physalis (on October 23, 2018, at 17: 37). The affected area was reported to be initially very painful with a burning sensation, and over the first 12 h, the right forearm and lower upper arm were also moderately sore. The skin lesion on the back of the right hand with pain and itching sensation continued to be present over the next 78 days after the event.\nTopical class II corticoid treatment was used until the end of the first week and discontinued because of lack of improvement. During the following weeks, sharply demarcated erythematous infiltrated plaques on the back of the hand remained. Pain, especially when tensioning the skin by making a fist, and recurrent itching of the skin remained over the next 10 weeks. Due to the pain when stretching the skin, the movement of the hand was also impaired. At week 10, also superficial ulcerations developed.\nBecause there were no signs of spontaneous healing of the skin lesions, an attempt was made to treat the affected erythematous skin area with extracorporeal shock wave therapy (ESWT) starting on day 72 after the event as shown in Figure (on January 8, 2019).\nThe English language public literature was searched using PubMed. ESWT treatment was performed in accordance with the manufacturer's operating instructions []. A planar/defocused shock wave (device: Storz Medical ESWT Duolith® SD1, T-Top version, C-Actor II handpiece, R-SW 1 V-Actor applicator) was used. Our treatment protocol consisted of 8 sessions with a focussed shock wave generated by a Duolith® SD1 machine plus a C-Actor® applicator, which defocuses the waves to adjust the penetration depth and energy applied. Treatment was administered twice per week. The focal depth of shock waves was 0–10 mm due to a 30-mm standoff on the C-Actor applicator to optimise the penetration depth with a total energy applied for each impulse of 0.33 mJ/mm2; wave frequency was 6 Hz, and duration of each session was 15 min (2,000 pulses for a session per 133 impulses/min, total energy 21.58 J applied).\nThe first shock wave therapy took place on January 8, 2019 (day 72). After the second consultation on January 11, the patient reported that the chronic pain and itching of the skin was already reduced approximately by half. ESWT treatments as described above of approximately 15 min duration were performed 2 times a week (Tuesdays and Fridays) over 4 weeks. The treatment itself was painless and tolerated very well as no side effects were reported. As shown in Figure , after 4 weeks the erythematous plaques had considerably been reduced and there were also no more signs of ulcerations.\nESWT treatments were stopped at day 110. The described skin changes on the back of the right hand had completely disappeared without any signs of scarring. Even at a follow-up examination 1 year after the end of treatment and more than 18 months after the initial event, there were no signs of reoccurrence. |
A 57-year-old male who had no family history of cancer complained of acute abdominal pain and fever and visited his local hospital. He was diagnosed with gastric perforation and referred to our hospital. We performed emergent surgery for peritonitis caused by a gastric perforation. We evaluated the abdominal cavity laparoscopically and found some cloudy ascites in the abdomen and a pin hole perforation at the anterior wall of the gastric antrum. Because the gastric wall around the perforation was thick and did not have any serous changes, it was difficult to assess whether the cause of the perforation was tumor related. The cytodiagnosis of ascites did not show any tumor cells. There was also a white nodule in the lateral segment of the liver, which was suspected to be a metastatic liver tumor. We performed a primary closure of the defect, applied an omentum flap and washed the abdominal cavity with 10 l of normal saline. The operation time was 120 min, and the volume of blood loss was 10 ml. The postoperative course was uneventful. We performed upper and lower endoscopy, which showed type II tumors in the gastric body (poorly differentiated adenocarcinoma, HER2 score 2+) (Fig. a, b) and transverse colon (well-differentiated tubular adenocarcinoma, RAS mutation) (Fig. c, d), and we considered that the gastric perforation was related to the presence of advanced gastric tumors. Furthermore, the CT showed irregular wall thickness with ulcers in the gastric body, which were suspected to be gastric cancer with lymph node metastases at station no. 3, irregular wall thickness of the transverse colon (Fig. a–c), which was suspected to be colon cancer, and nodules in liver segments 2/3 (60 mm) and in segment 6 (57 mm) (Fig. a, b), which were suspected to be liver metastases. We retrospectively reevaluated the CT findings from a local hospital and detected liver nodules in segment 2/3 (35 mm) and segment 6 (26 mm), and the tumors had dramatically grown in size in only 2 months. [18F]-fluorodeoxyglucose (FDG) positron emission tomography (PET)/CT showed colon cancer (maximum standardized uptake value (SUVmax) 14.4) and multiple liver metastases (S2/S3, unclear SUVmax; S6, SUVmax 11.3), but 18F-FDG uptake was not found in the gastric tumor and lymph nodes (Fig. a–d). The serum blood tests showed normal tumor marker levels (carcinoembryonic antigen, 4.6 mg/dl; carbohydrate antigen 19-9, 20.7 mg/dl) and normal liver function. We diagnosed the patient with synchronous cStage IV (cT3cN0cM1) transverse colon cancer with multiple liver metastases and cStage IIB (cT3cN1cM0) gastric cancer according to the 7th edition TNM classification []. Because of the gastric cancer perforation and the dramatic enlargement of the liver metastasis, we believed that a complete resection of the tumors would be difficult, and that chemotherapy treatment would be important to control tumor progression. We chose SOX plus bevacizumab therapy (on day 1 of each 3-week cycle, 7.5 mg/kg intravenous infusion of bevacizumab and 130 mg/m2 intravenous infusion of oxaliplatin were administered along with a dose of S-1 60 mg/m2 twice a day starting after dinner on day 1 until after breakfast on day 15, followed by a 7-day break). After 10 courses, the patient presented with oxaliplatin-induced peripheral neuropathy [], and another 10 courses were administered without oxaliplatin. An endoscopic examination did not detect gastric cancer (Fig. a) or transverse colon cancer (Fig. b). The liver metastases were dramatically reduced in size to 14 mm in segment 2/3 and 15 mm in segment 6 (Fig. a, b), and the FDG PET/CT findings showed no 18F-FDG uptake in any of the tumors (Fig. c, d). The serum blood tests showed the elevated tumor marker levels (carcinoembryonic antigen, 12.6 mg/dl; carbohydrate antigen 19-9, 43.1 mg/dl). After chemotherapy, the clinical diagnoses were ycStage IV (ycT3ycN0ycM1) transverse colon cancer and ycStage IIa (ycT3ycN0ycM0) gastric cancer. We planned a two-step procedure to completely resect the primary tumors and multiple liver metastases. We first performed resection of the colon and gastric tumors. Intraoperatively, there were broad adhesions between the omentum and the abdominal wall. The cytodiagnosis of the ascites showed no atypical cells. A small white nodule in segment 6 was detected. No signs of a “blue liver” were detected. We first laparoscopically performed resection of the transverse colon cancer in the right-side colon along with D3 lymphadenectomy. Then, we performed open distal gastrectomy for the gastric tumor with D2 lymphadenectomy (Roux-en-Y anastomosis). The operation time was 605 min, and the volume of blood loss was 840 ml. The patient was discharged home on postoperative day 18 and had an uneventful postoperative course. After 1 month, we performed liver resection for the multiple liver metastases. Intraoperatively, we detected tumors in segment 6 on the liver surface, and with sonography, we detected a tumor in segment 3, which was very close to the umbilical portion of the portal vein. We performed open lateral segment resection and partial resection at segment 3. The operation time was 402 min, and the volume of blood loss was 420 ml. The patient was discharged home on postoperative day 12 without any remarkable complications. The pathological findings showed ypT2 (MP) tumors with grade 2 therapeutic effects in the transverse colon and no residual gastric tumor and multiple liver nodules (pathological complete response). |
A 31-year-old gravida 4 para 0 African American woman at 22-weeks gestation presented with vaginal bleeding to an outside hospital. Her obstetric history was significant for 2 therapeutic abortions and 1 spontaneous abortion. During prenatal care, the fetus was noted to have a unilateral dysplastic kidney. The patient’s family history was significant for two family members with cervical cancer and two family members with endometrial cancer. She was placed on bedrest with inpatient admission. 48 h after admission she developed pelvic pain and uterine contractions. She expelled a mass vaginally measuring 11 × 9 × 5 cm with no fetal contents. The pathology from the outside facility showed a showed a highly cellular tumor composed of spindle-shaped cells and bizarre multinucleated giant cells with focal myxomatous change with mitotic count is greater than 50 per 10 high power field (Positive for CD10, SMA, ER, PR, EMA) with the differential diagnosis including endometrial stromal sarcoma and undifferentiated uterine sarcoma. The patient underwent examination under anesthesia, demonstrating a 3 × 3 cm defect of the posterior vaginal wall with active bleeding, which was sutured for hemostasis. The cervix was long, closed, and high with no evidence of bleeding and the fetus was intact with normal heart tones. She was discharged in stable condition.\nAt 27-weeks, the patient was referred to our institution for maternal-fetal medicine, and gynecology oncology consultation. On initial evaluation, she reported no prior abnormal gynecology history and no history of infertility issues. She denied dyspareunia and bulk symptoms prior to or during pregnancy, and her periods before pregnancy were normal. A pap smear at 27-weeks gestation was normal and negative for human papilloma virus. Her intake physical exam at our institution revealed a normal pelvic exam with no evidence of residual vaginal mass, and an ultrasound revealed no intrauterine myomas.\nGiven the differential diagnosis included an endometrial stromal sarcoma, an undifferentiated uterine sarcoma, and a primary vaginal sarcoma, the patient underwent magnetic resonance imaging (MRI) without contrast of the chest, abdomen, and pelvis at 27 weeks. MRI findings were notable for a peri-centimeter cyst in the cervix consistent with a Nabothian cyst, a multiloculated cystic structure in the fetal abdomen consistent with dysplastic kidney, and an anterior placenta. There was no evidence of metastatic disease.\nDue to the inability to ascertain whether the cancer had originated in the uterus, cervix or vagina, the patient desired definitive management for possible uterine sarcoma. In consultation with maternal fetal medicine and gynecology oncology specialists, the decision was made to proceed with a cesarean hysterectomy at 36 weeks. Betamethasone 12 mg intramuscularly was given for two doses 24 h apart with the first dose at 35 weeks and 6 days gestational age.\nAt 36 weeks and 1 day, she underwent planned exam under anesthesia of the vaginal canal with cesarean hysterectomy, bilateral salpingectomy, and peritoneal biopsies. She delivered a male infant weighing 2520 g with Apgars of 9 and 9. Operative findings were notable for normal appearing pelvic organs. There were no palpable or visibly enlarged pelvic/periaortic lymph nodes or evidence of metastatic disease throughout the peritoneal cavity. The neonate was admitted to the NICU post-delivery for prematurity. Neonatal US confirmed a right multi-cystic dysplastic kidney. He was transferred out of NICU on day 2 of life, and discharged home with his mother.\nThe final surgical pathology report was benign: no malignant or neoplastic cells were seen. The patient was seen at her 6-week postpartum visit and was doing well with a normal pelvic exam. At a follow-up surveillance visit 3 months postpartum, she was noted to have a 1 cm posterior vaginal wall lesion; biopsy results showed a high-grade sarcoma. The tumor was estrogen receptor and progesterone receptor positive. Computer tomography (CT) with contrast of the chest, abdomen, and pelvis showed no evidence of metastatic disease. The patient underwent an upper vaginectomy and proctoscopy. Operative findings were notable for 1.5 cm polypoid lesion in the posterior vagina wall 5 cm distal to the posterior vaginal apex. A 1 cm surgical margin was obtained circumferentially around this lesion where feasible, and surgical pathology showed high-grade sarcoma () of the polyp lesion with negative surgical margins. The tissue removed from this excision showed residual sarcoma, morphologically similar to the original pathology from the outside hospital pathology. All other vaginal biopsies were also negative for malignancy. Marker seeds were placed in the proximal and distal margins of the areas where the sarcoma was excised. She was treated adjuvant high dose rate vaginal brachytherapy using a multi-channel vaginal cylinder. She received a biologically equivalent 2 Gy dose (EQD2) of 45 Gy to the whole length of the vagina and 60 Gy to the post-operative bed (). A CT scan 36 months after surgery continue to show no evidence of disease. She remains disease free 58 months after completion of vaginal brachytherapy. |
A 65-year-old woman presented with a mass 6 cm in diameter located on her right frontoparietal region. The mass had appeared approximately three months earlier and had grown rapidly. The patient's history included resected colorectal adenocarcinoma followed by general surgery and was disease-free for five years. She was referred to us because the swelling and pain indicated a sebaceous cyst or a benign soft tissue tumor (). The mass was indurated, round, and adherent to the deep planes. Mild alopecia and skin expansion due to the mass were observed on her anterior scalp. The patient complained of tenderness and mild pain when the mass was palpated. Our clinical differential diagnoses included sebaceous cyst, lipoma, or localized soft tissue tumors. But the plain radiographs and the computerized tomography demonstrated a destructive mass which was eroding the bone over the right frontoparietal region ().\nA transverse incision and a meticulous blunt dissection that were performed under local anesthesia exposed a rubbery soft tissue mass extending to the outer table of the skull. The mass was dissected with care. However, we noticed calvarial bone destruction with an exposed duramater and lytic lesions 2 × 3 cm in size around the calvarial defect. The mass was completely resected. The mass was measured to be 5.5 × 5.0 × 2.0 cm (). The damaged bone sites were resected with a 1 cm security margin. A section of the duramater measuring 2 × 3 cm was removed and the defect was reconstructed using a tensor fascia graft by the brain surgeons. Hemostasis was achieved using electrocautery and bone wax. A bolster dressing was applied following the primary closure of the skin incision. The specimen was sent for histopathological analysis. The patient was monitored closely for one day and was discharged the next day.\nHistopathologic findings revealed many low differentiated adenoid, and apoptotic and necrotic spaces lined with atypical epithelial islands with vesicular nuclei. At high-power magnification, the specimens showed cytologic atypia with pleomorphic and hyperchromatic nuclei. Immunohistochemical analysis was positive for carcinoembryonic antigen (CEA), villin, and cytokeratins 7 and 20. The specimens were negative for vimentin, chromogranin, thyroglobulin, thyroid transcription factor 1 (TTF-1), and CDX2. The final diagnosis was metastatic low-differentiated adenocarcinoma compatible with the immunophenotype of colorectal adenocarcinoma (Figures , , and ). After the histopathological evaluation revealed metastatic adenocarcinoma, the patient was referred to the medical oncology department. |
An 85-year-old woman presented to hospital on 10 July with shortness of breath, fever and raised inflammatory markers and she was treated with levofloxacin for possible pneumonia. Her past medical history was notable for a tissue aortic valve replacement 11 years prior to this event, she had atrial fibrillation and was on apixaban and had hypertension and hypothyroidism.\nBlood cultures taken 2 days after admission were negative and a computed tomography pulmonary angiogram showed bibasal pleural effusions thought to be parapneumonic in nature. Deterioration in her clinical condition and new febrile episodes prompted repeat blood cultures to be taken on 28 July and these were positive for Campylobacter fetus identified by matrix-assisted laser desorption ionization time-of-flight analysis after 3 days of incubation. She did not have any loose stools during her admission at any point and had no known contact with animals. She was treated with 3 days of azithromycin. A transthoracic echocardiogram (TTE) was done and was reported as showing the prosthetic aortic valve was well seated with a preserved ejection fraction and mild diastolic dysfunction. She was discussed with the Cardiology team who felt that she did not warrant any further investigation as she did not display any features of endocarditis and her inflammatory markers had normalized, and she was discharged on 25 August.\nShe was then re-admitted on 6 September with multiple tonic-clonic seizures. In between episodes it was noticed she had a unilateral facial droop and had developed a fixed gaze. She was afebrile with a heart rate of 116 bpm and a blood pressure of 127/80 mmHg, and on examination it was noted that she had an audible ejection systolic murmur. Her C-reactive protein (CRP) level was 23 mg l−1 on admission and she had a white cell count of 13.0 g l−1 in the context of recurrent seizures.\nShe was initially started on ceftriaxone 2 g twice daily and acyclovir as there were concerns regarding central nervous system infection. However, as she slowly began to recover it became evident that she had a persistent right-sided facial droop, hemianopia and right-sided hemiparesis. An electroencephalogram done on admission was consistent with a vascular insult in the left hemisphere and not encephalitis and ceftriaxone and acyclovir were stopped. An initial computed tomography (CT) scan showed severe small vessel disease and a magnetic resonance imaging (MRI) scan done on 10 September showed bilateral emboli in both cerebral hemispheres. The working diagnosis at this point was that the patient had a cerebrovascular event secondary to her atrial fibrillation and she was transferred to the stroke team.\nOver the next 2 weeks her inflammatory markers started to climb and she started spiking temperatures to over 38 °C on 22 September and became less rousable. Aspiration pneumonia was suspected clinically and she was started on co-amoxiclav. Blood cultures were taken and were again positive 2 days later with C. fetus. As previously, the patient had no gastrointestinal symptoms. At this point clarithromycin was added pending MICs.\nGiven the repeatedly positive blood cultures, possibly septic emboli seen on MRI scan and prosthetic heart valve, endocarditis was strongly suspected. A repeat TTE on 26 September showed a significant change in the aortic valve appearance compared to the previous imaging, with thickening and restricted movement with possible thickening of the aortic root and an increase in the peak gradient to 67 mmHg although systolic function was maintained. It was felt that this warranted treatment for infective endocarditis due to C. fetus. A CT angiogram showed no evidence of aortitis but a possible wedge perfusion defect was noted in the spleen. She was not considered a surgical candidate and given her age, frailty and co-morbidities a transoesophageal echo was not performed.\nMICs were determined and were as follows: amoxicillin 0.50 µg ml−1, meropenem 0.016 µg ml−1, azithromycin 0.125 µg ml−1, gentamicin 0.38 µg ml−1, tetracycline 0.75 µg ml−1 and ceftriaxone 3 µg ml−1. Based on this information the antibiotics were switched to intravenous (IV) amoxicillin 2 g every 4 h to complete 6 weeks for prosthetic valve endocarditis along with 2 weeks of synergistic gentamicin at 1 mg kg−1 twice daily. Repeat blood cultures taken on antibiotic treatment were negative.\nThe patient gradually improved, her temperatures normalized and her CRP dropped to 7mg l−1 . She remained in hospital to complete her antibiotic treatment and multiple repeat blood cultures were negative. She remained afebrile with normal inflammatory markers while in hospital for 3 weeks after completing her treatment and was discharged to a rehabilitation facility on 28 November. There was no evidence of relapse of infection 2 months after discharge. |
A 48-year-old male was referred to our hospital due to abnormal brain MRI findings. He had undergone brain MRI for a mild headache without other neurological deficits such as weakness, paresthesia, or aphasia. The patient had no family history of intracranial lesions such as stroke, vascular malformations, or tumors. The initial brain MRI and computed tomography (CT) scan showed a single cystic mass located in the left basal ganglia with a fluid-fluid level and without peri-lesional edema (). Because the initial brain MRI was performed without enhancement at another hospital, an enhancing nodular lesion or peripheral rim enhancement could not be verified, however no nodular lesion or capsule-like appearance was detected using T2-, T1-weighted MRI. There was no evidence of calcification of the lesion in CT and gradient recalled echo T2-weighted imaging. To rule out vascular diseases, cerebral angiography was performed, which did not demonstrate tumor staining or a vascular malformation (). The first repeat brain MRI with Gadolinium enhancement performed 5 months after the initial brain MRI showed no evidence of peripheral or nodular enhancement. A spherical mass with a diameter of 16 mm had enlarged to 25 mm. Suspicious findings consistent with cavernous malformation were observed on axial T2-weighted MRI, which showed a reticulated "salt and pepper" pattern in the core and a peripheral halo of low signal intensity suggesting a hemosiderin rim (). These findings supported a diagnosis of cavernous malformation. The patient's symptoms were not remarkable; therefore, we decided to observe the natural course of the lesion using serial MRI. After 2 months, the patient attended our emergency department with a significant right hemiparesis of 3-hour duration. He was able to move the limbs on his right side through a full range of motion against resistance, but their power was weak. Brain MRI performed immediately showed an increase in the size of the lesion to 35 mm in diameter with heterogeneous internal signal intensity, multi-stage hemorrhage, and aggravated peri-lesional edema (). Based on the imaging findings, the lesion was diagnosed as a CM with recurrent bleeding. A left pterional craniotomy was performed, followed by a trans-sylvian approach, trans-insular cortisectomy for removal of the mass. The margin of the mass was clearly defined and the bulk of the mass had the gross appearance of a hematoma with a yellowish cystic fluid and no encapsulation. A gliotic plane surrounding the mass was observed. There was no active bleeding or evidence of abnormal vessels in the operative field. Intraoperative neuromonitoring included somatosensory evoked potentials (SSEP) and motor evoked potentials (MEP). Median and tibial nerves SSEPs on both sides showed normal values in latency at baseline and no significant interval changes during surgery. MEP decreased by nearly 50% in amplitude at the right extensor digiti minimi during resection of the tumor. Final MEP waveforms after skin closure showed improvement and decreased by < 50% in amplitude and increased < 10% in latency. A neurologic examination performed immediately after surgery showed no significant motor changes compared with the pre-operative examination. After the operation, the patient recovered rapidly from hemiparesis within 2 weeks and was discharged without neurological deficits. The histopathological features were consistent with CM (). After an additional 5 months, follow-up MRI showed no residual lesion () and the patient's neurologic status was normal. |
A 34-year-old woman presented to a major academic institute with history of pain in the right eye, she also developed rapidly progressive visual loss in the same eye over a course of 1 week. Her past history was not significant for any inflammatory pathology. Examination revealed visual acuity of 6/36 in the right eye and 6/6 in the left eye. Visual field charting showed right-sided cecocentral scotoma and the left eye was normal. Fundoscopic examination revealed right-sided mild superior and inferior temporal pallor, and the left side was normal. Visual evoked potential (VEP) showed delayed wave pattern in the right eye [] while it was normal for the left eye. Her workup for the inflammatory pathology was negative. Computed tomography scan of the orbit with contrast enhancement showed suspected optic nerve space occupying lesions, magnetic resonance imaging (MRI) of the brain with contrast enhancement showed right side optic nerve was markedly thickened compared to the left side, there was a hyper intense signal on T2-weighted images around the nerve, with intense enhancement around the optic nerve [] especially in the posterior half and extending as far as the orbital apex. On this basis, she was wrongly diagnosed to be having optic nerve glioma and was planned for surgery at another major academic center. She came to us to seek second opinion and on careful history taking we found that she had acute unilateral decreased visual acuity, which had worsened within 5-7 days with ocular pain aggravated by the eye movement which is quite typical for ON. On examination, we found that she had decreased visual acuity, grossly impaired color vision with central scotoma and afferent pupillary defect in the same eye. The careful scrutiny of the clinical and radiographic findings led to the diagnosis of ON. She was started on steroids and had complete resolution of her symptoms. Her repeat MRI and VEP showed marked improvement when compared with the previous ones [Figure and ]. |
A 16-year-old obese male, with no medical history, presented in emergency room with a closed trauma of the left knee secondary to a traffic accident. He was hit by a car while biking. The mechanism of the injury was a hyperextension and a valgus stress of the left knee. General examination revealed no other significant injury. On physical examination, the patient was totally unable to step on or move his left lower limb. There was a significant swelling around his left knee with no skin defect. The pulses of the A. poplitea, the Arteria tibialis posterior, and the Arteria dorsalis pedis were well palpable. The capillary filling of the toes was present. No motor or sensitive defect was found. Plain radiographs of the left knee showed a Salter and Harris Type 1 physeal fracture of the distal femur with an important displacement of the metaphyseal fragment (). At that time, in front of the spectacular femoral injury, the diagnosis of a Type 3 slightly displaced physeal fracture of the proximal tibia was missed. The patient was operated in the same day. Under general anesthesia, the patient was placed in supine position with the left lower limb pulled in its axis using a fracture table. After a gentle continuous traction, a downward pressure was applied on the distal epiphysis of the femur with the tight been locked. The reduction was found good when checked using the fluoroscopy. Fixation was done using two crossing 24 mm K-wires. A long leg cast was made. Post-operative radiographs showed a good reduction of the distal femur (). At this time, the tibial injury was diagnosed. The slight displacement of the epiphyseal fragment of distal tibia was accepted. Close physical observation in the first post-operative 24 h found no secondary vascular thrombosis. The cast was kept for 6 weeks. After the same period, healing of the fracture was obtained (), hardware was removed, and full weight bearing was allowed. 2 years later, the patient was pain free, with a complete range of motion of the left knee. No angular deformity or shortening of the left lower limb were noted (). |
A 67-year-old female with past medical history of congenital deafness presented to the emergency room with complaints of right-sided facial droop and right upper extremity weakness, tingling, and numbness. These symptoms were sudden in onset and lasted for a few minutes. Symptoms had completely resolved at the time of presentation. She did not have a history of any atherosclerotic risk factor including hypertension, diabetes, or hypercholesterolemia. The initial set of vital signs were normal; routine laboratory tests including complete blood count and basic metabolic panel were unremarkable. A computed tomography (CT) of the head without contrast as well as a magnetic resonance imaging (MRI) of the brain with and without contrast did not show any acute intracranial hemorrhage or infarction.\nThe patient was diagnosed with TIA, and further investigations were planned to determine the etiology. A magnetic resonance angiogram (MRA) of the head and neck with and without contrast did not show any arterial flow limiting stenosis or occlusion. A transthoracic echocardiogram (TTE) with bubble study using agitated normal saline contrast was performed and was found to be normal. Patient's heart rhythm was monitored with continuous cardiac monitoring, and no arrhythmias were noted during her stay at the hospital. At this point, the patient was identified as having cryptogenic TIA, having failed to determine the precise etiology from routine workup. Patient was started on aspirin therapy and discharged from the hospital on day 3 with further outpatient workup planned. Outpatient workup for hypercoagulability showed a high factor VIII activity of 153%, which potentially put her at increased risk of venous thromboembolism. However, this test was performed just one week after the thrombotic event and was hence difficult to interpret. Subsequently, a transesophageal echocardiogram (TEE) was performed that revealed a patent foramen ovale with right-to-left shunt. This raised the concern for paradoxical embolism as the cause of patient's TIA. Lower extremity duplex venous ultrasound showed no evidence of deep vein thrombosis. However, magnetic resonance venogram (MRV) of pelvis showed compression of the left common iliac vein just after its origin, which was suggestive of May-Thurner syndrome (). There was no evidence of venous thrombosis on the MRV. May-Thurner syndrome was recognized as the probable source of paradoxical embolism causing TIA in the patient. The patient was eventually referred for percutaneous PFO repair, which was performed without any complications. The patient had been regularly followed yearly at the cardiology clinic for 5 years now. She remains in good health with no further episodes of TIA. |
A 77-year-old Korean man who was a retired office worker was brought to our memory disorder clinic by his wife, who reported that the patient's main problem over the past 2 years was difficulty understanding what other persons said. At first, the caregivers thought the man had a hearing problem. Six months after symptom onset, after a hearing aid prescribed by a local otolaryngology clinic did not correct the problem, the caregivers brought the patient to a local neurology clinic where he underwent brain magnetic resonance imaging (MRI). MRI showed bilateral periventricular white matter hyperintensities that were slightly more severe on the right side than the left and multiple stenoses in the anterior, middle and posterior cerebral arteries.\nCaregivers recalled that at 3–4 months after onset, the patient started to have problems naming familiar, common, daily objects in addition to difficulty understanding speech. One year after onset, when asked questions, most of the patient's answers were unrelated to the question. However, the man was still able to read newspapers and use the Internet. Over the next year, his comprehension ability declined rapidly until he barely comprehended speech and never provided relevant answers to questions. At the same time, his memory was slightly impaired, for example, he could not recall a door-lock password. The patient started experiencing difficulty using public transit and taking medicines on his own. However, no obvious changes were noted in his behavior. When his family brought him to our clinic 2 years after symptom onset, he was moving slowly with a mildly shortened step.\nThe patient's past medical history was remarkable for hypertension, diabetes mellitus, hypothyroidism, and benign prostatic hyperplasia, but not for any history of developmental delay or disorder. The patient had received 16 years of education. Due to his severe language impairment, he could not perform actual motor skill tests to identify the dominant hand such as the Purdue Pegboard Test. However, the Edinburgh Handedness Questionnaire [] revealed that he was strongly right-handed, on which he scored right-handed for all 10 questions. Family history showed that his mother had a stroke, while there was no family history of left-handedness.\nA neurological examination showed that the patient was alert and able to utter sentences of 4–5 words and sounded fluent without being dysarthric or halting. However, his spontaneous speech often contained phonemic paraphasia or some jargon, making it difficult to understand what he was trying to say. The patient also showed severe auditory comprehension difficulty: for instance, when asked about his chief complaint, he asked ‘what?’ several times. The patient scored 0 of 30 on the Korean Mini-Mental State Examination because of language problems and cognitive impairment. His gait was slow with mild short steps. Cranial nerve functions, muscle volume, tone, power, and sensory functions were normal. Deep tendon reflexes were normal with no pathological reflexes.\nRoutine blood labs including serum vitamin B12, syphilitic serological marker, creatinine, and thyroid-stimulating hormone showed no abnormalities. Apolipoprotein E genotype was ε4/ε4. MRI showed bilateral periventricular white matter hyperintensities and diffuse brain atrophy on fluid-attenuated inversion recovery (FLAIR) sequence (fig. ). The patient had slightly more severe atrophy in the right temporal area than the left. In order to quantify the patient's cortical atrophy, we registered the patient's MR images into the MNI152 standard space using an affine linear transformation. Then, images were classified to white matter and gray matter using an artificial neural net classifier. The surfaces of the inner and outer cortices were restructured using 40,962 vertex points for each hemisphere automatically. Then, cortical thickness was computed applying the Euclidean distance between the linked vertex points of inner and outer surfaces. Subsequently, we measured W-scores based on the data collected from 55 normal elderlies (mean age: 68.8 ± 3.2, female sex ratio: 70.9%). W-score is the Z-score using the following formula with adjusting specific covariates: [(patient's raw value) – (value expected in the controls considering covariates)]/SD of the residuals in controls. Last, extracted values were reversed in order that positive numbers represent atrophic changes. The following special covariates were used: age, sex, education, and ICV. This patient showed decreased cortical thickness in all cortical area, and the right anterior temporal and parietal cortices were observed to be the thinnest regions (fig. ). 18F-fludeoxyglucose positron emission tomography (FDG-PET) showed moderate hypometabolism in the bilateral fronto-parieto-temporal cortex (fig. ). Quantitative analysis of FDG-PET was done after PET images were coregistered to T1-weighted MRI. The quantitative regional values were measured by an automated volume of interest (VOI) analysis tool using the automated anatomical labeling atlas. SPM version 8 through Matlab 2014 (Mathworks, Natick, Mass., USA) was used for this analysis. To measure regional glucose metabolism, we used the standardized uptake value ratios (SUVRs) which is the cortical to reference region uptake ratio. Pons mean activity was set as a reference. We selected 28 cortical VOIs from both hemispheres. The selected cortical VOIs were made up of the following cortices: bilateral frontal (superior and middle frontal gyri, medial part of superior frontal gyrus, opercular part of inferior frontal gyrus, triangular part of inferior frontal gyrus, supplementary motor area, orbital part of superior, middle, and inferior orbital frontal gyri, rectus and olfactory cortex), posterior cingulate gyri, parietal (superior and inferior parietal, supramarginal and angular gyri, and precuneus), lateral temporal (superior, middle and inferior temporal gyri, and Heschl's gyri), and occipital (superior, middle, and inferior occipital gyri, cuneus, calcarine fissure, and lingual and fusiform gyri). The patient's FDG-PET SUVRs of the right hemisphere were lower than those of the left (left vs. right SUVR: frontal cortex: 1.46 vs. 1.35; temporal cortex: 1.32 vs. 1.09; parietal cortex: 1.42 vs. 1.21; occipital cortex: 1.58 vs. 1.48) (fig. ).\nOne month after the first visit to our clinic, the Korean version of the Western Aphasia Battery (K-WAB) was performed to identify the patient's aphasia type. Throughout the test, he had difficulty concentrating on the test and kept on speaking what he wanted to say. On a spontaneous speech test, the patient spoke sentences using 4–5 words. He sometimes understood a few words of questions and repeated those words, but his speech contents were not related to the conversation. On seven questions to elicit spontaneous speech, he gave only a single right answer to the question about his name. When asked to describe a picture, he formed sentences without agrammatism, but frequently used substitutes and jargons, resulting in a 2/10 score for informed content and 7.5/10 for fluency rating. The auditory comprehension part of the K-WAB consists of three subtests: yes-no questions (maximum score 60), auditory word recognition (maximum score 60), and sequential commands (maximum score 80). The patient's impairment on auditory comprehension was so severe that he could not understand even single words, let alone complex questions. Thus, he only scored 3/60 in the yes-no subtest and scored none in other subtests. The patient also could not perform the repetition test and kept on speaking what he wanted to say (repetition: 0/10). When asked to name objects, the patient named 4 of 10 (object naming: 14/60). He was unable to perform tests about word fluency, sentence completion, and responsive speech. He also performed poorly at the reading (0/10) and writing (1.9/10) subtests. The overall results of the K-WAB were consistent with a severe degree of Wernicke's aphasia. The language disturbance precluded further neuropsychological tests.\nAt 3 years after symptom onset, the patient could not understand what others said and his utterance decreased drastically. At 3 years after onset, the patient underwent a Pittsburgh compound B (PiB)-PET which revealed amyloid accumulation mostly in the left fronto-parieto-temporal cortex (fig. ). PiB-PET was also analyzed using the same methods as FDG-PET except that for PiB-PET, the cerebellar gray matter was used as the reference area. Higher SUVRs were extracted in the right hemisphere than the left (left vs. right SUVR: frontal cortex: 2.01 vs. 1.75; temporal cortex: 1.82 vs. 1.57; parietal cortex: 2.07 vs. 1.76; occipital cortex: 1.68 vs. 1.50) (fig. ). 18F-T807-PET performed at 3.5 years after symptom onset showed that tau deposition was more predominant in the right fronto-parieto-temporal cortex than the left (fig. ). When SUVRs were calculated using the same process as PiB-PET, higher SUVRs were observed in the right cortices than the left (left vs. right SUVR: frontal cortex: 1.76 vs. 2.33; temporal cortex: 2.18 vs. 2.67; parietal cortex: 2.01 vs. 2.51; occipital cortex: 1.84 vs. 1.97) (fig. ). |
We report the case of a 90-year old female who was admitted to our traumatology department to fix an extracapsular hip fracture of the left femur which occurred during a low-energy trauma. The history of the patient revealed that she suffered from osteoporosis. In her eighties, she had been conservatively treated with a cast for a right wrist fracture. Previously the patient had been treated for four vertebral compression fractures with thoraco-lumbar orthosis. Spine and femur DEXA scan measurements at the time of the vertebral fractures showed T-Score of -4.9 and -3.7, respectively, confirming a severe osteoporosis.\nAfter routine clinical tests, the surgeon chose DHS to treat the pertrochanteric fracture. The operation was performed under general anesthesia, with the patient positioned on a fracture table. The fracture was manually reduced under image intensifier control. Thereafter the surgeon began placing two k-wires into the femoral head through the femoral neck with a lateral skin incision. A complication occurred in this step, as the surgeon found out that the first k-wire had been broken. A second K-wire was positioned. Next the cephalic screw was positioned in the middle of the femoral neck using K-wire to guide the insertion of the dynamic screw. Due to the severe osteoporosis, the surgeon decided to ream only the lateral cortex of the femur to boost the grip strength. The ensuing step was to slide the lateral plate to the cephalic screw until the internal side of the plate was seated on the lateral cortex of the femur. Owing to difficulties in placing the plate, the surgeon decided to use the mallet. During X-ray control of this surgical step, the cephalic screw appeared to have unexpectedly migrated into the pelvis (). Several attempts with different surgical forceps were made to remove it through the screw hole, but the screw had penetrated deeply into the pelvis making it impossible to retrieve it. The general surgeon was called to perform an abdominal approach to remove the screw, which was finally retrieved in a few minutes without any further complications. Meanwhile, the procedure of osteosynthesis was completed using a condylar plate (). The patient started postoperative physical reconditioning three days after, with only one day’s delay compared to standard procedure. |
A 68-year-old female underwent a partial left-sided chest wall resection, with partial removal of the 6th and 7th ribs and of the scapula angle for elastofibroma (). The chest wall defect was reconstructed by using a Mersilene mesh, secured by interrupted pericostal stitches, and covered by a sufficient volume of viable muscles. The postoperative course was uneventful; the radiographic aspect at discharge was normal (). The first symptoms in the form of pains in the region of the incision appeared five months after the operation, and computer tomography (CT) of the thorax showed a lung hernia in the region of the mesh covering the chest wall defect (Figures and ). The patient refused the proposed surgical correction, being only slightly limited in usual daily activities. During the next several months, the symptoms persisted with variable intensity under analgesic therapy, till the moment when pains significantly limited patient's daily activities, 22 months after the operation. The repeated chest CT showed a slight increase in hernia size, with no signs of tumour recurrence (), so that reoperation was planned.\nAfter the excision of the previous skin scar and the incision of the muscular layer, the mesh region was exposed, showing a lung protrusion (4 × 3 cm) along the anterolateral edge of the mesh (). The local situation is schematically presented on . The mesh suture line in the hernia region was completely disrupted, with a small piece of the herniated lung being completely detached from the mesh, the remaining lung surface under the mesh area being fully adherent to the mesh. By careful dissection, the mesh was separated from a firmly adherent lung and removed (). After adhaesiolysis and complete lung liberation, a wedge resection of the afunctional lung tissue of the superior segment of the lingula was done, just in the region of contact with the mesh. After the chest tube insertion, the chest wall defect was reconstructed by suturing a Mersilene mesh in two layers—single pericostal sutures for initial fixation and running suture for additional reinforcement (). A final chest wall stabilization was done by the fixation of two Synthes plates (DePuy Synthes J&J) over the 5th and 6th ribs (). The postoperative course was uneventful. The chest X-ray on discharge, on postoperative day 5, is presented in .\nAt the last contact with the patient, one year after the operation, the general condition was good, without the need for analgesics. |
The patient was a 48-year-old woman who worked as a healthcare professional with a medical history notable for four renal transplantations for idiopathic renal failure beginning at age eight. She was on chronic immunosuppression with tacrolimus and mycophenolic acid. She presented initially with cutaneous SCC two years previously with a nodule of the dorsal right hand involving the 3rd and 4th webspace. Biopsy confirmed squamous cell carcinoma and she underwent Mohs resection of the mass with subsequent reconstruction with chest wall flap. Resection included at least partial excision of the ring finger radial digital nerve due to perineural invasion. A year later, she noted a recurrent mass of the same webspace and underwent re-resection with negative margins. Over the next several months, the mass slowly re-grew in the same webspace, prompting a biopsy with pathology consistent with recurrent SCC. Magnetic resonance imaging (MRI) at that time showed a dorsal mass to the interspace between the heads of the third and fourth metacarpals measuring approximately 7 mm transverse × 5 mm volar-dorsal × 16 mm proximal-distal. The tumor significantly involved the webspace with some penetration of the lesion into the volar neurovascular bundle. Clinically, there was suspicion of dorsal, dermal invasion; however, MRI did not show bone invasion. She was seen in consultation at an outside institution where single (4th digit) to double ray resection (3rd and 4th digits) was recommended.\nThe patient presented to our institution for a second opinion. The recommendation of upfront surgical management was reinforced. As the patient refused ray amputation due to functional loss and cosmetic deformity, the option of initial external beam radiation therapy followed by surgical tumor debulking was discussed. The potential serious complications of surgery and radiation therapy were discussed in detail []. Due to the increased risk of wound healing complications, significant impairment of lymphatic drainage, fibrosis, swelling, and continued pain which could have limited the function of her hand, she declined this option as well. The option of EBRT alone using electrons was proposed. An initial plan using electrons was generated but the dose inhomogeneity was deemed unacceptable given the complexity of the target. Subsequently, she was referred to the brachytherapy group for consideration of interstitial brachytherapy (). After much discussion, a plan of surface and interstitial HDR brachytherapy was generated with the goal of balancing tumor control with preserving organ function. |
A 48 year-old female patient was transferred from another institution due to intractable bile leak after LC. It was reported that her primary diagnosis was calculous cholecystitis and all LC procedures were completed uneventfully according to standard techniques (). The patient discharged on the second day after LC, but 4 days later she readmitted due to abdominal pain and fever. Abdomen computed tomography (CT) revealed abnormal fluid collection in the subhepatic and subphrenic areas (). Endoscopic retrograde cholangiography (ERC) was performed under suspicion of bile leak, but no biliary injury was recognized at the time of the ERC procedure (). A retrospective review of the ERC images indicated that one of the right sectoral ducts was not visualized, but this finding was missed at the time of the ERC. Even after supportive care for 1 week, her symptoms had progressively become aggravated. Follow-up CT scans showed definite accumulation of a huge amount of perihepatic fluid (), which led to multiple pigtail insertion. The drained fluid was pure bile. A repeat ERC showed no evidence of a leak (), but pure bile of about 150 ml/day continuously drained from the abdominal pigtail catheters. Finally, she was transferred to our institution, 40 days after LC.\nWe assessed the extent of LC-associated bile duct injury. Magnetic resonance cholangiography (MRC) was carried out to delineate the biliary anatomy of the missing duct. The RAS duct had been transected close to the liver parenchyma, but there was no recognizable duct stump at the common bile duct except the cystic duct clipping (). This biliary anatomy implicates that the RAS duct might be aberrantly inserted into the cystic duct and both cystic duct and RAS duct were transected altogether (). Hepatobiliary scintigraphy showed major bile leak from the gallbladder fossa (). There was no intrahepatic duct dilatation in the right liver, implicating that bile drained freely from the transected RAS duct ().\nThe morphological features of this isolated injury to the RAS duct suggested that primary reconstruction using Roux-en-Y hepaticojejunostomy was not feasible or was technically very difficult and RAS resection appeared to be very complicated. Based on our previous experience with hepatic atrophy induction therapy for LC- and hepatectomy-associated segmental bile duct injuries, we decided to perform portal vein embolization (PVE) of the RAS portal branch to induce atrophy of the RAS parenchyma (). Thereafter, the amount of bile drained from the pigtail diminished dramatically, and the fluid color became much paler. CT scans taken after 1 and 4 weeks showed that the RAS parenchyma had atrophied markedly ().\nTwo weeks after PVE, one subphrenic pigtail catheter was removed. Ten weeks after PVE, the amount of pigtail drainage became scanty. Thus, the single subhepatic pigtail catheter was left clamped for additional 2 weeks. Thereafter, we removed the last clamp after ensuring the occurrence of heavy adhesions around the bile leak site. The pigtail was kept in place for a total of 3 months after PVE, as initially planned. At 6 months after PVE, the RAS parenchyma was definitely atrophied () and bile production disappeared completely on follow-up hepatobiliary scans (). She was free from any other complications during the first 12 months and to date. She will be followed up for 5 years including surveillance for hepatobiliary complications. |
A 64-year old woman presented with neurogenic claudication and a maximum walking distance of 50 m. She described a radiating pain from the gluteal region to the lateral legs on both sides with a predominance for the left side, with a symptom onset approximately 1 year ago. Physical examination revealed intact motor function and decreased deep tendon reflexes on the left side. Medical history consisted of a discectomy L5/S1 on the right side 13 years ago. MRI revealed a lumbar spinal stenosis at the level of L4/5 (Fig. ). Patient underwent a standard microsurgical decompression L4/5 on the left side, including an over the top undercutting to the right side. During surgery, no dural tear was noted. On the first postoperative day she was mobilised and described significant improvement of the preoperative pain.\nTwo days later, during a turning movement in bed, she experienced a sudden sciatica radiating in her left leg. The intense pain was not responding to any analgesics and was only tolerated in a standing position leaning on the right leg. After frustrating attempts to stay in the horizontal position despite intravenous opioid administration, she was put under general anaesthesia for an MRI scan. The imaging revealed no hematoma or significant intraspinal compression. However, an unclear facet joint effusion L4/5 on the left side was apparent, which was not present preoperatively (Fig. ). The decision for exploratory revision surgery was therefore manly based on her clinical presentation as at this point the meaning of this effusion was unclear. In early stages of revision surgery cerebrospinal fluid (CSF) leakage was noted and after careful dissection, a nerve root herniation through a small lateral dural tear with entrapment in the facet joint gap was discovered (Fig. ). The herniated root was repositioned and the dural defect was repaired using stitches in a watertight fashion and a sealant matrix. Postoperatively the patient’s symptoms disappeared, she was mobilised after 2 days using a lumbar brace and the further clinical course was uneventful. As she did not develop any further clinical signs of potential segmental instability, fusion surgery was not deemed necessary. |
A 42-year-old male presented to the Department of Head and Neck Surgery of a tertiary oncological centre because of right mandibular swelling and trismus. The patient had already been admitted to a secondary care hospital 3 months earlier with complaints of right mandibular discomfort and slight tumefaction. A biopsy was then performed and the diagnosis of ameloblastic carcinoma was made histologically. The patient was otherwise healthy with no significant past medical history, including alcohol, smoking or tobacco abuse.\nA complete head and neck examination revealed a painless, firm and fixed right mandibular mass with no cutaneous inflammatory signs. No ulcers or mucosal lesions were found in the oral cavity. Laboratory evaluation, chest radiograph and respiratory function tests were unremarkable. The patient underwent bronchofibroscopy, which revealed only mild laryngeal hyperaemia.\nBoth neck CT () and MRI () were performed, showing a large, solid tumour arising from the ramus and posterior body of the right mandible. The lesion extended to the surrounding soft tissues, with invasion of the masseter and medial pterygoid muscles and caused bulging of the buccal mucosa. The soft tissue component was hypointense on T1 weighted and hyperintense on T2 weighted MR images and showed avid enhancement after gadolinium administration on MR examination. CT scan disclosed striking sclerosis and irregularity of the mandibular ramus with some gas bubbles inside the medullary cavity and an expansive lytic component in the posterior body and angle with some bone-forming matrix inside. Prominent periosteal reaction was also identified, particularly in the outer cortical surface of the mandibular ramus with the typical pattern of a ruptured Codman triangle. No associated cystic lesion was found in the mandible. No enlarged lymph nodes were detected and the evaluation of the remaining cervical spaces was unremarkable. A thoracic CT scan was also performed, with no parenchymal lesions.\nThe patient underwent a right hemimandibulectomy and ipsilateral cervical lymph node dissection. Surgical resection also included the right submandibular gland and a segment of buccal mucosa that was swollen by the mandibular mass. Reconstruction was performed with free fibula graft. The surgical specimen included a large, white and solid tumour with 11 × 7.5 × 6.5 cm, corresponding to an invasive, moderately differentiated (G2) SCC (). Focal positive margins were found at the medial surface of the specimen. The resected buccal mucosa, submandibular gland and lymph nodes had no neoplastic tissue. Taking into account the imaging staging examinations and the post-surgical histological report, the final TNM stage was stated as pT3 N0 Mx. After surgery, the patient underwent adjuvant chemotherapy (cisplatin-based regimen) and radiotherapy. |
We report the case of a 75-year-old North African woman with no notable medical history, admitted for pelvic pain lasting for the last 4 months and unrelieved by analgesics; the transit was undisturbed. The symptoms were aggravated by melena and asthenia that motivated the medical consultation. A clinical examination at admission found a patient with a stable hemodynamic status, an abdominal examination revealed a palpable and mobile pelvic mass. Laboratory tests showed a hypochromic and microcytic anemia with low hemoglobin and hematocrit levels rating at 7.3 g/dL and 21%. An abdominal computed tomography (CT) scan showed a large pelvic mass measuring 11 × 9 cm involving the distal small bowel loops, the bladder dome, and the uterine body without peritoneal effusion (Fig. ). After primary care, our patient underwent an exploratory laparotomy. The intraoperative finding was a brownish pseudoaneurysmal mass of the small bowel located 80 cm from the Treitz’s angle; this mass was invading the bladder dome and the left ovary and closely adhering to the uterus (Fig. ). Limited small bowel resection with 10 cm margins on both sides of the tumor extended to the left annexes and to a portion of the bladder with end-to-end anastomosis was achieved. The postoperative management was uneventful and our patient was discharged on day 6. The surgical specimen was 18 cm in length and included a black solid tumor with exophytic growth infiltrating the small bowel wall until the mucosa (Fig. ). A histological examination revealed a malignant proliferation of large cells with prominent round nuclei and a cytoplasm with eosinophilic spots or the seat of melanin pigments. Tumor necrosis was estimated at 30%, the rest of the ileum was the seat of chronic ileitis (Fig. ). The immunohistochemical profile showed an intense and diffuse cytoplasmic positivity for HMB-45 antigens and for PS-100, suggesting a malignant melanoma (Figs. and ). An etiological investigation in search of a primary tumor of the small bowel melanoma was negative, an anoscopy, examinations of eyes and skin with multiple cutaneous biopsies were performed without finding any melanoma lesion. |
A 78-year-old woman suffering from generalized right leg pain applied to the outpatient department. From her medical history; she underwent total hip arthroplasty six years ago after being diagnosed as having primary osteoarthritis of the right hip. After surgery, she had not reported any problems about her right hip arthroplasty. Suddenly; she had restriction on full weight-bearing in her contralateral left side for seven months. She was prescribed painkillers by her general practitioner and started using a walking stick. Her relatives said that there was no specific trauma that could explain sudden onset of restriction on full weight-bearing in her contralateral left side. They criticized the general practitioner for not investigating her left side pain meticulously. We had no X-ray records of the patient during her application to general practitioner seven months ago.\nWhile she was walking for seven months after restriction of full weight-bearing of left hip, she had a fall after stumbling on the carpet, and was admitted to the outpatient department of orthopaedics for evaluation of severe right hip pain. The antero-posterior radiograph of the pelvis revealed Vancouver type-C periprosthetic fracture of the right femur and a neglected femoral neck fracture in her left hip (). According to the information gathered from her and her relatives, it was a simple fall that could not explain such a catastrophic periprosthetic fracture in the absence of any predisposing factor such as contralateral full weight-bearing for months.\nThe patient was 78 kg, 165 cm tall with a calculated body mass of 28.65 kg/cm2. She had undergone total hip arthroplasty with expansion cementless acetabular cup and cemented femoral prosthesis previously. After surgery, she had been followed up regularly for three years once a year and there was no complication such as significant leg length discrepancy or rotational abnormalities. Later she was lost to follow-up as she had no complication during those three years. She applied to outpatient department with periprosthetic fracture of the right femur and a neglected femoral neck fracture in her left hip. Pre-operative routine procedure was done by planning revision arthroplasty of the right hip. The only comorbidity was well-controlled blood pressure. C-reactive protein level and erythrocyte sedimentation rate were in normal range. She underwent revision arthroplasty of the right hip by using the previous anterolateral incision [modified Watson-Jones]. Both acetabular and femoral components were removed. Cementless acetebular cup with constrained polyethylene liner and 40 mm proximal body, 12/160 mm distal stem of fully porous coated long femoral prosthesis was implanted during revision surgery (). Five dall-miles cables were used to stabilize the femoral component. There were no post-operative complications and the patient was ambulated with partial weight-bearing on the left side and as much as she tolerated on the right side with the aid of a walker on the third day postoperatively. Hip musculature strengthening physical therapy was applied during her follow-up. Although total hip arthroplasty was offered for neglected contralateral femoral neck fracture, patient and her relatives were not keen for a second operation due to advanced age of the patient and also because she had relatively less complaints with her left hip. Weight-bearing had been increased incrementally during the follow-up of six and twelve weeks. The patient was ambulated with full weight-bearing six months after surgery. After seven months of neglected contralateral femoral neck fracture, patient was encouraged for weight-bearing on that side as much as her pain allowed her to bear. During regular follow-ups, she continued to have restricted weight-bearing in the contralateral side and was consistently reminded of the necessity for a total hip arthroplasty for that side at eachfollow-up visit. |
A 15-year-old female reported to our college with complaint of missing front tooth. She was a dwarf with a height of 4.5 feet and a weight of 31 kg (). History revealed that she was diagnosed as a case of hypopituitarism at 3 years of age. Her mother had a similar condition () and all her siblings were reported to be normal. Her basal growth hormone levels were 0.30 ng/mL at 3 years of age and 2.10 ng/mL at 11 years of age as per the records produced by her. Growth hormone replacement therapy was suggested for the patient by her endocrinologist which she had not taken. Her profile was mildly convex with competent lips (Figures , , and ). She had class I molars with proclined incisors and generalised spacing. She gave a history of trauma a few months back leading to the loss of her upper left central incisor (Figures , , and ). She had generalised microdontia with short conical roots as revealed by the OPG (). Her mother's OPG () too revealed microdontia which is consistent with the dental findings of congenital hypopituitarism []. Her lateral cephalogram revealed a class I skeletal base ( and ). A striking feature in the lateral cephalogram was the decreased sella size (). The size of sella turcica assessed from radiographs typically ranges from 4 to 12 mm for the vertical and from 5 to 16 mm for the anteroposterior dimensions [–]. The lower limits of normal for depth and length of sella on radiographs are 4 mm and 5 mm []. In our case on superimposing the sella tracing over a graph sheet with millimeter readings [], the vertical dimension was found to be 3.5 mm and anteroposterior dimension was 4 mm. The dimensions in our case are lower than the normal lower limit.\nSince the patient's main complaint was missing incisor, she was not interested in getting orthodontic treatment despite having proclined incisors. Hence, no orthodontic treatment was carried out for this patient. |
A 47-year-old female, with a history of left breast cancer and total mastectomy followed by neo-adjuvant radiation in 2009, presented to the Plastic and Reconstructive Surgery clinic in December 2014 to discuss options for breast reconstruction. Other significant past medical and surgical histories at the time of her initial presentation included a left posterolateral thoracotomy for complicated pneumococcal pneumonia, three cesarean sections, and 1 ppd tobacco use. Her disease status was closely monitored by her oncologic team and was deemed disease-free as evident in her then most up-to-date negative screening mammogram in 2013.\nUpon successful cessation of her tobacco use for a period of six months after the initial consultation, the patient was taken to the operating room in June 2015. With goals to recreate a full C-cup sized breast in a patient with a very thin body habitus (body mass index = 24), our team utilized a supercharged muscle-sparing fTRAM flap in order to safely utilize all four zones of the abdominal flap. Utilizing a two-team approach, the recipient site of the left chest was entered from her previous mastectomy incision, along with resection of the surrounding radiated fibrotic skin. A laterally based pectoralis flap was utilized to dissect up to the second rib, which was then partially resected to expose the left internal mammary artery and two veins. Simultaneously, the transversely oriented abdominal flap was elevated by the second surgeon, being particularly careful to preserve both the superior and inferior epigastric vessels on both hemi-abdominal flaps. Dissection proceeded in the prefascial plane above the anterior rectus sheath in a lateral to medial approach. After the favorable right-sided lateral row perforators were identified, the fascia was incised and the rectus muscle exposed, allowing us to trace the vessels down to the origin of the deep inferior epigastric artery and vein in the right lower quadrant. Care was taken to preserve both the superficial and deep inferior epigastric vessels on both sides of the flap during all steps of the dissection. Once the medial portion of the right-sided muscle-sparing TRAM flap was elevated in a MS-2 design, it was found to be quite thin, and so this portion of flap was folded on itself during the harvest. Once the flap was fully islandized, adequate perfusion and bleeding was seen along the entire flap, including zone 4 after the de-epithelization process (Figure ).\nOnce the flap was dissected, de-epithelized, and transposed to the left chest, it was folded in half and the following vessels of the tissue were microsurgically anastamosed to those of the left internal mammary system: the superficial inferior epigastric vein in zone 4 of the contralateral fTRAM flap to the medial internal mammary vein, the ipsilateral deep inferior epigastric artery to the internal mammary artery, and the ipsilateral deep inferior epigastric vein to the lateral internal mammary vein. Subsequent additional contouring was performed on the flap and sutured to the inframammary fold of the left chest wall for reinforcement, in lieu of the fibrotic and tenuous local tissue quality. Lastly, the abdominal donor site was closed primarily (2-0 PDS [Ethicon Inc., Somerville, NJ], 3-0 Monocryl [Ethicon Inc.], and 4-0 Monocryl [Ethicon Inc.]) without mesh reinforcement given the minimal tension and healthy quality of her innate tissue. At the conclusion of the case, the flap exemplified excellent color and strong Doppler signals along the entire skin paddle.\nThree months after the initial reconstruction, the patient underwent a symmetrizing augmentation of the right breast with a subpectoral silicone implant, and revision of the left breast for improved superior medial fullness and reconstruction of the nipple using a C-Y trilobed flap (Figure ). The patient expressed that she was highly satisfied with overall symmetry and her final result. |
A 49-year-old Hispanic woman presented to the ambulatory medicine service for worsening oedema of her right upper extremity. Six years prior to presentation, she noticed painless swelling of her right thumb, which she initially attributed to an injury that occurred while working in a chicken-processing plant. She reported receiving repeated local injections of an unknown antibiotic in Honduras over the course of 2 weeks without relief. As time progressed, so did the swelling with extension from her hand to shoulder with eventual involvement of her right chest wall and hemithorax (). Her medical evaluation at that time was extremely limited and included a complete blood count and an X-ray, both of which she reported were normal, and hence, no further workup was sought. Eighteen months before presentation to the clinic, she developed dyspnoea on exertion with occasional cough. She reported right arm and back pain from the significant swelling with associated right chest wall discomfort. Apart from postprandial nausea, she denied any fever, night sweats, weight loss, or lower extremity oedema. A computed tomography (CT) scan of the chest with contrast revealed soft tissue oedema without evidence of thrombus or obstruction in the venous system (). Further evaluation for multiple infectious aetiologies, including tularaemia, coccidiodes, and malaria, were negative; however, she did have a positive purified protein derivative (PPD) test for which she received and completed latent tuberculosis treatment. Subsequent lymphoscintigraphy showed evidence of lymphatic obstruction at the level of the distal forearm and she was referred to a lymphedema clinic for compressive therapy. Her oedema continued to progress resulting in a large right-sided exudative chylus pleural effusion requiring thoracentesis. Fluid analysis for at that time was negative for malignancy and cultures were negative for acid-fast bacilli. She again presented two months later with dyspnoea and was found to have a re-accumulation of her pleural effusion at which time she underwent pleuroscopy with biopsy and pleurodesis with pleural drain placement. Although repeat pleural fluid analysis was negative for malignancy, thoracoscopy revealed a pleural space that was densely adherent in multiple locations with gelatinous, firm material. Pathological examination showed sections of very nodular neoplastic proliferation of small spindled to ovoid cells with eosinophilic cytoplasm and bland nuclei within a predominantly myxoid stroma. A ‘whorled’ cellular morphology was identified multifocally without mitotic figures or necrosis. Immunohistochemically, the tumour cells involved were positive for epithelial membrane antigen and progesterone receptor but negative for Ewing sarcoma break point 1 gene translocation, smooth muscle antigen, cytokeratin AE1/AE3, S100, calretinin, microphthalmia-associated transcription factor (MITF), and claudin 1 (). The cytogenetics from the cell cultures of the pleural biopsy revealed complex chromosome translocations and deletions 45,XX,-6,inv(9)(p24q32)x2[14]/90,idemx2[3]/44,idem,dic(19;19)(q13.4;q13.4)[1]/90,idemx2,-6,+7,+11,dic(19;19)[1]/46,XX[1]. Surveillance magnetic resonance imaging (MRI) brain showed no intracranial or meningeal masses. Tissue specimens were referred to a sarcoma expert who identified it as an unusual variant of myxoid sarcoma that could not be classified. Chemotherapy was initiated with doxorubicin that she received every 21 days for 4 cycles. After one cycle of chemotherapy, she had a drastic improvement in her symptoms and physical appearance (). Due to her poor performance status in addition to side effects from her prior regimen, her chemotherapy regimen was changed to gemcitabine. Unfortunately she continued to decline and ultimately opted for hospice care. |
Our patient was a 2-year-old female infant. She presented with three painless vesicles on the lower lip that had emerged 3 weeks earlier. The vesicles had been left untreated due to their fluctuation in size. However, a local dentist who saw the patient for a routine checkup recommended a thorough examination of the vesicles, and thus referred her to our hospital. Her medical and family histories were unremarkable. At the initial visit, three well-circumscribed round masses, each measuring approximately 5 mm in diameter, were found on the lower lip. The lesions were bluish fluctuant masses covered with normal mucosa. No pain or congestion was noted. The facial appearance was symmetric without any skin rashes. No swelling or tenderness was present in the submandibular or cervical lymph nodes. The patient had no fever and her food intake was good. The differential diagnosis of mass lesions in lower lip is salivary gland tumors, fibroma, and hemangioma. Salivary gland tumors of the minor glands can occur on the lip but are more commonly seen in the upper lip rather than in the lower lip. Fibroma is commonly pink and nonfluctunt. Hemangioma can undergo transient reduction in size under pressure. Based on these findings, the lesions were diagnosed as multiple mucoceles of the lower lip. The mucoceles were individually excised, along with the surrounding minor salivary glands, under local anesthesia. We removed three spherical masses, each measuring 5 × 5 × 5 mm in size (Figure ). Histopathological analysis revealed that the cysts were present within connective tissues and lacked lining epithelium. In the minor salivary gland tissues, lymphocytic infiltration was noted around the ducts. These histopathological findings led to a diagnosis of mucoceles (Figure ).\nHer postoperative course was uneventful without any infection or wound dehiscence. To date, 3 years after the operation, the patient has not experienced any recurrence. Informed consent was obtained from the patient's parents, and the procedures were performed in accordance with the Helsinki Declaration. |
A 73-year-old woman with history of Crohn's disease and cholangiocarcinoma invading the duodenum and pancreas underwent pancreaticoduodenectomy (PD). A surveillance CT scan performed several weeks after the surgery revealed two new hepatic lesions (). She had no clinical symptoms. She was referred to the interventional radiology service for biopsy of a liver lesion and for placement of a venous infusion port. Complete blood count, liver function tests, coagulation profile, and basic metabolic profile were all within normal limits. The liver biopsy was performed under moderate sedation with the patient in the supine position. Under CT guidance, a 2.5 cm lesion in segment 8 was accessed with a 19-gauge/20-gauge automatic core biopsy gun (Temno, CareFusion, Waukegan, IL) via a lateral intercostal approach (). A good core of tissue was obtained. A touch preparation was made by placing the core of tissue on a glass slide and rolling the specimen gently around the slide. The sample was immediately evaluated for adequacy by an on-site cytotechnologist. No neoplastic cells were seen on the initial touch preparation. Ultimately five core samples were obtained from different areas of the lesion; each appeared visually adequate, but none of the touch preparation samples contained neoplastic cells on the on-site evaluation. The biopsy was terminated based on CT imaging confirmation of adequate sampling of the lesion. The specimens were submitted for both cytopathologic and surgical pathologic evaluations, which revealed liver parenchyma with chronic active inflammation, granulomas, and a reactive bile ductule proliferation (). No carcinoma was seen. Special stains for mycobacteria and fungal organisms were negative. No microbiology specimens were sent due to lack of clinical suspicion for infectious etiology.\nThe patient developed right upper quadrant pain in the recovery area. She did not have any clinical signs of hemorrhage or sepsis. The pain was controlled with 50 micrograms of intravenous fentanyl. She remained hemodynamically stable throughout a three-hour postbiopsy observation period. The pain was attributed to minimal blood or bile leaking from the puncture site irritating the diaphragm. She was sent home on oral pain medications. The day after the biopsy, the patient continued to complain of right upper quadrant pain. She also developed fever and was brought back to the hospital for further evaluation. She had an elevated white blood cell count of 13.6 K/mcL, increased from prebiopsy value of 5.1 K/mcL. CT scan showed a new right perihepatic/subcapsular fluid collection. This was aspirated under CT guidance and yielded 600 mL of serosanguineous fluid (). The patient was discharged home with instructions regarding signs of hemorrhage and infection. No drainage catheter was placed due to the benign gross character of the fluid. Culture results returned positive for Escherichia coli. The patient was placed on oral ciprofloxacin. In the following 2 weeks, the patient's pain persisted and she developed dyspnea; a CT scan of chest was performed to rule out pulmonary embolism. This showed subsegmental bilateral pulmonary artery embolism and also enlarging, recurrent right perihepatic/subdiaphragmatic collection with new loculations. It was drained with an all-purpose drainage catheter and intracavitary alteplase instillation. Culture showed Escherichia coli. Pulmonary embolism was treated with anticoagulation therapy. The patient was treated and discharged with ciprofloxacin for her perihepatic infection. The collection resolved and the catheter was removed three weeks after placement. CT scan of the abdomen was repeated 3 months after the biopsy, which showed resolution of the hepatic lesions, including the lesion that was biopsied (). The patient had not received any chemotherapy since biopsy. |
A 50-year-old Caucasian female visited the regional dental office due to uncertain pain in the right mandibular region. No significant diseases were mentioned in the anamnesis. The patient was diagnosed with gingivitis, and treated with a non-steroid anti-inflammatory drug and oral rinsing with chlorhexidine. The condition of the patient did not improve significantly. When she returned a week later, she had no alleviation, but labial paresthesia on the right side of her lip. As the cause of the new symptom remained unidentified, she was referred to the oral surgery group. Initially, the paraesthesia affected only the right side of the lower lip, but by this time it had spread to the skin of the right side of her chin. The oral surgeons decided to extract the lower right wisdom tooth, but the labial paraesthesia still persisted. Since there was an extended composite restoration in the lower right first molar, and translucency around its apical region was visible, the lesion was diagnosed as periapical granuloma. The lesion on tooth 46 was not close to the mandibular canal; this can be seen on Fig. . Therefore, root canal treatment was carried out, despite the uncertain result of the percussion, palpation and sensibility test. The transparent lesion around the apex and the negative sensibility test lead to the decision of endodontic treatment. Diagnosis was made by a dentist who is not a staff member of our Department. The treatment revealed that the pulp was vital. Our oral diagnostic team examined the patient, and we diagnosed the case as cemento-osseous dysplasia based on the panoramic radiograph showing apical translucency lesions around the anterior teeth with vital pulp (Fig. ). The paraesthesia has been continually present ever since. Because the lesion around the lower right canine had already involved the cortical bone by CBCT scan (Fig. ), a root canal filling and removal of the apical lesion were performed. On the CBCT image the involvement of the entire periapical region can be seen including the lingual region and the buccal cortical (Fig. ). It became clear during the planning of the surgery that the blood vessels and nerves of tooth 43 would be injured.\nThe root canal treatment was carried out on the lower right canine in a single-visit treatment using local anaesthaesia. We explored the entire root canal length using a size #15 hand instrument. The working length was determined using an apex locator, (Woodpex III, Guilin,China), then the length was also confirmed with radiographic imaging. Following the length determination, the root canal was shaped using Wave One (Densply Maillefer, York, USA) rotary instrument. The root canal was obturated using guttapercha and AH Plus sealer (Dentsply DeTrey GmbH, Konstanz, Germany) with lateral condensation technique. Glass ionomer cement (Fuji IX GP, GC Co., Tokyo, Japan) was then applied to seal off the access cavity, while the permanent restoration was done.\nWe prepared an intraoral mucoperiostal flap using an L-shaped incision and the surgery was carried out by using a surgical microscope (Aspheron, Schmidt and Bender Hungaria, Budapest, Hungary).\nWe opened up the buccal cortical bone using a surgical bur, thereafter we removed the lesion surrounding the apex of the root. The root apex was resected and a retrograde root canal filling was placed using mineral trioxid aggregate (MTA+, Cerkamed, Stalowa Wola, Poland).\nThe area of the lesion was augmented using gentamicin impregnated BoneAlbumin (OrthoSera Dental Zrt., Gyor, Hungary). We seeked to lower the chance of osteomyelitis occuring with the use of gentamicin. Sutures were then carefully placed to achieve tensionfree closure of the flap for optimal healing.\nThe 19-year-old Caucasian woman is the daughter of the first patient. CBCT showed a radiolucent lesion around the root of the lower right incisor (Fig. ). She is presently asymptomatic after a one-year follow-up, but she occasionally felt tension and moderate pain in the right side of the mandibular region eradiating to her ear, approximately 2 years earlier. The symptoms had no dental background, and ceased gradually.\nPulp tests and periapical pathology diagnoses were made by authors on teeth 33,32,41,43 with the use of percussion, palpation and sensibility test.\nPanoramic radiograph and CBCT scans of the first patient showed radiolucent lesions located in the periapical bone, specifically on lower incisors and canines – in the premandibular, and in the right molar region of the mandible with radiopaque parts showing the lesions inside (Figs. and ). The affected teeth were asymptomatic, CBCT proved the presence of apical pathology. The buccal cortical involvement was discovered with the aid of CBCT, which also affected the treatment plan because 2D imaging methods provide no information on bucco-lingual dimension. Prior to the surgical intervention the use of CBCT was motivated by the fact that only 3D imaging could ascertain the precise shape, location and involvement of surrounding anatomical landmarks of the lesion.\nFigure shows the postoperative condition (Fig. ). At the 6-months periapical follow-up, the bone healing is being processed (Fig. ). 18 months later it can be seen on the CT image that the buccal cortical bone surrounding the root of tooth 43 has regenerated (Fig. ). In the case of the second patient, there were no notable changes in the radiographic status 1 year later, and she has no complaint at present.\nThe removed tissue pieces contained both connective tissue and calcified areas. In the calcified area, beside the irregular trabecular – lamellar bony formations, oval and globular cementum-like structures were present. In the non-calcified area connective tissue was found and connective tissue filled out the centre of another bony sample as well. No elements or remnants of a capsule were visible. The histological diagnosis was cemento-osseous dysplasia (Fig. ). |
A 76-year-old female presented to the emergency department in March 2019, with progressive weakness, fatigue, and weight loss of over 20 pounds over a period of 3 months and was admitted to the Internal Medicine Service. Her past medical history included stage-3 chronic kidney disease, peripheral vascular disease with multiple bypass surgeries, gastroesophageal reflux disease, gout, appendectomy, and hysterectomy for uterine cancer. She was also a former smoker with a 35 pack-year history having quit 22 years prior.\nInitial investigations found her significantly malnourished and anemic, with multiple electrolyte disturbances including profound hypercalcemia. A computed tomography (CT) scan of her abdomen demonstrated a 6 cm mass-like lesion arising from the upper pole of the right kidney with a single prominent retrocaval lymph node measuring 1.1 cm, with no evidence of further metastatic disease []. Her clinical condition improved with medical optimization including blood transfusions, hydration, and oral nutritional support. Initially, this mass was felt to be consistent with xanthogranulomatous pyelonephritis by the consulting urology service. Follow-up and biopsy of the lesion were arranged as an outpatient, with a tentative plan for a nephrectomy in April 2019, once the patient had been medically optimized.\nThe biopsy demonstrated a poorly differentiated carcinoma with squamous differentiation. With this diagnosis, further investigations including cystoscopy, urine cytology, and CT chest imaging were performed, all of which were normal. Throughout these investigations, her overall performance status began to deteriorate with progressive weakness and anemia after her discharge from hospital.\nGiven the patient's decline, and after a discussion of her case at multidisciplinary oncology rounds, the plan was to proceed with a right radical nephrectomy with the goal to improve her symptoms and oncological outcomes. It was thought that her anemia and functional decline would progress further without active management of her cancer. A repeat preoperative CT scan demonstrated a significant interval progression of the disease in a 20-day period []. The renal mass was now found to be locally invasive with possible invasion into the right psoas muscle and the right renal vein abutting the inferior vena cava.\nA radical right nephrectomy with inferior vena cava thrombectomy with patch graft angioplasty was performed in early April 2019. Intraoperatively, the mass was noted to be significantly adhered to the adjacent organs including the inferior vena cava. The final pathology demonstrated pT3aN1 SCC measuring 8.5 cm with 50% tumor necrosis with invasion into the renal sinus and collecting system with 1/1 hilar lymph nodes positive [Figures and ].\nThe patient recovered quickly and her overall condition improved. She was discharged home in 6 days. Follow-up investigations demonstrated that the patient was normocalcemic, improvement in her anemia, and a CT scan of the abdomen demonstrated no residual tumor or recurrence 2 months postoperatively. Functionally, the patient had a near complete resolution of her preoperative symptoms. |
A 41-year-old female patient was referred to our pain clinic for management of chronic multiple joint pains with a recent diagnosis of EDS-type 3. The patient worked as a laboratory technician and suffered from chronic joint pains for more than 5 years. Although she was under treatment she reported worsening pain and an inability to function. She had worse pain in both knee joints, followed by hands and wrists, and shoulders. Intensity of pain was assessed by 0 to 10 cm visual analog scale (VAS) and pain interference was assessed using pain disability index (PDI).[ Her baseline intensity of pain was 4 to 5 cm in and could get as high as 7 to 8 cm on activity. Any activity, including walking, standing, or folding laundry, would trigger increase in pain. She indicated more disability in the domains of social activity and family/home responsibilities. Her pain was worse in the mornings associated with stiffness. A genetic testing in 2016 had confirmed a diagnosis of EDS-type 3. Further elicitation of her history confirmed that she had hypermobile joints since her childhood with easy bruising on her legs, without any provocation. But she did not have a history of epistaxis, or bleeding from the gums or intra-articular bleeding. She had sustained musculoskeletal injuries before including an ankle, elbow, and humeral fracture and also had undergone ulnar nerve transposition at the age of 19. Her history revealed that she did have kinesiophobia concern about movement-related injuries as she was prescribed with exercises. Her profile did not reveal any obvious contributory psychological factors such as anxiety, depression, or high catastrophizing. She was being treated with long acting tramadol 200 mg once a day along with celecoxib as a rescue medication daily. Overall, she felt that her pain relief was not better than 30% to 40% with these interventions. Her comorbidities included hypertension for which she was on medications. Her examination revealed that she had lax skin at the neck and upper chest area. No signs of swelling or injury to the joints were observed. She was not using any braces for her symptom control. We did not engage in an extended range of joint mobility for fear or possibility of subluxation.\nFrom the very beginning, patient was assessed by a team, which included a pain physician and a nurse, a pain psychologist, and a kinesiologist (with special training in pain management), as part of our multidisciplinary pain program, as a part of pain evaluation and treatment. As a treatment strategy, the group agreed that the patient will need measures to better control her pain, improve her coping skills and decrease her kinesiophobia along with measures to stabilize her musculoskeletal symptoms. Although we considered treatment with baclofen to reduce her morning symptoms, we decided it was more appropriate to start her on a tricyclic antidepressant to decrease her pain and desensitize her symptoms. She was started on nortriptyline 20 mg bed time dose and patient was asked to continue with her tramadol in the morning and use celecoxib on as needed basis. She was provided with self-management strategies to improve her coping and use appropriate pacing techniques. She was also provided education around postural awareness and improved body mechanics, during work and relaxation. The appropriate use of kinesio-taping measures was suggested to stabilize her joints. An exercise prescription with graded exercises, including pool activity was also provided. A follow-up visit after 2 months revealed that the patient expressed significant decrease in pain intensity (VAS of 2 cm at rest and 4 cm on activity) and improvement in her ability to engage in family responsibilities and social activity. Presently, more than 18 months after the initial visit, the patient continues to be on good pain control with nortriptyline and tramadol only on a daily basis and is confident of managing her symptoms using her self-management tools. |
A 58-year-old Caucasian woman presented at our institution with immobilizing back pain, known for 4 years but exacerbating for 3 months. The past history included smoking (about 15 pack-years), appendectomy and tonsillectomy as a child, and extensive endometriosis which required surgical excision of sigmoid colon and hysterectomy sixteen years earlier. Conventional X-rays of the thoracic and lumbar spine revealed degenerative alterations only. However, magnetic resonance imaging (MRI) demonstrated multiple lesions in all vertebral bodies with a pathologic fracture in the 8th thoracic vertebral body. Blood count was completely normal. Suspecting bone metastases of a yet unknown primary tumour, a thoracoabdominal CT scan was performed, revealing disseminated small nodules in both lungs (max. 1.0 × 0.5 cm in diameter) and small polypoid intraluminal lesions in the gall bladder infiltrating the liver (), the latter showing additionally some very small lesions measuring only a few millimeters in diameter. An ultrasound confirmed a solid tumour mass in the fundus of the gall bladder, highly suspicious for primary gall bladder cancer. Upper endoscopy and lower endoscopy were inconspicuous. External biopsies of a suspected pulmonary lesion and the thickened conglomerate of gall bladder wall towards liver were both not diagnostic. Repeating one of these interventions seemed not promising. Palliative radiotherapy (5 × 4 Gy) of the thoracic spine was performed. Unfortunately, severe mood depression and panic attacks occurred, delaying further diagnostic and therapeutic interventions. Six weeks later, a repeated CT scan revealed progression of the bone lesions and significant thickening of the gall bladder wall. MRI did not detect brain metastases. For further diagnosis, we discussed with the patient pulmonary wedge resection, vertebral biopsy, or cholecystectomy. With the two unsuccessful attempts in mind, she chose the latter procedure, suggesting the highest probability to finally reach a diagnosis. This intervention was then performed without complications and provided the following findings.\nMacroscopically, the gall bladder specimen was 9 cm long and 3 cm in diameter. In the lumen, a friable broad based papillary nodule in the fundus (5 cm maximum diameter) and two polypoid satellite nodules in the neck (1 cm maximum diameter) were present. In the cystic duct, a small gallstone was identified. Histopathologically, surprisingly, the nodules corresponded to a malignant melanoma. The tumour was almost completely confined to the mucosa () with only focal invasion of the muscularis propria and subserosa. It was composed of pigmented polygonal cells with vesicular nuclei and prominent eosinophilic nucleoli (). Numerous mitotic figures could be encountered. The lining of the villi of the gall bladder mucosa consisted of tall columnar cells with in-between lying pigmented neoplastic cells and macrophages (). Junctional activity, meaning presence of microscopic aggregates of melanoma cells at the junction of epithelium and lamina propria, was present (). Tumour cells stained positively for S100, HMB45, Melan A, and CD117 by immunohistochemistry and melanin pigment with Fontana-Masson stain. Neither PDGFRA nor kit mutations were detected.\nAs neither BRAF inhibitors nor checkpoint inhibitors were yet available in clinical practice, a palliative chemotherapy with temozolomide (200 mg/m2 daily for 5 days) and monthly infusions of zoledronic acid were installed. Temozolomide was preferred to the former standard of care Dacarbazine, as equal efficacy was demonstrated [] and the oral application convened to the patient.\nUnfortunately, the patient developed severe myelosuppression CTCAE grade 4, requiring several transfusions of erythrocytes and platelets, as well as the application of G-CSF. Another CT scan performed after first chemotherapy cycle showed progression of the lung and liver metastases, newly developed spleen metastases, and pathologic fractures of the lumbar spine on levels 3 and 5. Because of the severe side effects without rendering any effect on tumour progression, the patient refused another series of palliative radiotherapy as well as another attempt of systemic therapy and decided to look for alternative treatment regimens. Two months later, she presented again with exacerbating bone pain, severe anemia (hemoglobin: 5.8 g/dl), and thrombocytopenia (36 G/l) as a result of progressive bone marrow infiltration. After installation of an analgesic therapy, the patient was transferred to a hospice institution, where she died a few days later. |
A 56-year-old female with a past medical history of COPD, GERD, and bipolar disease was transferred to our institution following inpatient treatment at two outside hospitals. She had been admitted to the first hospital with abdominal pain and found to have gallstone pancreatitis, cholangitis, and Streptococcus salivarius bacteremia. While a cholecystectomy was considered, she instead underwent ERCP to remove the stone and a percutaneous cholecystostomy tube was placed by interventional radiology. After three weeks, she was discharged with a course of oral antibiotics.\nTwo weeks later she was admitted to a different facility with right upper quadrant abdominal pain, nausea, and vomiting. Liver enzymes and total bilirubin were elevated. The patient revealed that she had not taken the full course of oral antibiotics. ERCP was performed with small stones seen. The patient underwent a planned cholecystectomy which began as laparoscopic and was converted to open. This procedure was unsuccessful and a cholecystostomy drain was placed with intraoperative cholangiogram showing an obstruction in the common bile duct. Gastroenterology made the decision to abort the procedure and instead place drains in the biliary tree.\nOne week later the patient was transferred to our institution for higher acuity of care. An open cholecystectomy with common bile duct exploration and common bile duct repair was performed. Postoperatively labs remained elevated with white blood cell (WBC) 19.1 K/UL, alkaline phosphatase 1060 U/L, total bilirubin 15.79 mg/dL, and direct bilirubin 9.86 mg/dL. A series of cholangiograms revealed intrahepatic biliary strictures and bile casts in the intrahepatic biliary tree. Interventional radiology attempted drainage of bile but was unsuccessful. The patient's WBC count and LFTs continued to rise and she progressed to liver failure. Cardiology consult was obtained but the patient refused a recommended pharmacologic stress test. A psychology consult commented that the patient was not psychologically stable for a liver transplant.\nGiven the past procedural and surgical attempts to drain biliary casts, the decision was made to attempt to dissolve the casts using methyl tert-butyl ether (MTBE). The patient was brought into the operating room where she was given general anesthesia via an endotracheal tube. MTBE was directly injected into the biliary system with 30 mL ether injected in the right biliary system and 46 mL injected into the left biliary system. Following MTBE treatment, there was marked improvement of the right biliary duct system and modest improvement of the left biliary duct system (). The patient's alkaline phosphatase decreased to 590 U/L, total bilirubin 14.44 mg/dL, and direct bilirubin 8.79 mg/dL, but WBC remained at 15.3 K/UL. She continued to have persistent Gram-negative rod infection for which she remained on long term antibiotics. Twenty days following MTBE injection, and a little over 2 months after being admitted to our institution, the patient was discharged to a rehabilitation center. |
A 65-year-old nondiabetic male presented to us with history of chest pain of 3-hour duration two days ago, associated with vomiting and one episode of syncope. For these complaints, he had been evaluated at a local hospital and diagnosed as acute inferior wall with posterior wall MI with atrioventricular (AV) dissociation, for which he was managed medically, and transvenous right ventricular apical temporary pacing was done. Due to persistent hypotension and recurrent rest pain, the patient was referred to us on the third day of the index event. His presentation ECG at our centre showed evidence of inferior wall MI with 2 : 1 AV block (). Echocardiogram showed hypokinesia in the left circumflex (LCx) territory, with an ejection fraction of 50% and moderate mitral regurgitation. Pacing threshold was normal and remained so throughout the observation period. In view of postinfarction angina, hypotension, and persistent AV block, the patient was taken up for coronary angiography with an intent to revascularise. His coronary angiography showed a left dominant circulation with 90% stenosis in proximal segment of left circumflex artery and 70% stenosis in the middle segment of the left anterior descending (LAD) artery. The LCx was balloon dilated followed by deployment of a drug eluting stent; the LAD was planned for a staged procedure. The angioplasty was uneventful and the patient was shifted to ICCU.\nAfter four hours of percutaneous coronary intervention (PCI), the patient suddenly developed palpitations and ECG showed monomorphic ventricular tachycardia (). He was managed with 150 J biphasic DC cardioversion followed by intravenous amiodarone bolus and infusion, considering it to be secondary VT. The serum electrolytes were within normal limits. There were multiple hemodynamically significant episodes of both monomorphic () and polymorphic VT () over the next few hours. Significant ischemia was effectively ruled out by a check angiography that showed patent LCx stent with TIMI 3 flow as well as an insignificant rise in CPK-MB.\nCareful inspection of serial ECG tracings of the patient showed that each episode of either monomorphic or polymorphic VT began after a fusion complex consisting of the paced and intrinsic beats (Figures and ). The temporary pacing lead was removed and there were no VT episodes after that. The patient had an uneventful convalescence; he was taken off amiodarone and discharged in a hemodynamically stable condition few days later. Staged PCI to LAD was done one month later; the procedure was uneventful. The patient is currently in our follow-up and doing well. |
At 18 year old, a Caucasian female patient without any systemic diseases or drug use was presented for evaluation of gingival problems around her upper right premolar. Marginal periodontitis was diagnosed by her Dentist and the patient was referred to a Periodologist for root scaling. During the next two years, the patient was undergone perio-treatments under anti-inflammatory medications and had root canal treatment of her second premolar one year later. Biopsy of the lesion was not taken but the lesion was not gone but slowly progressed and therefore the patient was referred to a maxillofacial surgeon at our clinic. During her first consultation with the maxillofacial surgeon, the patient only complained about gingival recession in the palatal area of her upper-right-side teeth. The initial examination showed that the canine and both premolars had second-grade mobility (Fig. ). The probing depth of teeth 13, 14, and 15 was < 3 mm on the buccal side and 5 mm at the palatal side as the palatal gingiva were recessed leaving exposed the fist premolar and canine and less the second premolar whose exposed root surface was covered with dental plaque. The patient’s jaw underwent 3D computed tomography, which revealed bone destruction in the defect area reaching the maxillary sinus, whose mucosa was locally thickened (Fig. ). The condition of periodontium around other teeth was fine. The probing depth was < 3 mm around all other teeth. The chronic long-term lesion, with its unknown cause and unusual localization (defect in the palate side, while the marginal buccal surface bone was uninjured), caused confusion to the Oral Surgeon as the clinical findings were not characteristic of oncological tumors: such as the absence of induration of adjacent soft tissues, the boundaries were clearly visible, and the unpleasant smell characteristic of tissue collapse was not detected from the lesion. Moreover, no regional lymph nodes were palpable, and none of the patient’s close relatives were having similar lesions.\nIt was decided to remove all three teeth (13th, 14th and 15th), perform a removal of the altered soft tissues, and evaluate them histologically. After radical surgery of the defect, it was decided to reconstruct the area using allogenic bone transplant. Surgery was performed under local anesthesia. The initial prosthetic treatment plan was to insert dental implants in the area of teeth 13 and 15 about 6 months after the excision of the altered tissues and to make a fixed 3-tooth bridge on the dental implants afterward. However, after the diagnosis was histologically confirmed, prosthetic treatment was delayed for 12 months after excision in case relapse did not occur.\nThe teeth were removed during surgery, and the altered soft tissues were removed based on the clinical view, within the boundaries of healthy tissues. Soft tissues with granulations and teeth were easily separated from the bone. The bone relief was uneven but hard and was covered in a compact bone layer. The formed bone defect was more similar to uneven bone lysis than to destruction (Fig. a, b). All of the resected tissues were sent for histomorphological evaluation. After the teeth with altered tissues were removed, the bone window (uzura) to the sinus was visible, but the integrity of the membrane was intact. The membrane, as visible through the window, looked unchanged.\nSoft tissue deficit was present from the crest to the hard palate. Tumor-free margins were about 20 mm wide mesiodistally and about 15 mm wide mediolaterally (Fig. a, b). Sticky bone graft with platelet concentrate made from venous blood according to the PRGF Endoret® method, in combination with demineralized freeze-dried bone allograft was chosen for restoration of bone defect []. The soft tissue defect was covered with free gingival flap from the buccal to the palatal side. Antibiotic therapy (875 mg of amoxicillin with 125 mg clavulanic acid twice a day for 7 days) and painkillers (25 mg of Dexketoprofen according to the patient’s needs, no more than 3 times per day for 5 days) were prescribed after surgery. The wound healed without complications, and the sutures were removed after 10 days.\nThe excised gingivae and adjacent oral mucosa sent for histological examination were partially ulcerated while the underlying submucosa was consisted of complexes of atypical epithelial cells of different shapes in fibromycoidic stroma (Fig. a–d). These cells had eosinophilic cytoplasm, insignificantly polymorphic oval cores, and isolated mitoses. Part of the cytoplasm of the atypical cells contained vacuoles with erythrocytes in openings. Immunohistochemical staining with endothelial markers CD31, CD34, and ERG was positive for 100% of the tumor cells (Fig. a–c), and reaction with the epithelial marker PanCK was positive for 10% of the tumor cells. The proliferative activity of Ki-67 was about 5%. ''The included bone did not reveal any infiltration from tumor cells. The final histomorphological conclusion was epithelioid hemangioendothelioma pT1b of low malignancy in the periodontal tissues and palatal mucous membrane.\nThe further examination of patient after histopathtological results was set up during the consultation with oncologist, who indicated the need of thoracic X-ray and abdominal ultrasound to identify any possible distant metastasis. An ultrasound head and neck region examination revealed isolated II A group neck lymph nodes up to 0.5 cm in diameter, which were considered to be reactive. Suspicious lymph nodes were not found. Ultrasound of the abdomen and chest X-ray did not show any pathology.\nAfter 31 months, there have been no clinical signs of relapse. Three-dimensional computed tomography of the jaws was carried out 5 months after surgery and showed that the augmented bone had retained its shape but without complete mineralization. Considering possible relapse, the patient was scheduled for additional consultations and examinations after 3, 6, and 12 months. Prosthetic treatment was started 12 months after the tumor’s excision. Two dental implants were inserted in the area of the 13th and 15th teeth. Six months after implantation, prosthetic implants were installed with a 3-unit zirconium ceramic bridge (Fig. ). The patient had no complaints about her condition 12 months after this prosthetic treatment. |
In May 2018, an immunocompetent 65-year-old man with no familial history of skin cancer was recovered due to a large ulceration involving the proximal portion of the right leg causing hypofunctionality of the limb and osteomyelitis with loss of neuronal substance. A cutaneous biopsy demonstrated an SCC. The ulceration was too large to be subjected to local excision; therefore, after a complete radiological staging with chest and abdomen computed tomography that did not show distant metastases, amputation of the middle third of the right leg was made. The definitive histological examination showed an ulcerated area of 13 cm, necrotic, haemorrhagic, incorporating and full-thickness infiltrating from skin to the underlying bone tissue, compatible with poorly differentiated SCC. For persistence of secretions, the wound was revised and cleaned several times. One month after amputation, a radiological restaging with chest and abdomen computed tomography showed multiple bilateral lung lesions compatible with distant metastases and a dubious intracardiac nodularity. The patient was then studied for a possible infectious disease but all the exams (including tuberculosis screening) were negative. We performed a fibrobronchoscopy with biopsy of a mediastinal lymph node and bronchioloalveolar lavage, resulting in positive SCC in both samples. The following positron emission tomography highlighted multiple lung localisations (Standardised uptake value 20), widespread skeletal accumulation referable to osteomidullary activity compatible with substitutive meaning and a left and right atrial nodularity compatible with metastases (standardised uptake value 7, 4). Echocardiography revealed a solid lesion on the atrial side of the anterior tricuspid flap; the ejection fraction determined to be 85%. A transesophageal echocardiography documented in the right atrium a voluminous tripartite formation with a sessile spherical base of 3 cm in diameter and two other non-homogeneous mobile formations of 2 and 5 cm in diameter, each comes into contact with the atrial margin of the tricuspid valve. In the left atrium, evidence of a roundish 2 cm diameter formation anchored by a peduncle to the atrial roof was compatible with metastases (–). A cardiac surgery consultation determined that the extent of the disease, including at the pulmonary level, suggested that surgical intervention was inappropriate. A cardiac nuclear magnetic resonance was unsuccessful since the patient experienced rapid dyspnoea that arose from lying in a supine position. Despite the extent of the disease and the cardiac involvement, considering the young age of the patient and the conserved cardiac function, we proposed a platinum-based chemotherapeutic treatment; however, when informed of the possible risks and benefits of the treatment, the patient preferred not to undertake any oncological treatment. |
A 51-year-old male with a history of uncontrolled hypertension, morbid obesity, and sleep apnea presented initially to an outside hospital with findings of an acute myocardial infarction from a thrombus in the RCA requiring emergent percutaneous catheterization intervention with a drug-eluting stent (DES). The following day, CT scan revealed a type A aortic dissection with the dissection flap extending infrarenally. This prompted immediate admission to our institution for further management. As the patient was on prasugrel for the DES and was hemodynamically stable, surgical intervention was delayed to allow for platelet function recovery. Six days after admission, the patient was brought to the operating room for repair of his type A aortic dissection. Intra-operative 2D TEE was consistent with RCA obstruction and demonstrated mild right ventricular dysfunction, moderate left ventricular systolic dysfunction, and an akinetic inferior wall. 3D TEE provided dynamic visualization of the dissection flap protruding onto the non and right coronary cusps and into the aortic valve causing aortic regurgitation with normal aortic valve leaflets. In addition, the right coronary orifice was clearly visualized to be within the false lumen [].\nSurgical exploration confirmed 3D TEE findings of the false lumen extending to the aortic valve annulus with complete avulsion of the origin of the RCA. A Yacoub valve sparing root replacement was performed along with partial replacement of the aortic arch using an ascending aortic arch graft, including an innominate and left carotid bifurcation graft. The patient also required coronary artery bypass of the RCA with removal of the RCA stent. A mitral valve annuloplasty ring was also placed to repair significant mitral regurgitation. Post repair, worsening right ventricular function required implantation of a right ventricular assist device (RVAD). The patient remained on an RVAD with an oxygenator after surgery with successful weaning and removal of the RVAD about 1-week later. After a long hospital course, the patient was discharged to a rehabilitation center with good recovery at follow-up several months later. |
A 54 year-old Korean man visited the orthopedic surgery department due to persistent pain in his right hip joint starting a month previously and a gait disturbance was caused by this pain. He received a stent due to coronary artery occlusive disease in 2006 and received arthroscopic surgery for a rotator cuff tear in 2012. He did not have any notable medical history related to his joints, such as septic arthritis, tuberculosis, and rheumatoid arthritis. He was not undergone joint injection before. The pain started without any trauma about a month prior and worsened with exercises and walking. On a physical examination, he showed severe limping, no signs of rebound tenderness, and a positive sign to a Patrick’s test including a severe decrease in joint ROM (flexion, 100°; extension, 0°; abduction, 30° internal rotation, −5°, external rotation, 20°). Spine and sacroiliac joints did not show any significant findings upon physical examination. A simple radiograph showed no remarkable findings thus an magnetic resonance imaging (MRI) was conducted. Exudation in the right hip joint was increased, and his acetabular articular cartilage and acetabular labrum were almost completely obscured (; ). His C-reactive protein and erythrocyte sedimentation rate were in the normal range. Serum markers for rheumatoid arthritis, including the rheumatoid factor, were also normal. The patient did not have a family history of rheumatoid arthritis. The patient was initially diagnosed with transient synovitis of the hip and arthrocentesis was administered after hospitalization. The findings of the hip joint fluid were all normal. After hospitalization, he was enrolled in a conservative treatment, including traction and physical therapy, yet he did not noticeably improve. Accordingly, a total hip replacement arthroplasty was performed one week later (). The findings in the operating room revealed extensive chondrolysis of the articular cartilage in the head of the femur (). From the biopsy, nothing other than the synovial vitreous body was found. Polymorphonuclear cells were not found ().\nThe patient did not experience any pain on the operated hip joint and revisited the hospital due to pain in his left hip joint one year after the surgery. The pain occurred without any trauma as in the first incidence. Similar to his previous visit, he severely limped, and again, displayed no signs of rebound tenderness, a positive sign to the Patrick’s test and a severe decrease in the joint ROM (flexion, 90°; extension, 20°; abduction, 30°; adduction, 10°; internal rotation, 0°; external rotation, 20°). Significant neurological findings in the lower body were not found. There were no remarkable findings on the simple radiography so again, an MRI was ordered. The MRI revealed an increased amount of joint effusion as noted in his left hip joint with much debris and bone marrow edema at the left acetabular roof and at the lateral portion of the femoral head, revealing cartilage thinning at the left acetabulum. Progressed degeneration of left labrum was compared to the prior MRI (; ). We suspected chondrolysis of the left femoral head as was the same with the right side and thus the patient received a left-side, total hip replacement (). Extensive chondrolysis of the right femoral head was uncovered during the surgery (). The HHS (Harrison hip score) was 67 points preoperatively, which improved to a 96 points at the last follow-up. The VAS (visual analogue scale) score was 7 points preoperatively, there was not any pain at the last follow up. No complication was found during follow period. Currently, at 21 months after the right hip joint surgery and 9 months after the left hip joint surgery, the patient is not complaining of any symptoms and continues to enjoy his daily life. |
A 41-year-old woman presented to the emergency room after sustaining 2 shotguns wounds in the abdomen and left thigh from a short distance. Upon admission, the patient was hemodynamically stable with a normal blood pressure and respiratory rate. Physical examination showed multiple ballistic wounds in the abdomen, both thighs and groins, associated with a deep wound on the upper-outer side of the left thigh. No exit wound was identified. Palpation found an upper abdominal guarding. Peripheral pulses were normally palpated in the 4 extremities.\nNo abnormality was detected on the focused assessment with sonography for trauma. Whole-body CT scan using spiral acquisition with intravenous contrast administration and iterative algorithm for metal artifact reduction reconstruction was performed. It showed multiple small pellets within thighs, groins, abdominal wall and inside the abdomen and pelvis (). Mild pneumoperitoneum was present without any evident associated organ or vascular injuries. At the supra-diaphragmatic level, an isolated metallic foreign body was seen at the lower part of the right ventricular wall (). Its exact location was unknown due to metallic artifacts and cardiac motion, the patient was tachycardiac with a heart rate of 115 during the acquisition.\nThe patient underwent a laparotomy which showed no evidence of digestive or vascular injury. The transthoracic echocardiogram (TTE) showed a small echogenic foreign body, located in the infero-lateral wall of the right ventricle, causing posterior acoustic shadowing (). No pericardial effusion was detected. The patient was hospitalized for the next 2 weeks for wound debridement and care.\nThe metallic object seen on CT scan and TTE could be a penetrating pellet which directly reached the mediastinum. Another possibility is a migrating pellet through the venous system to the right heart cavities even though there were no evident associated vascular injuries seen on the whole-body CT scan.\nECG-gated Cardiac CT scan was performed 5 days after admission using retrospective gating and iterative algorithm for metal artifact reduction reconstruction after administrating 10 mg of intravenous Tenormine (Atenolol) reducing the heart rate from 100 to 75. It confirmed the presence of a right ventricular intracavitary metallic pellet measuring 3 mm entrapped within the trabeculations throughout the cardiac cycle (). There was no evidence of a penetrating thoracic injury or pericardial abnormality. The most likely diagnosis was therefore a metallic pellet embolism to the right ventricle migrated from the femoral veins.\nTTE was repeated 3 days later during the hospitalization showing the pellet within the same location in the right ventricle. It was decided after a multidisciplinary team meeting to follow a conservative management and to start prophylactic anticoagulation for 1 month. It was controlled by serial TTE rather than CT scan to avoid radiation exposure, looking for signs of stability, migration or myo-pericardial complications. On the first 2 monthly follow-ups, it was stable and no further investigations or workups were done. |
A 42-year-old male presented to the Department of Head and Neck Surgery of a tertiary oncological centre because of right mandibular swelling and trismus. The patient had already been admitted to a secondary care hospital 3 months earlier with complaints of right mandibular discomfort and slight tumefaction. A biopsy was then performed and the diagnosis of ameloblastic carcinoma was made histologically. The patient was otherwise healthy with no significant past medical history, including alcohol, smoking or tobacco abuse.\nA complete head and neck examination revealed a painless, firm and fixed right mandibular mass with no cutaneous inflammatory signs. No ulcers or mucosal lesions were found in the oral cavity. Laboratory evaluation, chest radiograph and respiratory function tests were unremarkable. The patient underwent bronchofibroscopy, which revealed only mild laryngeal hyperaemia.\nBoth neck CT () and MRI () were performed, showing a large, solid tumour arising from the ramus and posterior body of the right mandible. The lesion extended to the surrounding soft tissues, with invasion of the masseter and medial pterygoid muscles and caused bulging of the buccal mucosa. The soft tissue component was hypointense on T1 weighted and hyperintense on T2 weighted MR images and showed avid enhancement after gadolinium administration on MR examination. CT scan disclosed striking sclerosis and irregularity of the mandibular ramus with some gas bubbles inside the medullary cavity and an expansive lytic component in the posterior body and angle with some bone-forming matrix inside. Prominent periosteal reaction was also identified, particularly in the outer cortical surface of the mandibular ramus with the typical pattern of a ruptured Codman triangle. No associated cystic lesion was found in the mandible. No enlarged lymph nodes were detected and the evaluation of the remaining cervical spaces was unremarkable. A thoracic CT scan was also performed, with no parenchymal lesions.\nThe patient underwent a right hemimandibulectomy and ipsilateral cervical lymph node dissection. Surgical resection also included the right submandibular gland and a segment of buccal mucosa that was swollen by the mandibular mass. Reconstruction was performed with free fibula graft. The surgical specimen included a large, white and solid tumour with 11 × 7.5 × 6.5 cm, corresponding to an invasive, moderately differentiated (G2) SCC (). Focal positive margins were found at the medial surface of the specimen. The resected buccal mucosa, submandibular gland and lymph nodes had no neoplastic tissue. Taking into account the imaging staging examinations and the post-surgical histological report, the final TNM stage was stated as pT3 N0 Mx. After surgery, the patient underwent adjuvant chemotherapy (cisplatin-based regimen) and radiotherapy. |
A 10-year-old boy presented with mild shortness of breath that aggravated on physical exertion. Cardiovascular examination revealed a normal size heart with an evidence of small to moderate sized VSD with a left to right shunt. There was a short flow murmur across the mitral valve suggestive of significant left to right shunt. His 2D echo color Doppler showed a PMVSD measuring 7–8 mm on the LV aspect with two jets emerging on the right ventricular (RV) side; the larger jet measuring 6 mm. He had a TV aneurysm tending to cover the VSD with a resultant mild TR. In view of his symptoms and significant left to right shunt, he underwent device closure of PMVSD using 10 × 8 ADO I. The procedure was uneventful and he was discharged the morning after the procedure with a tiny residual shunt through the additional fenestration and with a mild TR. Six weeks later, during the follow-up visit, his TR had progressed to little more than mild grade. However, at 6-month follow-up, the TR became moderately severe although he continued to remain asymptomatic. His RV showed mild volume overload with normal contractility. He was then advised surgical intervention; however, the family deferred the same for almost a year at which time the TR became severe [Figure , and Video , ] with right atrial and RV enlargement.\nAt the time of surgery, the right atrium was opened in the usual manner under cardioplegic arrest. Part of the anterior leaflet and the septal leaflet was found torn and part of the leaflet was flail []. In addition, half of the septal leaflet was stuck to the VSD device. These two components of immobility of the septal leaflet and torn anterior leaflet were responsible for the TR. The tricuspid annulus was not dilated. During repair, the adherent leaflet was mobilized and flail removed by a triangular-shaped incision with the apex pointed toward the orifice and base at the annulus. A triangular pericardial patch treated with glutaraldehyde was stitched in the gap with continuous sutures []. Valve function was checked with cold saline injection and was found to be good with minimal TR and no tricuspid stenosis. The TV chordae and papillary muscles were intact. His postoperative recovery was uneventful. His 2D echo color Doppler examination at 3-month follow-up revealed the device to be in the position with a tiny residual shunt. There was a mild residual TR [Figure , and Video , ]. His right atrium and RV showed remarkable reduction in their size. |
A 67-year-old female with past medical history of congenital deafness presented to the emergency room with complaints of right-sided facial droop and right upper extremity weakness, tingling, and numbness. These symptoms were sudden in onset and lasted for a few minutes. Symptoms had completely resolved at the time of presentation. She did not have a history of any atherosclerotic risk factor including hypertension, diabetes, or hypercholesterolemia. The initial set of vital signs were normal; routine laboratory tests including complete blood count and basic metabolic panel were unremarkable. A computed tomography (CT) of the head without contrast as well as a magnetic resonance imaging (MRI) of the brain with and without contrast did not show any acute intracranial hemorrhage or infarction.\nThe patient was diagnosed with TIA, and further investigations were planned to determine the etiology. A magnetic resonance angiogram (MRA) of the head and neck with and without contrast did not show any arterial flow limiting stenosis or occlusion. A transthoracic echocardiogram (TTE) with bubble study using agitated normal saline contrast was performed and was found to be normal. Patient's heart rhythm was monitored with continuous cardiac monitoring, and no arrhythmias were noted during her stay at the hospital. At this point, the patient was identified as having cryptogenic TIA, having failed to determine the precise etiology from routine workup. Patient was started on aspirin therapy and discharged from the hospital on day 3 with further outpatient workup planned. Outpatient workup for hypercoagulability showed a high factor VIII activity of 153%, which potentially put her at increased risk of venous thromboembolism. However, this test was performed just one week after the thrombotic event and was hence difficult to interpret. Subsequently, a transesophageal echocardiogram (TEE) was performed that revealed a patent foramen ovale with right-to-left shunt. This raised the concern for paradoxical embolism as the cause of patient's TIA. Lower extremity duplex venous ultrasound showed no evidence of deep vein thrombosis. However, magnetic resonance venogram (MRV) of pelvis showed compression of the left common iliac vein just after its origin, which was suggestive of May-Thurner syndrome (). There was no evidence of venous thrombosis on the MRV. May-Thurner syndrome was recognized as the probable source of paradoxical embolism causing TIA in the patient. The patient was eventually referred for percutaneous PFO repair, which was performed without any complications. The patient had been regularly followed yearly at the cardiology clinic for 5 years now. She remains in good health with no further episodes of TIA. |
It is a case report of a 25 years old male presented to us in outpatient departments with chief complaint of discharging wounds over mid part of his left arm since six months. On the complete history taking he revealed that he had an episode of trauma one year back over his left arm due to fall on the ground and swelling develops at that time. For it he did not take any medical advice and he took few pain killers and repeated massage over his left arm. Subsequently he noticed that the swelling over his whole left arm is permanent. After six months of this incident he felt that now mid of his arm is pain full and after 2 to 3 episodes of fever pus discharge came out. Since then, on and off the pus (sometimes with chunk of bone) is coming out.\nOn physical examination there were multiple discharging sinuses (with the protruding bony chunk) over his lateral aspect of his mid part of the left arm with puckered and hyper pigmented skin []. On palpation his left arm was thickened and tenderness was found around the arm. Range of movements over left shoulder and elbow were terminally restricted and pain full. Vital signs of patient were stable and distal neurovascular status was intact. Clinical provisional diagnosis of chronic osteomyelitis of left humerus made and his hematological examination and X-ray sought. His hematological study was near normal except the erythrocyte sedimentation rate (ESR) which was raised (Westergren’s method=35 mm/hour).\nX- Ray was astonishing, entire humerus bone was normal, and there was no any osteomyelitic change in the humerus. There was a geographic appearance of myositis ossificans around the upper two third of left arm [].\nAfter consent taking, the patient got operated. Sinuses curetted out and sequestrum (infected part of myositis mass, because it was not feasible to remove the all myositis mass) removed and sent for biopsy and culture sensitivity. Now after the one year of follow-up, there is no recurrence of the myositis ossificans. And the patient is free from discharging sinuses and has resumed his work. |
A 71-year-old woman was referred for further evaluation of an uneven surface mucosa in the esophagus, in a Barrett’s area on the anterior wall of the distal esophagus where well-differentiated adenocarcinoma was diagnosed with biopsy. The patient has a history of cerebral infarction at age 50 years, from which she recovered, except for moderate left hemiparesis. Although she also has a history of chronic heart failure, her cardiac function was improved. Her physical examination was normal; blood and serum chemistry tests including coagulation studies were normal; and chest and abdominal computed tomography scans showed no metastatic lesions. She has been routinely followed up for the development of malignant lesions in a long segment of Barrett’s esophagus.\nAccording to the Prague classification, the circumferential extent (C) of Barrett’s esophagus was 4 cm and the maximum length (M) was 7 cm. By using a laser imaging system, we found reddish lesions on the anterior and posterior walls of the distal esophagus in white light laser images (). In addition, another lesion in the middle esophagus was detected as a brownish area, by using FICE and BLI (). All three lesions were 0-IIc early cancers with slight reddish color changes in the laser white light images: two lesions measuring 15 and 10 mm in diameter on the anterior and posterior walls of the distal esophagus, respectively, and another lesion measuring 5 mm in the middle esophagus. FICE and BLI enhanced all three lesions with high contrast, compared with the surrounding mucosa. Magnification BLI images revealed an irregular surface pattern on the brown lesion compared with the surrounding mucosa and an irregular vascular pattern in one portion of the tumor. All biopsy specimens showed atypical cells, suspicious for well-differentiated adenocarcinoma.\nThree synchronous early adenocarcinomas in a long segment of Barrett’s esophagus were diagnosed. ESD was scheduled for the two lesions in the distal esophagus at the first resection and for the lesion in the middle esophagus 3 months later.\nAll lesions were resected completely through ESD with sodium hyaluronate solution without difficulties. The en bloc resected mucosa measured 27 mm for the anterior wall lesion in the distal esophagus, 26 mm for the posterior wall lesion, and 25 mm for the middle esophageal lesion. The tumors were 10, 5, and 13 mm, respectively.\nPathological diagnosis of the resected specimens showed well-differentiated adenocarcinoma in all lesions (, , ). All tumors were located within the lamina propria mucosa with tumor-free vertical and horizontal margins. Double muscularis mucosae were seen in the area beneath the malignant cells in the resected specimens from the anterior wall of the lower esophagus and the middle esophagus. No tumor cells invaded the deep muscularis mucosae. Esophageal glands were found in the submucosa of the posterior wall of the lower esophagus. Lymphovascular invasion was not seen in the three resected specimens. These findings are all characteristic of adenocarcinoma in Barrett’s esophagus.\nThe patient was followed up with endoscopy with laser imaging every 6 months, and a fourth lesion was detected 2 years after the final ESD procedure. This new lesion had a slightly reddish area on white light laser images and an irregular surface pattern on BLI bright images (). Histopathology revealed well-differentiated adenocarcinoma. However, this lesion was not resected because the patient’s cardiac function had worsened. |
Following a coronary bypass surgery with mammary graft to left anterior descending coronary artery and venous graft to posterolateral ventricular branch four years previously, a 61-year-old diabetic hypertensive man presented with acute lateral wall myocardial infarction. Due to remote access to a cardiac catheterization laboratory for a primary angioplasty, he was managed with thrombolysis initially. After 2 weeks, he presented with New York Heart Association (NYHA) Class II dyspnoea to our centre. Clinical examination showed sinus tachycardia at 106 per minute, normal blood pressures, left ventricular third sound, absence of murmurs, and basal lung crepitations. His electrocardiogram revealed an old inferior wall infarct and chest radiograph showed cardiomegaly with pulmonary venous congestion. There was a left ventricular free wall rupture forming a pseudoaneurysm measuring 8 × 15 cm with a 16 mm mouth of entry from the left ventricle between the two heads of the papillary muscles, identified on transthoracic echocardiography (Figure , and ). The systolic colour Doppler flows from the ventricle into the large pseudoaneurysm and diastolic flow back into the left ventricle created a to-and-fro spectral Doppler pattern and reduced ventricular efficiency. Regional strain analysis identified the scarred akinetic inferoposterior and lateral segments (Figure ).\nThree-dimensional transoesophageal echocardiogram delineated the morphology and extent of the large pseudoaneurysm, which extended posterolaterally from the left ventricle and distorted the mitral annulus (Figure ). A reconstructed three chamber long-axis view showed the exact location and extent of the leak (see and ). Multislice gated computed tomogram showed patent bypass grafts, non-dominant left circumflex artery with very small obtuse marginal branches, lack of need for further revascularization, and delineated the aneurysm well (Figure ). After femoral venous and arterial cannulation that assisted to establish cardiopulmonary bypass, a left posterolateral thoracotomy was performed to gain access to the pseudoaneurysm. Under hypothermic circulatory arrest, the aneurysm was opened from outside and the mouth was closed using bovine pericardial buttressed patch successfully. The symptoms of dyspnoea improved significantly and tachycardia subsided on 1-month follow-up with normal clinical findings. Echocardiography confirmed normal ejection fraction and complete exclusion of the pseudoaneurysm. |
A 55-year-old woman presented with dull aching pain on the right side of her abdomen. Abdominal examination revealed an ill-defined lump in the right lumbar area extending to the hypogastrium and iliac fossa. Ultrasound revealed a right-sided renal mass with an empty left renal fossa with an ectopic kidney at the L4 level. Dedicated triphasic CECT of the abdomen was performed for preoperative surgical planning. The CT revealed crossed fused renal ectopia on the right side (). The right renal moiety was lying opposite the L1-L4 vertebrae. The left moiety was seen lying horizontally opposite the L4-L5 vertebrae with its pelvis lying anteriorly and forming an L-shaped crossed fused ectopia. A 15-cm lobulated, minimally enhancing mass lesion was noticed to arise from the anteromedial aspect of the orthotopic kidney in the lower and interpolar region. The mass lesion was infiltrating the renal sinus fat and the upper pole region of the left moiety with ill-defined fat planes with the second part of the duodenum and liver.\nThe arterial phase of the CECT revealed three renal arteries arising from the abdominal aorta. The proximal renal artery was seen arising at the L1 level at the 9 o'clock position and was supplying the right renal moiety along with the mass lesion. The mid renal artery was arising at the upper border of the L3 level at the 10 o'clock position and was supplying the mass lesion along with the ectopic moiety. The distal renal artery originated at the mid L3 level and was supplying the ectopic moiety. Two renal veins were at the L1-L2 level draining the orthotopic moiety along with the mass lesion. Another renal vein at the lower border of the L3 level was draining the mass lesion and the left renal moiety into the inferior vena cava. The left moiety was also drained by another renal vein into the common iliac vein at the L4 level. Both renal moieties showed normal contrast excretion. The right ureter was seen coursing along the medial margin of the mass lesion, creating a plane between the mass and the left renal moiety. The left ureter was crossing the midline and was draining into the left vesicoureteric junction. No significant regional lymphadenopathy was present. The patient's serum creatinine was 1.01 mg/dL.\nAfter the preanesthetic checkup, the patient underwent robot-assisted radical heminephrectomy on the right side under general anesthesia (). The patient was placed in the left lateral decubitus position and was well secured with adequate pressure padding. Pneumoperitoneum was created by using a Veress needle through an umbilical incision. A 12-mm trocar was inserted at the umbilicus and a 30-degree scope was introduced. Two 8-mm robotic working ports were placed at the lateral border of the rectus, one between the xiphoid process and the umbilicus and the other between the umbilicus and the pubic symphysis. Another 12-mm assistant port was placed in a midline supraumbilical location. Docking of the robot was done. The right colon was mobilized medially. By use of blunt and sharp dissection, the renal mass was mobilized at the superior pole followed by mobilization at the inferior pole with identification of the right ureter between the renal mass and the ectopic kidney where it was clipped and divided. On the basis of the preoperative cross-sectional imaging, tumor-feeding vessels were clipped and divided. The lower limit of the tumor was meticulously dissected off from the ectopic moiety with preservation of its vasculature. The parenchyma at the isthmus was quite thin, and bleeding was controlled by electrocautery. The collecting system was not opened. Thus, no formal repair of the thin raw area was needed. In an attempt to avoid iatrogenic injury to the mesenteric vessels and blood vessels supplying the healthy ectopic moiety, dissection was kept close to the tumor. The large size of the tumor was impairing vision and light focusing. While dissecting superiorly and posteriorly, the plane of dissection went through the upper polar normal parenchyma of the orthotopic kidney, which was supplied by a separate vascular twig, which kept on oozing until the kidney was completely mobilized. Identification, clipping, and division of this vascular pedicle achieved hemostasis. The large tumor size hindered its retrieval with an indigenous specimen delivery bag. For specimen delivery and hemostasis, an anterior subcoastal incision was made. A 22-Fr abdominal drain was placed. Wounds were sutured and the patient was shifted to recovery after extubation.\nThe total operating time was 120 minutes (16 minutes for port placement and 2 minutes for docking). Blood loss was 600 mL. Gross examination of the specimen revealed an 18 cm×12 cm mass arising from the right kidney with areas of hemorrhage and necrosis. The patient was ambulatory by the second postoperative day. Final histopathology revealed chromophobe renal cell carcinoma with negative surgical resection margins. |
A 68-year-old female with PD sustained a right closed (OTA type A3) distal humerus fracture in her dominant arm. She takes levodopa-carbidopa 150 mg po twice daily for her PD. The patient was treated with a total elbow arthroplasty and an ulnar nerve anterior subcutaneous transposition (). Postoperatively, she noticed swelling and gradual increasing stiffness and loss of range of motion (ROM). Her activities of daily living were affected as she was unable to feed herself or adequately perform self hygiene. HO development was noticed radiographically at 6 weeks (Figures and ). ROM at that time was limited to 30° extension to 100° flexion. Initial treatment included physiotherapy as well as dynamic splints.\nThis patient clinically and radiographically was followed until cortication and maturation of the HO with no change in HO size were noted on consecutive radiographs 6 weeks apart (Figures and ). At this point, her ROM had decreased to 45° extension to 90° flexion despite splinting and physiotherapy, indicating a class IIa lesion by the Hastings classification. CT imaging was then performed to plan for operative excision of the ectopic bone ().\nAn open excision of HO and soft tissue releases () was conducted. A lateral humeral column approach was used with the Kaplan interval (between the extensor digitorum communis and extensor carpi radialis longus) anteriorly and the posterior part of the Kocher interval (between the anconeus and extensor carpi ulnaris). Careful dissection and reflection allowed anterior and posterior access. Extensive bone was removed but the components were secure and not revised. The ulnar nerve was not explored. Intraoperative tranexamic acid was applied locally into the wound to decrease bleeding from the bony surfaces. A drain was inserted during closure to prevent hematoma formation and was discontinued within 48 hours postoperatively.\nWithin 24 hours of the HO excision, the patient received a single fraction of 800 cGy parallel opposed pair beam radiation. Continuous passive motion machine therapy was initiated on the first postoperative day; however, the patient was unable to tolerate the machine due to her PD and it was discontinued. The patient was started on immediate active physiotherapy and dynamic splinting, as well as oral NSAID for 1 month. At 2 weeks follow-up, her ROM was 20° extension to 130° flexion; by 3 months, it was 15° to 140°. At her latest follow-up, 10 months after the excision and radiation treatment, the patient did not have any residual pain and had a ROM from 10° extension to 140° flexion. Her wound was well-healed, and there was no significant recurrence of the HO on follow-up radiography (). |
We present a case of an 8-year-old boy with involuntary movements of his limbs and trunk. He had hypoxic encephalopathy because of cardiopulmonary arrest after choking on food at the age of 2. He lost consciousness for 18 min owing to the cardiopulmonary arrest and was treated with hypothermia and steroid pulse therapies at our hospital for 3 months. He was then transferred to another hospital to undergo rehabilitation and returned home after 5.5 months. He underwent outpatient rehabilitation at our hospital since the age of 6. Results of the auditory reception subtest of the Illinois Test of Psycholinguistic Abilities indicated that he had an intellectual level of 5 years and 8 months; that is, he was normal in intellectual level. Although he had severe dysarthria, he was able to communicate using gestures or a communication board. Magnetic resonance (MR) imaging of his brain at the age of 6 showed a mild degree of cerebral atrophy with no clear BG lesion (). Fiber tractography using diffusion tensor imaging revealed no serious damage to the corticospinal tracts on either side (). His family history, perinatal period, developmental history, and past medical history were all normal. In his clinical assessments, manual muscle testing scores of the upper extremities, trunk, and lower extremities were 4/5, 2/5, and 2/5, respectively, with no side-to-side differences. Although his muscle tone and somatosensory function were normal, he was unable to maintain his posture or stabilize his extremities because of involuntary movement that involved the whole body. In addition, his involuntary movement was exacerbated before he initiated a voluntary movement. In daily life, he required a trunk belt to sit on a chair, and he could not stand without the support for his weight with both hands.\nThe patient was instructed to remain standing with his plastic ankle-foot orthosis for 10 s, leaning forward on a desk on his forearms. Movement of his head during standing was measured using an accelerometer attached to the glabella for 10 s at three time points: before, during, and after tDCS. Acceleration data were recorded at a sampling rate of 1 kHz using an analog-to-digital converter (Power Lab 16/35; AD Instruments, Aichi, Japan) () and were analyzed using specialized software (Lab Chart 7; AD Instruments, Aichi, Japan) to calculate and sum the power spectra within 1–5 Hz for each axis on three dimensions (x-, y-, and z-axis) (). We compared the ratios of power spectrum data (% power) during and after tDCS and divided this by the data prior to tDCS.\nFor the effective stimulation of tDCS, before this experiment we had already administered both anodal and cathodal stimulation to SMA and M1 of the patient and selected the cathodal stimulation to SMA from the other stimulations. Cathodal or sham tDCS was transcranially delivered to SMA at 1 mA for 10 min using a DC stimulator (neuroConn GmbH, Ilmenau, Germany) with 7 × 5 cm electrodes. Each stimulation session was conducted for 3 days, at least 2 days apart. The cathodal electrode was placed over SMA, identified as 2 cm anterior to Cz in the International 10/20 EEG System, and the anodal electrode was placed over the left supraorbital region. In the stimulation period, the participant started the task 2 min after stimulation began. During stimulation, he was asked to keep standing for 10 s at three time points without interruption.\nThe safety of tDCS was assessed by interviewing the patient and his mother, as well as through the examiner's observations. The patient was monitored and frequently asked whether he experienced pain or discomfort during and after tDCS. Before and after tDCS sessions, the experimenter carefully checked the skin where the electrodes were placed. The study protocol was approved by the Ethics Committee of Tohoku University Graduate School of Medicine. The patient and his parent gave informed written consent.\nPercent power data were analyzed via two-way analysis of variance (ANOVA) with the factors “stimulation” (cathodal stimulation and sham stimulation) and “time” (before, during, and after tDCS). In post hoc analysis, multiple comparison was performed using the Bonferroni correction. The level of significance was set at p < 0.05. All analyses were performed using SPSS for Windows (version 20.0; IBM, Armonk, NY, USA).\nshowed results of % power data. The two-way ANOVA showed main effects of “stimulation” (F = 7.3, p < 0.05) and “time” (F = 4.2, p < 0.05). There was a statistically significant interaction of “stimulation” × “time” (F = 4.8, p < 0.05). Specifically, the % power during tDCS was significantly reduced compared with that during sham stimulation. The % power during tDCS was also significantly reduced, compared with that before tDCS but not with that during sham stimulation. Post hoc analysis for the factor “stimulation” showed that % power during tDCS was decreased, compared with that during sham stimulation. Post hoc analysis for the factor “time” showed that % power during tDCS was significantly decreased, compared with that before tDCS. The study sessions were completed with no adverse effects. |
A 54-year-old woman presented with a painless, gross hematuria lasting three months. The patient had undergone a hysterectomy 24 years previously because of the uterine cancer. The patient had frequency, residual urine sensation, nocturia and edema of the left leg. On vaginal sonography, there was a 3.8×4.3 cm sized solid mass in the urethra which bulged to the wall of the urinary bladder (). The upper part of the lumen of the urinary bladder was clear. The computed tomographic urography revealed a 4-cm low attenuated mass with a cystic portion adjacent to the vaginal stump (). Many metastatic lymph nodes of 0.5-3 cm in size were present in both the paraaortic space and the pelvic cavity. Based on urine cytology findings, the patient diagnosed with adenocarcinoma. On the surgical field, the patient had a lobulated tumor mass of about 3 cm in diameter in the urethra and had the clear urinary bladder.\nThe voided urine cytology revealed moderate cellularity with small clusters of globular or papillary cells in the inflammatory background (). The necrotic debris and inflammatory cells were present within some clusters of tumor cells (). These tumor cells were enlarged and had abundant clear or granular cytoplasms with cytoplasmic clear vacuoles. Some clusters showed hobnail features (). The nucleus showed granular or vesicular chromatin with prominent nucleoli. It was enlarged and had a round shape with folded, irregular nuclear membrane. It showed a moderate nuclear pleomorphism (). Small globular hyaline bodies were present in clusters of tumor cells (). Benign urothelial cells were rarely observed.\nA histological section showed a tubulo-papillary pattern with solid portions (). The cells had an abundant clear or granular cytoplasm. The cells were cuboidal or columnar and hobnail cells were often present. The nucleus was vesicular with prominent nucleoli. Nuclear pleomorphism ranged from moderate to severe (). Several eosinophilic, round hyaline bodies were scattered in the tumor (). These histological findings were identical to those of the CCA of the female genital tract. The tumor cells were positive for cytokeratin (CK) 7 and P504S but negative for CK 20, p63 and prostatic-specific antigen (PSA) (). |
A 16-year-old black African girl was admitted to the emergency room of the Parakou University Hospital Center for altered consciousness and tumor of the left breast. The tumor had been progressing for approximately 6 months before admission, according to her parents. Our patient did not smoke tobacco or drink alcohol. Her medical, surgical, and psychosocial history was unremarkable. She had normal pubertal development. Her parents are of low socio-economic status. The onset of symptoms was marked by the appearance of a small nodule in her left breast, and an evolution marked by a rapid increase in the volume of the tumor, which motivated traditional herbal treatments of unknown nature. No notion of radiotherapy or previous chemotherapy was reported.\nOur patient was admitted in a state of apparent death. She had no blood pressure or pulse. Her temperature was 35 °C. She had an estimated Glasgow Coma Scale of 3. She was pronounced dead on clinical examination 10 minutes after admission. Blood samples for laboratory tests of hepatic and renal function and serological tests could not be taken before she died. The parents claimed to have no knowledge of breast cancer in their family. The parents claimed to have consulted two traditional healers and an herbal treatment of unknown nature was administered to the girl, but without improving her health.\nA postmortem examination of the body showed cachexia and a bulky, multinodular, exulcerated, blackish red hemorrhagic mass of the left breast, 35 cm in circumference, partially overflowing on the right hemithorax (Fig. ). A biopsy sample of the tumor mass was carried out postmortem. Histopathological examination after staining with hematoxylin and eosin showed tumor proliferation made up of anastomosed vascular networks. The tumor cells were spindle shaped, oval, or rounded in places and were not very cohesive. There was a marked anisokaryosis and numerous foci of tumor necrosis (Fig. ).\nThe result of a immunohistochemical analysis was positive for CD31 and factor VIII (Figs. and ). The proliferation index (Ki-67) was estimated at 30%. The expression of the tumor cells was negative for smooth muscle actin (SMA), desmin, melanin A, and S-100. A diagnosis of grade 3 angiosarcoma according to The National Federation of Centers of Cancer Control (FNCLCC) was confirmed. |
A 63-year-old female presented with pelvic abscess and rectovaginal fistula several months after undergoing a low anterior resection at another institution complicated by intra-abdominal sepsis from dehiscence of her anastomosis. This had been treated with diverting loop ileostomy and closure of her abdominal fascia with biologic mesh. Since that time she had developed purulent drainage from her vagina and rectum. She had been started on per oral amoxicillin-clavulanate. She was taken to the operating room for low anterior resection with coloanal anastomosis, small bowel resection, primary repair of parastomal hernia with mesh, and drain placement. The patient was on broad spectrum antibiotics at the time of surgery. Specific preoperative antibiotics were not given. On postoperative day three she developed nausea and vomiting. On postoperative day four she developed hypotension. A nasogastric tube was placed with return of bilious output. She became hypotensive and was transferred to the surgical intensive care unit (SICU) for fluid resuscitation. She was afebrile, with moderate abdominal tenderness, reduced urine output, and elevated creatinine. The decision was made to proceed to the operating room. At the time of reoperation her anastomoses were intact, but the abdomen could not be closed. An ABThera Open Abdomen Negative Pressure Wound Therapy System was placed and she was returned to the SICU where she was started on intravenous metronidazole and oral vancomycin due to concern for small bowel C. difficile enteritis after pseudomembranes were noted on the ileostomy. A C. difficile toxin assay was positive at that time. Red rubber catheters were placed in both sides of her ileostomy and her rectum taking care not to disrupt her coloanal anastomosis. 500 mg of vancomycin was placed in each of these tubes and down her nasogastric tube every 6 hours.\nHer hospital course was further complicated by ventricular tachycardia, heart failure intolerance of enteral feeds, cholestatic jaundice, need for reoperation for placement of absorbable mesh for closure of the abdominal cavity, leukocytosis, need for total parenteral nutrition (TPN), tracheostomy, development of enterocutaneous fistula, multiple intraabdominal abscesses, a repeat episode of C. difficile enteritis, atrial fibrillation with rapid ventricular rate, and a urinary tract infection. She remained in the hospital for two months.\nAt the time of discharge, her tracheostomy had been removed; she was tolerating snacks of thickened liquids, had a nasoenteric feeding tube in place, and was receiving TPN and intravenous metronidazole. A wound manager was being used to control her fistula output. The fistula subsequently closed, and she remains healthy but with a large ventral hernia. |
A 73-year-old woman presented with intermittent abdominal pain and weight loss of 15 kg for the past 2 years. Her pain was frequently accompanied by nausea and/or diarrhea and tended to be aggravated by food intake. Her past medical and familial histories were unremarkable. On examination, vital signs were stable but she appeared a little chronically ill. Her abdomen was soft with no palpable mass; bowel sounds were somewhat increased and mild tenderness was noted in the right lower quadrant (RLQ). Initial laboratory blood tests were normal.\nColonoscopy revealed an erythematous polypoid mass at the proximal ascending colon and it seemed to have a huge stalk thicker than the mass itself (). With air being inflated during colonoscopy, it was not seen in the colonic lumen and abruptly disappeared. As the endoscope was advanced through the ICV, there was a polypoid mass in the terminal ileum and multiple tiny hemorrhagic spots on the ileal mucosa; the mass intussuscepted into the colon through the ICV had reverted back to its original site in the terminal ileum (). The thick stalk must have been an intussusceptum of the terminal ileum with the mass as a lead point. In its original position in the ileum, a short stalk was identified on the base of the polypoid mass. Additionally, abdominal computed tomography (CT) scan revealed a well-demarcated pedunculated subepithelial mass with fat attenuation indicating a lipoma of the terminal ileum ().\nThis ileal lipoma was thought to have been responsible for her abdominal pain and weight loss on account of repeated ileocolic intussusceptions causing intermittent intestinal obstructions. Because the patient strongly refused surgical treatment, we planned to endoscopically remove her ileal lipoma considering potential sequelae of ileal resection. In order to perform endoscopic procedure in the spacious colonic lumen, we tried to retract the mass into the colonic lumen through the ICV but unfortunately failed. Therefore, to keep a clear visual field within the narrow lumen of the ileum, a cap-assisted colonoscopy technique was used. After submucosal injection into the base of the mass, a snare polypectomy was performed within the ileum (). And then, three clips were placed to prevent delayed hemorrhage or perforation (). There were no immediate complications after the polypectomy. The histopathology report confirmed a 2.6×2.7 cm subepithelial lipoma. At 2 days of fasting after the procedure, oral intake was started and the patient complained of no symptoms related to food intake. Since resection of the lipoma, her body weight has gradually restored. |
Case 14 reports a 60-year-old male who showed objective response (Figure b). After pancreatoduodenectomy, gemcitabine treatment started in October 2008 and liver metastasis was found 3 months later. Followed by TS-1 chemotherapy, we found that metastatic lesions in the liver progressed after the condition of SD during 3 cycles of TS-1 treatment. After 1 cycle of the peptide vaccine, one target lesion of liver metastases located at S8 was shrunken. This lesion kept shrinking until September 2009, and became hardly detectable by CT scan. Similarly, a metastatic lesion in the lymph node was significantly shrunken until September 2009. However, the other target lesion (S4) in the liver showed no response to the vaccine treatment and the tumor progression was promoted after 2 cycles. Finally, the patient died at 220 days after the start of the vaccination.\nIn case 24, a 74-year-old male also showed objective response (Figure c). After distal pancreatectomy in August 2007, adjuvant chemotherapy utilizing gemcitabine was performed for 6 months and then switched to TS-1 because of the side effect. Bone metastasis was found in the xiphoid process by CT scan in April 2009. Radiation therapy was performed to the xiphoid process in May 2009, but the tumor did not respond well. The patient was enrolled into the peptide vaccine trial in July 2009 after one month of cooling off period. Bone metastasis started to shrink after one cycle of the peptide vaccine treatment. The precordial pain was rapidly diminished and well controlled without opioid treatment. After the 5th shot of the peptide, Grade 3 interstitial pneumonia was observed and the treatment was discontinued. The patient was hospitalized in one week of treatment without any steroid therapy and then well recovered. Even without the vaccination, pain was well controlled and tumor markers kept decreasing for the next two months. After the re-progression of the disease, gemcitabine was administered and no clinical effect was observed. Since the patient desired to receive the peptide vaccine again, we obtained an approval of the re-entry of this case from the Ethical committee. The vaccine treatment was restarted with careful monitoring, while neither adverse events nor clinical effect was observed in this second round of drug administration. His overall survival period from the first day of administration was 495 days.\nThe median overall survival time of 31 patients was 142 days, and the progression free survival period was 56 days (Figures a and b). In comparison with the control group without the vaccine treatment, who are the patients visited Chiba Tokushukai Hospital in the period between January 2007 and January 2009 (MST: 83 days), overall survival of the patients with the KIF20A-peptide vaccination was statistically significant (p = 0.0468, MST: 142 vs. 83 days) (Figure c). Moreover, MST of the patients who received BSC was 63 days. Compared to the control group in multi-center, Overall Survival of the vaccinated patients was significantly improved (p = 0.0020, MST: 142 vs. 63 days) (Figure c). Taken together, we concluded that the cancer vaccination utilizing KIF20A-derived peptide was significantly effective as immunotherapy against advanced pancreatic cancer. |
A 58-year-old Iranian postmenopausal woman referred to the hospital because of a abdominal wall mass and hernia. She had a history of three cesarean sections with a midline abdominal incision. Her last cesarean section was performed 20 years earlier. Physical examination demonstrated a partially mobile and large mass in the anterior abdominal wall in the midline cesarean scar, extending bilaterally to the midclavicular line. The upper margin of the mass was adjacent to her umbilicus, where there was a large umbilical hernia above the mass (Fig. ). Pelvic ultrasound showed an irregular 12 × 8 × 8 cm mass in the abdominal wall anterior to the rectus muscle involving subcutaneous tissue, a large umbilical hernia containing omentum, and a well-defined left solid adnexal mass (4.1 × 3.7 × 5.1 cm in diameter). An abdominopelvic CT scan confirmed the ultrasound findings (Fig. ). The peritoneal surface of the anterior abdominal wall was intact, and other abdominal and pelvic organs were normal. The chest X-ray was normal.\nA local incisional biopsy of the abdominal wall mass was performed before referral to our department. The histology implied clear-cell carcinoma\nThe patient was scheduled for surgery as determined by the MDT. Midline laparotomy was performed. The abdominal wall mass invaded the lower half of the rectus muscles. The mass reported in the left adnexa was indeed a large pelvic lymph node adjacent to the left external iliac artery. Uterus and ovaries had a normal appearance. The patient underwent a complete excision of the abdominal wall mass including the rectus muscles and fascia, peritoneal washing, resection of the enlarged pelvic lymph nodes, TAH, and BSO. Due to the large abdominal wall defect following the excision of the abdominal wall mass, reconstructive surgery was performed to repair the abdominal wall. The defect was covered with a synthetic mesh. The results of the permanent histology of the abdominal wall mass and the enlarged pelvic lymph node were similar, indicating clear-cell carcinoma (Fig. ) without any marginal involvement. The pathology results of ascites and surgical specimens were negative. Twenty days after the reconstructive surgery, the patient initiated chemotherapy with six cycles of paclitaxel and carboplatin every 3 weeks. An abdominopelvic CT scan was performed 6 months after the completion of the treatment and revealed normal findings. At present, the patient is alive without any recurrence (26 months after diagnosis). The basic characteristics of the patients, imaging results, surgical procedure, and adjuvant treatment are summarized in Table . |
A 56-year-old man who developed ankylosing spondylitis at the age of 33 in 1986 complained of dyspnea and back pain for 10 days before his first admission to the pulmonology department of our hospital via the emergency room in 2009. At another hospital in 2002, he had been treated with internal fixation of the thoracic and lumbar spine due to an ankylosing spondylitis-associated spine deformity. He was clinically diagnosed with smear-negative pulmonary tuberculosis in 2004 and received incomplete treatment. In 2007, he was diagnosed with reactivation of pulmonary tuberculosis and took anti-tuberculosis agents for 9 months . Medical records review showed that he was clinically diagnosed with smearnegative, culture-negative pulmonary tuberculosis based only on radiological findings and without mycobacterial confirmation. At the time of admission to out hospital, the patient was not taking drugs other than anti-tuberculous agents and nonsteroidal anti-inflammatory drugs for intermittent pain control. He has smoked half a pack of cigarettes daily for 15 years with occasional alcohol intake.\nThe patient was diagnosed with pneumonia and an exacerbation of chronic obstructive pulmonary disease at first admission. Therapy with daily intravenous antibiotics was begun following multiple blood, sputum , and urine cultures. Oral and inhaled bronchodilators were prescribed and oral prednisolone (30 mg) was maintained for 10 days. Since discharge in 2009, he has maintained medical treatments and respiratory rehabilitation. Treatment consists of oral medication, including methylxanthine, a long-acting beta 2 agonist (LABA), with inhaled therapy of a long-acting anticholinergic, inhaled corticosteroid and LABA combination.\nThe patient's initial chest radiograph, taken by the orthopedic department in 2006, showed apical linear fibrotic opacity with cystic changes similar to sequelae of pulmonary tuberculosis (). A nother chest radiograph, taken in the pulmonology department at first admission in 2009, showed patchy consolidations and ground-glass opacities in both lungs, and apical fibrosis with bullous changes suggesting stable tuberculosis and emphysema in both lungs (). Over the next 8 years, the patient's serial chest radiographs () and HRCT findings () showed bullous fibrocystic changes on both upper lobes, and the bullous cysts increased in size with disease duration. HRCT findings () revealed linear fibrocystic opacity, calcified granulomas, and traction bronchiectasis accompanying parenchymal lung destruction in both upper lungs compatible with post-tuberculosis sequelae, and pneumonic consolidations wi th ground-glass opacities in both lower lobes.\nDuring a follow-up period of 6 years from 2009 to 2014 in the pulmonology department of our hospital, the patient experienced frequent respiratory events and was hospitalized ten times. Among his respiratory events, pneumonia was the most common reason requiring a visit to the hospital, followed by acute exacerbation of chronic obstructive pulmonary disease and hemoptysis. The patient was hospitalized via the emergency room due to massive hemoptysis following superinfection of the cavitary-destroyed lung with mycetoma, and underwent bronchial artery embolization in combination with antifungal agents at the age of 60 in 2013. The pathogen was not confirmed by sputum or blood culture, but since aspergillus antigen was confirmed positive in serum, the lung lesions were thought to be associated with a fungal infection. Chest radiography showed that a fungal ball had been present in a fibrocavitary lesion of the left upper lung since 2011. The patient complained of symptoms such as worsening dyspnea and required oxygen therapy at home because of chronic respiratory failure.\nIn analyses of radiologic serial images with chest X-ray and HRCT, the progression of parenchymal lung destruction was correlated with disease duration in this patient with advanced ankylosing spondylitis. Chest X-ray () and HRCT () showed that the initial apical fibrosis in 2009 progressed to progressive fibrocystic bullous changes that involved both upper lobes with cavitations. In serial films, frequent superinfections of cavitary lung lesion with mycetoma in both upper lungs were noted. Although the patient was clinically diagnosed with smear-negative pulmonary tuberculosis and reactivation during previous treatment at another hospital, acid-fast staining and all subsequent mycobacterial sputum cultures were negative, and an interferon-γ release assay failed to detect mycobacterial infection.\nFollowing the investigation and retrospective review of medical records and radiologic findings by a pulmonologist, a rheumatologist, and a chest radiologist, the patient's progressive fibrocystic bullous lung changes were interpreted as pleuropulmonary involvement of extra-articular symptoms of advanced ankylosing spondylitis. The patient is currently managed at home with supportive care, maintenance medical treatment, and oxygen therapy. Considering the generally poor performance of patients with advanced-stage disease and the high risk of complications related to active treatments, surgical resection procedures, such as bullectomy, and anti-tumor necrosis factor α (TNF-α) agents are not being used. |
A 43 year old gentleman with no past medical history of note presented to his General Practitioner with 2 episodes of frank haematuria. An ultrasound scan was carried out which demonstrated a large mass in the right kidney. A follow-up contrast enhanced computerised tomography (CT) scan of the thorax, abdomen and pelvis was carried out which demonstrated a large right intrarenal tumour with extracapsular spread into the right adrenal gland and vascular invasion of the right renal vein and inferior vena cava (IVC). An echocardiogram demonstrated no invasion of the right atrium.\nA right radical nephrectomy with exploration of the IVC was performed jointly by a urological and cardiothoracic surgeon. The operation was performed entirely via a laparotomy using an L-shaped anterior abdominal incision to facilitate mobilisation of the suprahepatic IVC. The diaphragm was divided radially from the xiphoid down to the IVC. Cross clamping of the IVC below the right atrium allowed removal of the tumour from within it, thus no sternotomy or cardiopulmonary bypass was used. At the end of the operation the diaphragm was repaired from below. The pericardium was opened in order to facilitate removal of the tumour and a pericardial drain was placed following the procedure. The operation was without complication, however 3 days post-operatively the patient developed a pulmonary embolism (PE) and was anticoagulated with treatment dose tinzaparin. The patient recovered well and was discharged on day 10 post-operatively.\nOn the 19th post-operative day the patient presented to his local emergency department with abdominal discomfort, nausea and bilious vomiting which had lasted 24 h. On examination there was mild abdominal tenderness and epigastric fullness. His observations were stable, ECG showed normal sinus rhythm and bloods demonstrated no clear abnormality other than a C-reactive protein (CRP) of 50 and an alkaline phosphatase (ALP) of 172. Following a period of observation, he was discharged home. The working diagnosis given at this time was viral gastroenteritis.\nThe following day the patient returned to the emergency department in cardio-respiratory failure. He had a tachycardia of 120, was severely agitated and was hypoxic with oxygen saturations of 82%. He was coughing up large amounts of blood stained frothy sputum. His blood tests demonstrated rising leucocyte count of 14 and CRP of 99, which increased overnight to a leucocyte count of 17.9, CRP 117 and of note a troponin of 478. The decision was made to intubate in order to facilitate a CT pulmonary angiogram (CTPA) as a recurrent PE was the initial working diagnosis. Following sedation he was profoundly bradycardic and hypotensive, necessitating rapidly increasing pharmacological cardiovascular support, and on insertion of the endotracheal tube a large volume of pulmonary oedema fluid was suctioned. The CTPA demonstrated no PE, however there was extensive pulmonary airspace shadowing in both lower lobes consistent with acute respiratory distress syndrome (ARDS), as well as a highly abnormal appearance to the left atrium with a smooth margined filling defect extending from the posterior wall anteriorly compressing the lumen. There was a 6 cm pericardial effusion seen. Furthermore, a transthoracic echocardiogram demonstrated a huge cystic lesion in the left atrium with a thick wall containing fluid and debris. These scan results were discussed with the regional cardiothoracic unit and an emergency transfer was performed.\nThe patient was taken straight to theatre and the transoesophageal echocardiogram confirmed a large (6.68 × 7.25 cm) loculated posterior pericardial effusion which at this point was entirely compressing the left atrium. An emergency sternotomy was performed, a large posterior loculated effusion was found which was made up of old venous blood, 450 ml was drained and a pericardial drain placed. The patient’s cardiac output immediately improved and norepinephrine requirement decreased. The pleura was opened bilaterally with straw coloured fluid drained 500 ml on the left and 300 ml on the right, drains were placed bilaterally.\nHe was extubated the following day, and biochemically improved significantly over the following 4 days with his leucocyte count completely normalising. Serial chest radiographs demonstrated significant improvement with reduction in pulmonary oedema, this was mirrored by his oxygen requirements completely reducing. The patient was discharged home and outpatient follow up demonstrated an excellent recovery. |
A 43-year-old male reported to the Department of Oral Medicine and Radiology with the chief complaint of tooth pain on the right lower back tooth region. Extraoral examination revealed multiple nodules on the conjunctival surfaces of the upper and lower eyelids []. The lips were diffusely enlarged and appeared everted and bumpy on palpation []. On intraoral examination, multiple nodules were seen on the tip and lateral borders of the tongue []. Tongue appeared enlarged, and no functional abnormality was detected. Nodular masses were also seen on the buccal mucosa [] and labial mucosa []. The nodules were asymptomatic, oval-to-round shape, measuring 0.8 cm × 0.5 cm in dimension with sessile base and smooth surface. The patient gave a history of carcinoma of the thyroid, and a total thyroidectomy was done in a cancer institute 20 years back. There was no history of cancer and thyroid abnormality in his family. The patient gave a history of chronic constipation since childhood. Anterior maxillary and mandibular teeth appeared to be protruded from side profile view []. On hard tissue examination, multiple root stumps were present in all the four quadrants. Extraction and prosthetic rehabilitation was advised. Midline diastema was clearly evident in the maxillary anterior region [].\nGeneral examination failed to reveal abnormalities such as cervical lymphadenopathy, café-au-lait spots and diffuse pigmentation of neurofibroma on the trunk or extremities. Blood pressure and pulse rate were within the normal limits ruling out pheochromocytoma. A proper medical record of previous investigation and treatment could not be obtained from the patient since they were not maintained properly. At present, he was not under any treatment. Based on the clinical features and history, a clinical diagnosis of MEN2B was made. There was no history of cancer and thyroid abnormality or similar syndromes in his family. Since none of the family members were affected, this case was considered to be due to de novo mutation. Biochemical analysis of the patient was within the normal limits. Incisional biopsy was done from nodule on labial mucosa. Histopathological examination revealed bundles of disorganized and tortuous nerve fibers surrounded by a thickened perineurium which was suggestive of mucosal neuromas []. Numerous bundles of elongated cells with wavy dark nuclei were identified. The patient was further referred to the Department of Endocrinology for further management. |
A 50-year-old Caucasian female visited the regional dental office due to uncertain pain in the right mandibular region. No significant diseases were mentioned in the anamnesis. The patient was diagnosed with gingivitis, and treated with a non-steroid anti-inflammatory drug and oral rinsing with chlorhexidine. The condition of the patient did not improve significantly. When she returned a week later, she had no alleviation, but labial paresthesia on the right side of her lip. As the cause of the new symptom remained unidentified, she was referred to the oral surgery group. Initially, the paraesthesia affected only the right side of the lower lip, but by this time it had spread to the skin of the right side of her chin. The oral surgeons decided to extract the lower right wisdom tooth, but the labial paraesthesia still persisted. Since there was an extended composite restoration in the lower right first molar, and translucency around its apical region was visible, the lesion was diagnosed as periapical granuloma. The lesion on tooth 46 was not close to the mandibular canal; this can be seen on Fig. . Therefore, root canal treatment was carried out, despite the uncertain result of the percussion, palpation and sensibility test. The transparent lesion around the apex and the negative sensibility test lead to the decision of endodontic treatment. Diagnosis was made by a dentist who is not a staff member of our Department. The treatment revealed that the pulp was vital. Our oral diagnostic team examined the patient, and we diagnosed the case as cemento-osseous dysplasia based on the panoramic radiograph showing apical translucency lesions around the anterior teeth with vital pulp (Fig. ). The paraesthesia has been continually present ever since. Because the lesion around the lower right canine had already involved the cortical bone by CBCT scan (Fig. ), a root canal filling and removal of the apical lesion were performed. On the CBCT image the involvement of the entire periapical region can be seen including the lingual region and the buccal cortical (Fig. ). It became clear during the planning of the surgery that the blood vessels and nerves of tooth 43 would be injured.\nThe root canal treatment was carried out on the lower right canine in a single-visit treatment using local anaesthaesia. We explored the entire root canal length using a size #15 hand instrument. The working length was determined using an apex locator, (Woodpex III, Guilin,China), then the length was also confirmed with radiographic imaging. Following the length determination, the root canal was shaped using Wave One (Densply Maillefer, York, USA) rotary instrument. The root canal was obturated using guttapercha and AH Plus sealer (Dentsply DeTrey GmbH, Konstanz, Germany) with lateral condensation technique. Glass ionomer cement (Fuji IX GP, GC Co., Tokyo, Japan) was then applied to seal off the access cavity, while the permanent restoration was done.\nWe prepared an intraoral mucoperiostal flap using an L-shaped incision and the surgery was carried out by using a surgical microscope (Aspheron, Schmidt and Bender Hungaria, Budapest, Hungary).\nWe opened up the buccal cortical bone using a surgical bur, thereafter we removed the lesion surrounding the apex of the root. The root apex was resected and a retrograde root canal filling was placed using mineral trioxid aggregate (MTA+, Cerkamed, Stalowa Wola, Poland).\nThe area of the lesion was augmented using gentamicin impregnated BoneAlbumin (OrthoSera Dental Zrt., Gyor, Hungary). We seeked to lower the chance of osteomyelitis occuring with the use of gentamicin. Sutures were then carefully placed to achieve tensionfree closure of the flap for optimal healing.\nThe 19-year-old Caucasian woman is the daughter of the first patient. CBCT showed a radiolucent lesion around the root of the lower right incisor (Fig. ). She is presently asymptomatic after a one-year follow-up, but she occasionally felt tension and moderate pain in the right side of the mandibular region eradiating to her ear, approximately 2 years earlier. The symptoms had no dental background, and ceased gradually.\nPulp tests and periapical pathology diagnoses were made by authors on teeth 33,32,41,43 with the use of percussion, palpation and sensibility test.\nPanoramic radiograph and CBCT scans of the first patient showed radiolucent lesions located in the periapical bone, specifically on lower incisors and canines – in the premandibular, and in the right molar region of the mandible with radiopaque parts showing the lesions inside (Figs. and ). The affected teeth were asymptomatic, CBCT proved the presence of apical pathology. The buccal cortical involvement was discovered with the aid of CBCT, which also affected the treatment plan because 2D imaging methods provide no information on bucco-lingual dimension. Prior to the surgical intervention the use of CBCT was motivated by the fact that only 3D imaging could ascertain the precise shape, location and involvement of surrounding anatomical landmarks of the lesion.\nFigure shows the postoperative condition (Fig. ). At the 6-months periapical follow-up, the bone healing is being processed (Fig. ). 18 months later it can be seen on the CT image that the buccal cortical bone surrounding the root of tooth 43 has regenerated (Fig. ). In the case of the second patient, there were no notable changes in the radiographic status 1 year later, and she has no complaint at present.\nThe removed tissue pieces contained both connective tissue and calcified areas. In the calcified area, beside the irregular trabecular – lamellar bony formations, oval and globular cementum-like structures were present. In the non-calcified area connective tissue was found and connective tissue filled out the centre of another bony sample as well. No elements or remnants of a capsule were visible. The histological diagnosis was cemento-osseous dysplasia (Fig. ). |
A 56 year old Indian male patient was admitted for post-traumatic avascular necrosis of the head of the femur leading to degenerative arthritis of his left hip joint (). He was a known case of hypertension on medication and a chronic smoker as well as an alcoholic. A cemented total hip arthroplasty was performed through the Southern Moors (posterior) approach (). Immediately following the surgery, his pulses were felt but had been feeble. However, three hours post-operatively the resident doctor noticed that the patient’s left lower limb had become cold and the dorsalispedis artery, popliteal artery, and femoral artery pulsations were absent on the left side. There were no ankle movements. Since the patient was on epidural anesthesia, the patient did not complain of any pain. An emergency Doppler scan suggested narrowing of the left iliac and left common femoral vessels with monophasic flow. A C T angiogram revealed complete occlusion of the left common femoral artery ().\nWith an urgent opinion from cardiothoracic and vascular surgery, the left femoral artery was exposed and a thrombus was noticed (). Angioplasty was carried out using Forgarty’s catheter and the vessel was recanalized (). On passing the catheter proximally, one layer of thickened intima was removed followed by clots. Good blood flow was established on removal. When the catheter was passed distally, not many clots were present. After removal of the occluding thrombus measuring 2.5 cm in length and intimal plaque of one cm length confirmed by histopathological examination (), the artery was closed using 6-0 prolene sutures (). Peripheral pulses were felt. After 12 hours, there was active movement of the limb. A post-operative Doppler scan showed good flow of blood within the affected vessel (). Though a postoperative CT angiogram would have further affirmed the successful findings, it was not performed due to the financial constraints of the patient. The patient was started on anticoagulants for a course of three weeks. Since the remainder of the post-operative period was uneventful the patient was discharged 17 days later and advised to return for a follow-up and repeat Doppler scan after one month. He was also strictly advised to stop smoking. |
A 14-year-old boy, who was initially examined for continuous coughing, was referred to our hospital owing to an anterior mediastinal mass identified on chest X-ray (). Systematic examination revealed a 20 × 10 cm sized mass at the right anterior mediastinum that involved the right pulmonary vein and elevated levels of alpha fetoprotein (AFP), a tumor marker, at 3825 ng/ml. Needle aspiration was performed, but only necrotic tissue could be collected. We did not analyze the karyotype since the symptoms suggesting the Klinefelter syndrome were not observed. Therefore, a clinical diagnosis of malignant teratoma was made, and three courses of cisplatin-based chemotherapy were administered because AFP levels continuously increased. After chemotherapy, AFP levels decreased, although the size of the tumor did not change, as evident in a computed tomography (CT) scan. Next, tumorectomy and total right lung extraction were performed. The pathological diagnosis of the extracted tumor was malignant teratoma with areas of yolk sac tumor (). No cancer cells were found at the edges of the area where the tumor was removed. Two courses of cisplatin-based postoperative chemotherapy were administered until AFP levels were normal.\nAfter 3 months of follow-up, the patient experienced pain in his right hip joint while AFP was still normal. The 99mTc-methylene diphosphonate bone scan showed increased tracer uptake at the left forehead and right hip joint (). Head and pelvic MRI also revealed signs of metastasis at the left forehead and right hip joint. Abdominal CT showed a metastatic region in the liver. Bone biopsy was performed at the left forehead. The pathological diagnosis was metastatic malignant melanoma originating from an immature teratoma of mediastinum (). Large heteromorphic cells with melanin were found in the original mediastinal malignant teratoma by retrospective re-examination. This suggested that a section of the malignant melanoma in the original malignant teratoma, which was composed of various components, metastasized. Because of the very fast disease progression, after consultation with the family, aggressive treatment was discontinued, and palliative therapy was provided. He died 15 months after diagnosis of the original malignant teratoma. |
An 88-year-old woman visited our clinic due to a small mass on her right upper eyelid that had developed 6 months prior to her consultation at the department of ophthalmology. The mass was previously proven to be a sebaceous carcinoma by an incisional biopsy done at the department of dermatology. The tumor was located in the lateral region of the right upper eyelid margin, with a size of approximately 5 mm. It was a painless, yellow tumor with no other distinctive characteristics (). No regional lymph node involvement or distant metastasis was observed on ultrasonography or magnetic resonance imaging.\nAs the tumor was on the border of the upper eyelid, we planned a full-thickness wide surgical resection with a safety margin of 5 mm. The full-thickness defect after tumor excision was covered by a combination of a nasal septal chondromucosal graft and a temporal forehead flap.\nSurgery was performed under general anesthesia (). The excision of the tumor with a 5-mm safety margin resulted in a 2-cm-diameter full-thickness defect. The defect was larger than one-third of the total length of the upper eyelid. The levator aponeurosis was exposed on the upper margin, the tarsal plate on the medial margin, and the lateral orbital rim on the lateral margin.\nSince chondromucosal composite grafts should be harvested with cartilage and mucosa attached, local anesthesia (1:100,000 epinephrine mixed with 2% lidocaine solution) was administered only between the nasal mucosa and septal cartilage of the opposite side to prevent inappropriate hydrodissection. A 2× 3 cm graft was then obtained. The septal donor site was covered by a mucosal graft harvested from the buccal mucosa and compressed using a silicone sheet and nasal tampon to prevent septal hematoma. The buccal mucosa donor site was primarily closed.\nThe harvested graft was trimmed into the shape of the eyelid defect, and the cartilage was sutured with the levator aponeurosis on the upper margin, the orbital rim on the lateral margin, and the tarsal plate on the medial margin.\nA Fricke flap, or a forehead transposition flap, was designed just above the eyebrow, including a line of eyebrow that would replace the eyelash excised during wide resection. The width of the flap was set to fit that of the defect, and the length was determined such that the width-to-length ratio did not exceed 1:4 to ensure an appropriate blood supply. The subcutaneous flap was then raised carefully to prevent it from becoming too thick. A tunnel was made between the flap origin and defect, and the flap was de-epithelialized, except for the distal portion, to cover the defect. The flap was then passed through the tunnel, and the distal portion of the flap was inserted on the chondromucosal graft. The flap was sutured along the margin of the eyelid defect. The flap donor site was closed primarily, and tarsorrhaphy was performed.\nA light compressive dressing and tarsorrhaphy were maintained for a few days, and the stitches were removed after 8 days. Early complications such as hematoma, graft loss, flap necrosis, and nasal septal perforation were not observed.\nOne year postoperatively, no local recurrence or metastasis was observed on ultrasonography and magnetic resonance imaging, and the wound healed completely without any late complications. Lagophthalmos, scleral show, and asymmetry on both upper eyelids during the opening or closing of the eyes were not observed. The appearance of the eyelashes was satisfactory. There was no trichiasis or alopecia, and the lining of the new eyelash appeared natural in accordance with the adjacent eyelash. The scars of the eyelid and flap donor site were both acceptable. The overall results were satisfactory, and the patient was pleased (). |
A 30 year old male presented to the outpatient department in January 2010 with complaints of increasing pain and stiffness in the left wrist for the last 2 years following a fall on outstretched hand. He took treatment in form of massage for two and half months and no radiological investigations were done for his injury. He returned to stitching clothes, his profession, after two and a half months. His wrist pain increased progressively along with restriction of movements to such an extent that he had to discontinue his job. On examination his left wrist (non-dominant) showed gross restriction of motion with just 10 degrees of dorsiflexion and 15 degrees of palmar flexion. There was mild tenderness on radial side of wrist, which was more marked in the anatomical snuffbox. Radiographs showed a nonunion of scaphoid with avascular necrosis of distal pole with no evidence of radiocarpal arthritis []. The lateral intrascaphoid angle as observed in the lateral view was 60° indicating a mild humpback deformity; however, luno-capitate angle was 0°, indicating absence of carpal instability. There was no loss of carpal height. The carpal height ratio was 50% (45-60%), ruling out a carpal collapse. Computed tomographic (CT) scan of right wrist showed the distinct fracture line in the scaphoid with cystic changes in the adjacent region of bony fragments and a sclerotic distal fragment [].\nAn open reduction and internal fixation of the nonunion with k-wires supplemented with a pronator quadratus muscle pedicle bone graft was planned. The scaphoid was exposed through a standard palmar approach after extending the incision beyond the distal palmer crease by 2 cm []. Nonunion was exposed and fractured surfaces cleared of fibrous tissue. The distal fragment was retrieved, which was approximately 1 × 0.5 cm in size. No bleeding could be observed in the distal fragment on curettage of its fractured surface, confirming avascular distal fragment as preoperative MRI was not done in this patient for assessing avascularity. The nonunion was reduced and a 2.0 mm k-wire was passed across the fracture site from distal to proximal fragment, under fluoroscopic guidance. We had planned fixation with Herbert's screw, however, because of the fear of crushing the small sized distal fragment, we preferred k-wire. The pronator quadratus muscle pedicle bone graft was harvested and transposed to the nonunion site. A second k-wire was now introduced, which held the graft in position and supplemented the fixation of the fracture []. The wound was closed in layers with a suction drain in place. The wrist was immobilized in a below elbow plaster of paris cast in neutral flexion The postoperative radiographs showed a reduction in the lateral intrascaphoid angle to 46° from a preoperative 60° []. The pop cast was continued for 12 weeks followed by gradual mobilization and range of motion exercises. At 6 months, sound union at the fracture site was observed radiographically and was confirmed by CT scan [Figure and ]. He returned to (stitching clothes) job at 4 months and resumed his part time farming activities after k-wire removal at 6 months, postoperatively. At 2 years followup, he was able to perform all activities of daily living and had attained his pre-injury functional level. He still experiences mild pain after heavy work and has restriction of terminal 10° of palmar flexion []. His Disabilities of the arm, Shoulder and Hand (DASH) Score was 4.25, which is consistent with good hand function. The magnetic resonance imaging (MRI) scan of the left wrist showed evidence of revascularization 2 years post-injury suggesting successful incorporation of pronator quadratus muscle pedicle bone graft []. |
A 57-year-old female was admitted at our facility because of 2 days history of hearing loss in both ears. She experienced tinnitus in both ears 2 weeks prior to the hearing loss. She has undergone multiple surgical treatments for urinary calculi. She is also a known hypertensive for the past 5 years. Electrocardiogram (ECG) also detected coronary heart disease 4-years after establishing the hypertension. She was admitted at a local hospital a month prior to the above symptomatology due to severe urinary tract infection (UTI) with thrombocytopenia. She was treated with intravenous (IV) antibiotics for 5 days and discharged home. General cranial nerve examination did not yield much. However, Webber and Renner tests revealed severe left side hearing loss and a mixed hearing loss in the right ear. General physical examination was unremarkable. Routine chest X-ray was normal. Also, routine laboratory investigation at the current presentation was grossly normal.\nCT scan of the urinary system revealed a calculus at the right ureter (Fig. A). The stone was located at 2nd and 3rd lumbar vertebral body planes. Also, the whole right ureter was slightly dilated. She was given hyperbaric oxygen and intramuscular dexamethasone at a maximum daily dose of 16 mg for 4 days and reduced to a maximum daily dose of 8 mg for another 3 days in attempts to resolve the sudden deafness. She also complained of backache, chills, and fever. Thus, a diagnosis of UTI secondary to ureteric calculi was made and she was put on IV Ceftriaxone Sodium 2 g daily for 5 days.\nHer condition suddenly aggravated 5 days after establishing the UTI although she was on antibiotics. The patient went into hypertensive encephalopathy. She was transferred to the intensive care unit (ICU) immediately and an emergency chest CT showed left lung pneumonia (Fig. B), while a repeated CT scan of urinary system showed a descend of the right ureter stone with hydronephrosis (Fig. C). Blood as well as urine cultures and sensitivities revealed staphylococcus hemolyticus which was sensitive to piperacillin, tazobactam, and levofloxacin. Therefore, she was put on the above antibiotic to treat both UTI and chest infection. Two days later, she developed seizure so we performed EEG at the bedside which revealed abnormal occipital lobe apical waves consistent with acute seizure disorder (Fig. A).\nHead CT scan and CT angiography (CTA) showed no abnormalities (Fig. B and C) while MRI showed abnormal signals at both temporo-parieto-occipital (TPO) cortices, the subcortical area, as well as the left hippocampus (Fig. D–F) which was consistent with the diagnosis of posterior leukoencephalopathy syndrome. Magnetic resonance angiography (MRA) also showed no vascular abnormality (Fig. G). Also, cerebrospinal fluid examinations were grossly at normal ranges. She was put on dexamethasone injection 16 mg 6 hourly for 3 days. The posterior leukoencephalopathy syndrome improved remarkably.\nFive days later, pelvic CT scan revealed a total descend of the right ureteric stone into the bladder and a resolution of the dilatation of right ureter (Fig. A). Also, the patient's temperature returned to normal range. Furthermore, a repeated MRI of the head revealed disappearance of the abnormal signals at both TPO cortices, the subcortical area, as well as the left hippocampus (Fig. B–D). Thus, a definitive diagnosis of RPLS secondary to obstructed right ureteric calculus with superimposed UTI and left lung pneumonia was made.\nThe patient's hearing loss was also restored and she was discharged home after 2 months stay in admission in the hospital. Her pre-existing hypertension which was also high at time of admission was adequately controlled. Two years follow-up revealed no recurrence of her symptomatology. Nevertheless, she is on follow-up because of possible recurrence of the renal stones which could also cause a recurrence of the leukoencephalopathy syndrome. |
A fifteen-year-old female adolescent was admitted to the child and adolescent acute psychiatric unit of our hospital for the first time after remaining isolated in her home for four months. She had initiated treatment in the outpatient clinic of her district in June 2013, attending only one visit and refusing all psychiatric treatment thereafter.\nThere was a family history of schizophrenia in second degree relative on her mother's side. The patient's medical history, according to her family, included recurrent acute bronchitis until 10 years of age. No history of alcohol or tobacco consumption or psychoactive substance use was reported. The patient was very selective and restrictive in her meals since her childhood. She only ate biscuits, ham croquettes, bread, and omelettes.\nRegarding school, she described acceptable performance until the previous academic year when she presented some difficulties, and her teachers decided to change her to another classroom with a lower academic level.\nHer premorbid personality was described as quiet and shy, with some difficulty in social relationships, but with a group of friends at the beginning of the episode. She lived with her parents and an 18-year-old sister in a mid-size city (with a population of about 216,000 inhabitants).\nThe patient reported that during the previous academic year she had been separated from her friends due to the change of classroom and at the same time she had begun to suffer digestive cramps and headaches, and as a result she stopped going to school. She stayed at home, with no interest in leaving the house, playing computer games and surfing the internet all day from April 2013 until her admission in our department 6 months later. The family explained that she had also become withdrawn from them, showing little interest in conversation, giving only monosyllabic answers, and gesturing in response to her parents' questions. She showed neglect in her personal hygiene and apathy regarding her physical appearance.\nAt the time of her admission, the mental status examination showed she was conscious and oriented in time, place, and person, though unkempt and exhibiting psychomotor retardation, poor eye contact, and limited language, answering questions with monosyllables or gestures. She also displayed emotional lability when discussing sensitive issues like friends and school and showed hypothymia, poorly reactive mood, apathy, weakness, and anhedonia and was only interested in computer usage. She did not exhibit impaired sensory perception, self-referentiality, or psychotic behavior, though she was sleeping from 4:00 am to noon. Her food intake was very selective, limited to certain specific foods, but there was no loss of appetite. The patient denied having suicidal ideation.\nIn the first interview, apathy and depressive moods were observed when she spoke about life events related to her friends at school and how this situation interfered with her functioning. Physical examination revealed a weight of 83.1 kg, height of 1.63 m, body mass index of 31.3, body mass index standard deviation score of 2.689, and blood pleasure of 120/80 mmHg. She denied muscular pain and no goiter or myxedema.\nSoon after admission, a blood test and electrocardiogram (EKG) were performed. EKG, liver function tests, electrolytes, and whole blood count were within normal limits. Due to her isolation at home and selective food intake, levels of some vitamins were analyzed; vitamin D3 and folic acid levels were insufficient (vitamin D3: 20.9 ng/mL insufficient with levels above 30 ng/mL and folic acid: 2.3 ng/mL insufficient with levels above 3 ng/mL). The thyroid function test showed thyroid-stimulating hormone (TSH) levels of 24.159 mIU/L (reference range: 0.400–4.00), free thyroxine (FT4) 0.93 ng/dL (reference range: 0.80–2.00), and triiodothyronine (T3) 1.08 ng/mL (reference range: 0.70–1.90), with antithyroid peroxidase antibodies >1300 IU/mL (reference range: <35).\nWe requested interconsultation with the endocrinology department where hypothyroidism secondary to Hashimoto's thyroiditis was diagnosed, and treatment with levothyroxine 50 micrograms/day was recommended. We began folic acid (5 mg/day) and vitamin D (0.266 mg/alternate days) supplementation too.\nDuring her hospital stay, the patient adapted well to the rules of the unit, was sociable with peers, and showed no behavioural disturbances at any time.\nAfter 8 days in our acute unit, the patient showed significant improvement in mood and social interaction and was discharged. Monitoring continued in the outpatient clinic, while the patient maintained the same pharmacological treatment. At six weeks, the patient was asymptomatic; she had returned to school showing acceptable performance and socialization with family and classmates. The new thyroid function test showed TSH 9.096 mIU/L and FT4 1.21 ng/dL. Six-months after discharge, the improvement persisted, and thyroid function was TSH 6.935 mIU/L and FT4 1.35 ng/dL and antithyroid peroxidase antibodies decreased, with 75 micrograms/day of levothyroxine. |
82 years old married male working as a farmer was suffering from bipolar affective disorder for the last 50 years. He had multiple episodes of mania and depression which were treated with various psychotropic medications. He was maintaining well for the last 3 years and hence had stopped all medications and was only taking lorazepam 1 mg on as and when required basis for sleep.\nHe now presented with complaints of over talkativeness, big talks, over activity, decreased need for sleep and excessive cheerfulness for the last 1 month. There was no associated history of fits, urinary incontinence, fever, forgetfulness or other symptoms suggestive of organicity. His personal and family history were non significant. Medical history revealed patient to be suffering from benign hypertrophy of prostrate grade I. Mental status examination revealed elated affect, increased psychomotor activity, grandiose ideation and absent insight. His Mini Mental Status Examination score was 25/30. A diagnosis of BPAD currently manic episode was made. He was started on divalproex 250 mg HS, olanzapine 5 mg Hs increased to 10 mg HS after 5 days and lorazepam 2 mg HS. The patient after taking medications for about two days reported excessive sleep and consequently stopped divalproex. He however, continued olanzapine and lorazepam in the dose prescribed. However, after about two days of 10 mg olanzapine, the family members noticed that the patient would get up in the night at around 2.00 a.m. He would then start roaming about in the house, would not recognize the family members, would start saying that he should go to work and would not recognize the time and place also. He would urinate at wrong places. On occasions, he would start picking up the bed sheets or the pillow covers saying that ants were sitting there, though the family members would not see anything there. He would on occasions stand on the table and start speaking to self or would try to reach for some invisible objects in the air. He would then go to sleep at around 5:00 a.m. in the morning and would get up at 9:00 a.m. He would not recollect the happenings of the night time. His day time activities however, started to decrease. These episodes continued regularly every night. A diagnosis of delirium was made and he was investigated. Hematological investigations, including serum electrolytes, liver function tests were normal except for increased blood Urea-54 mg% (15-45 mg% normal range) and increased S. Creatinine-2 mg% (0.5-1.0 mg% normal range). Computed tomography scan head was normal. Lorazepam was increased to 4 mg per day and olanzapine was continued in the same dose. The manic symptoms decreased in intensity. The family members then stopped olanzapine on their own. Two days after this, the family members reported improvement in delirious symptoms. He would now take only lorazepam 4 mg per day and would have improvement in manic as well as the abnormal behavior suggestive of delirium. Hematological investigations done after the improvement in abnormal behavioral episodes of delirium revealed similar findings. |
A 35-year-old female patient presented herself as an outpatient in 2012 with a two-week history of pain in the right leg and hyposensibility of the right calf. For further investigation, an MRI of the lumbar spine was performed showing a right-sided cystic tumor of the pelvis instead of a suspected disc herniation. In the following, an abdominal CT scan was performed revealing an osteolytic involvement of the right ileum, os sacrum, and the right-sided neuroforamina s1–s4 ().\nOn physical examination, the patient showed a moderate swelling of the right gluteal region with a slight reduction in range of motion of the right hip due to severe pain during leg movements. Otherwise the exam was unremarkable with normal blood circulation in both legs and intact sensitivity except for the right dorsal calf.\nTo further evaluate the tumor, an MRI scan of the pelvis and an open biopsy were performed ().\nThe suspected diagnosis of an aneurysmal bone cyst was confirmed by histopathological examination. Treatment options were discussed in an interdisciplinary tumor board. Surgical intervention was not recommended due to inaccessibility. Therefore, selective arterial embolization was performed by our radiologists. Additionally, the off-label use of 60 mg of Denosumab, once every four weeks, was initiated. It was decided to start the patient with 60 mg and not 120 mg to assess a potential therapeutic effect while keeping side-effects low. Due to the unstable situation of the right pelvis, the patient was advised to use crutches with no weight bearing of the right leg. Follow-up examination and CT imaging were performed every three months and revealed a significant reduction in pain and a notable increase in cortical thickness, while less mineralization of the cyst cavity was seen (). Therefore, Denosumab dosages were not changed. Additionally, an increase to partial weight bearing of the leg up to 20 kg was possible and therapy with Denosumab was discontinued after a total course of twelve months. During this time no remarkable side-effects were detected. During interdisciplinary discussions, surgical treatment via curettage and a combination of bone grafting and cement implantation of the defect was planned. Prior to surgery embolization of the remaining arteries was performed again.\nSurgery included curettage, implantation of autologous spongiosa, alloplastic bone grafts, artificial bone material, and bone cement.\nA follow-up CT scan six months after surgery revealed a significant tumor progress with decreased cortical thickness. Since there was a good response to Denosumab before, the treatment was restarted.\nFollow-up imaging showed a significant response with tumor reduction, recalcification, and increasing stability of the pelvis again. Thus Denosumab therapy was continued, controlling the aforementioned lesion and allowing the patient to fully bear weight on the said leg. In the following, laboratory tests revealed signs of low turnover osteoporosis with low calcium, phosphate, and 25-hydroxyvitamin D3 levels combined with increased parathyroid hormone (PTH) levels resulting in discontinuation of Denosumab after a total of 17 months during the second cycle. Until today the overall patient follow-up is about four years with clinical examinations being performed in our outpatient clinic every 6–12 months. Denosumab has been discontinued for fifteen months with clinical and radiological examination showing a stable disease so far (). |
A 40-year-old female presented with chronic headache with infrequent exacerbations. She presents with worsening headache for three months with associated vertigo, nausea, and vomiting not responding to analgesics or vestibular sedatives. Her symptoms initially started twelve years ago as a diffused mild headache, which persisted through the day. Gradually, the headache worsened to a severe headache episodically associated with vertigo, nausea, and vomiting. These episodes lasted for two to three days and got resolved. She was treated with flunarizine for suspected basilar migraine but did not show any response. From the last year, she had monthly exacerbations of headache associated with distressing vertigo, unsteadiness of gait, and right-sided body numbness. In between these episodes, she had a significant dull diffuse headache not responding to simple analgesia. She did not complain of fever or night sweats but had constitutional symptoms lasting for several months. All of these symptoms severely affected her daily activities and functionality.\nDuring the last 17 years, she had repeated episodes of neurological deficits. Even before the headache appeared, she has presented with visual impairment of the right eye and right lateral rectus palsy and was treated as retrobulbar neuritis with good response to methylprednisolone. One year later, she developed left-sided visual impairment, which fully responded to methylprednisolone. MRI imaging at that time revealed normal results. Few months after this event, she got admitted with right hemisensory loss with hemiplegia, and a demyelination disease or hemiplegic migraine was suspected. Second MRI was performed at this admission, and no abnormalities were detected again. Eight years ago, she had developed a left lower motor type facial nerve palsy, which was attributed to Bell's palsy. Within the last year, she was diagnosed to have depression and anxiety for which she was treated for few months. Other than the first two instances, she was not treated with steroids thereafter. She did not complain of weight loss and did not have constitutional symptoms or chest symptoms during these periods.\nOn examination, she is an averagely built female with a BMI of 23 kg/m2. She is afebrile, pale, and did not have lymphadenopathy. Her GCS was 15/15, and she was conscious and rational with normal pupillary response, visual acuity, visual field examination, and fundoscopy. There was no neck rigidity, and she had residual left lower motor VII palsy. She had an ataxic broad-based gait with unsteadiness. Upper and lower limb examination is clinically normal. Her respiratory, cardiovascular, and abdomen examinations were unremarkable.\nInvestigations revealed a hemoglobin count of 9 g/dL with normal white cells and platelets. ESR was elevated to 86 mm/1st hour. Renal- and liver-related biochemical investigations were normal with an alkaline phosphatase within the normal range. Initial MRI scans of the brain done 10 years ago did not reveal any abnormalities such as demyelination, optic nerve enhancement of focal lesions in the cerebrum, or cerebellum. CSF examination revealed an elevated protein level of 55 mg/dl with normal glucose and cells with negative oligoclonal bands or TB-PCR. Vasculitis investigations including ANA, ANCA (ELISA and Immunofluorescence), and RF were negative. Syphilis serology and HIV testing were also negative. Chest radiograph, ultrasound abdomen, and CT scan of chest and abdomen did not reveal any mediastinal lymphadenopathy or focal lesions in visceral organ or evidence of any malignancy. Serum ACE levels (19 µ/l) and ionized calcium levels were normal. We performed a new MRI scan of the brain with contrast, which revealed a diffuse and patchy meningeal thickening and enhancement mainly in the right frontoparietal and left occipital regions with a minor enhancement of bilateral optic sheaths (). Her NMO antibodies were normal, and the MRI did not show any areas of demyelination. Therefore, she underwent a dural biopsy from the thickened dura, which revealed large areas of caseous necrosis surrounded by epithelioid histiocytes and lymphoid cells with a few isolated giant cells in the adjacent vicinity (). There were no features of vasculitis or sarcoidosis. TB-PCR of tissue and acid-fast bacilli were negative. Final conclusion was necrotizing granulomatous inflammation suggestive of dural tuberculosis. This diagnosis was presumed by the presence of necrotizing granulomatous necrosis with caseation with a strongly positive Mantoux test of 25 mm (), and later was supported by a marked response to antituberculous medication.\nWe initiated her on antituberculous therapy (all four drugs for 3 months and 9 months of rifampicin and isoniazid) without streptomycin as she is already having vestibular symptoms. Steroids were added concurrently (1 mg/kg) and was continued for 6 weeks and was tailed off over a month. She experienced a marked improvement of her headache, and she could do her daily activities normally. After a year of anti-TB medication and follow-up, she did not complain of any worsening of symptoms. |
A medically free 14-year-old male who was obese sustained an injury to his left knee after jumping from 3 stairs. The patient mentioned that he predominantly landed on his left lower limb with his left knee in full extension and in external rotation. The patient started complaining of left knee pain limiting his range of motion and ability to bear weight immediately after the fall. He was brought to the Emergency Department (ER) of King Saud Medical City (KSMC) by his parents immediately after the injury. On physical examination, the left knee was profoundly swollen and bruised. There was tenderness over the tibial tuberosity and lateral joint line. He was unable to actively move the knee joint. The passive range of motion was painful. There were no signs indicating compartment syndrome or neurological or vascular injury. X-ray radiographs revealed a Watson-Jones type IIIB avulsion fracture of the tibial tuberosity apophysis (). A CT scan showed a step of the articular surface more than 2 mm extending to the posterior-lateral epiphyseal part of the proximal tibia (lateral tibial plateau) ().\nThe patient was admitted and was prepared for operative management. A procedure was planned and done on a radiolucent table under general anesthesia. A tourniquet was used to avoid excessive bleeding during the procedure. The tourniquet was inflated after pulling down the quadriceps to avoid blocking the reduction due to the extensor mechanism. The leg is prepped and draped according to the standard orthopedic protocol. Anterolateral approach of the knee was used with an incision starting from the lateral upper border of the patella to 10 cm down. Deep fascia was opened anterior to the iliotibial tract. The fracture line was identified; the reduction of the articular step was done using a reduction clamp and assured using a portable image intensifier on flexion and extension of the knee without arthrotomy. We avoided arthrotomy of the joint to not make it vulnerable to infection and possible scarring. Stabilization of the reduction was maintained using a k-wire. Definitive fixation was achieved with three 3.5 mm partially threaded cancellous screws placed under fluoroscopic guidance for the tibial tuberosity fracture. A proximal tibial plate was slid laterally and was used to buttress the lateral tibial column. Careful placement of the screws was done to not cross the physis with the help of a C-arm (). After fixation, good hemostasis was achieved, drain was placed, and the range of motion was assessed which was full. Closure was done layer by layer, then dressing after. The postoperative plan was to immobilize the knee in a cylindrical cast for 3 weeks with no weight bearing on the left lower limb with the use of crutches for ambulation.\nPostoperative knee CT scan is requested to ensure that the fracture is anatomically reduced. The patient received analgesia and antibiotics, and drain was removed 24 hours post-op. The patient was seen in an orthopedic clinic after 3 weeks, there were no signs of surgical site infection, and the clips were removed. The controlled range of motion was advised using a hinged knee brace throughout the day for 4 weeks. A follow-up X-ray () shows that the fracture is aligned with no loss of reduction or displacement. Physiotherapy is advised 7 weeks postoperative management focusing on the range of motion and strengthening. The patient was seen 6 weeks later, he had full range of motion with no deformity, and there were no complaints reported by the patient like locking or pain. |
A 16-year-old male with moderate mental retardation presented to our clinic with 2 years history of multiple episodes of chewing movements accompanied by a state of altered consciousness, during which he would not respond to his name being called out and would continue to give a vacant stare in one direction. There was no history of unconsciousness, repeated rhythmic movements of the limbs, loss of tone resulting in falls, tongue bite, and urinary incontinence. Each episode was abrupt in onset and lasted about 5–10 min, after which the patient would spontaneously become responsive to the surroundings once again. A clinical impression of complex partial seizure was made based on the semiology, and the patient was started on CBZ 200 mg twice daily (8 mg/kg/day) for the same.\nThe patient was brought to the emergency department 4 days later with acute dystonia of the neck which had started approximately 2 h back. His neck was found to be held firmly in an extended position with the head tilted toward the right. He had been fully conscious, oriented, and responsive to external stimuli throughout this period, though he was noticed to be agitated. He was administered promethazine 50 mg intramuscularly with the dual purpose of achieving sedation, as well as using its anticholinergic properties to relieve the dystonia. The dystonia was seen to resolve completely within the next 25–30 min. A serum CBZ level estimation was done, and levels of 8 mcg/ml were obtained. Since it was within the normal range (5–12 mcg/ml), it was considered unlikely that the CBZ toxicity could have led to the dystonia. A literature review also confirmed that there was very little likelihood of the dystonia resulting from the use of CBZ, especially below toxic levels.\nTherefore, the patient was advised to continue CBZ and kept under observation. He developed the cervical dystonia once again after 3 days. Supportive care was provided and promethazine 50 mg was administered via the intramuscular route. The dystonia resolved within a period of 25–30 min. It was considered that CBZ could be the offending agent, responsible for the episodes of dystonia. Thus, it was decided to stop CBZ and shift the patient over to another anticonvulsant drug.\nThe patient was shifted over to sodium valproate at 1200 mg/day (24 mg/kg/day) for seizure prophylaxis and was followed up for a 2-week period during which the patient remained seizure free, and no recurrence of dystonia was found. |
A 21-year-old female was referred to the Department of Periodontics, with chief complaints of gingival enlargement of both maxilla and mandible on right side and an inability to chew on that side []. Before one year, she noticed a gingival over growth []. She did not respond to non-surgical periodontal management or the use of chlorhexidine mouthwash.\nAn extra-oral examination revealed facial asymmetry and swelling extending anteriorly from angle of the mouth to the angle of the mandible posteriorly and superiorly from the lower eyelid to the base of the mandible inferiorly. The sub mandibular and sub mental lymphnodes were enlarged and palpable.\nAn intra-oral examination showed occlusal surfaces of all the posterior teeth on right side covered with plaque and calculus. A periodontal examination showed the bluish red gingival extending from maxillary canine to second molar and mandibular first premolar to second molar [], with soft and oedematous consistency. A diffuse type of gingival enlargement which we classified as Grade III Enlargement was apparent in affected area. It covered three quarters or more of the crown (Bőkenkamp et al. , 1994). Generalized bleeding on probing with suppuration was present in relation to the area of gingival enlargement along with insufficient width of attached gingival.\nOn hard tissue examination, we found traumatic occlusion with pathological migration of the teeth. Mobility in relation to upper right premolars and molars and lower right molars was also noted.\nRadiographic examination revealed horizontal bone loss from distal aspect of mandibular first premolar to second molar and maxillary premolars on the right side and vertical bone loss in relation to maxillary molars in the same region [].\nAfter clinical and radiographic diagnosis, a complete treatment was planned to get the best possible results. Treatment started with non surgical periodontal therapy including scaling, root planing and polishing with oral hygiene instructions given to the patient, which was completed in three consecutive appointments. Occlusal correction was done to relieve the traumatic occlusion. Inflammation persisted on the right side even after non surgical periodontal therapy. So, a ledge and wedge technique was planned to excise the enlarged tissue, expose the surgical site to approach to furcation involvement and areas of bony defects and contour gingival morphology. After excising the tissue the surgical site was closed with interrupted loop sutures for better approximation. At the 7th day after surgery, patient presented good healing. Follow-up appointments were performed at 14th, 28th day, 3rd month, and 6th month []. An incisional biopsy was done and specimen sent for histopathological examination.\nOn histopathology examination, it showed normal to hyperplastic stratified squamous epithelium with elongated reteridges []. The underlying connective tissue showed thick, long collagen fibers arranged in bundles with thick plump fibroblasts intermixed with histiocytes and plasma cells with a scattering of fibrocytes. It also showed mononuclear and multinucleated giant cells []. Clusters of odontogenic epithelial rest cells were seen in few areas []. A diagnosis of peripheral giant cell fibroma, a benign tumor, containing multinucleated fibroblasts was made. |
A 20-year-old woman presented to the emergency department with complaints of severe abdominal pain, multiple episodes of vomiting and obstipation for one day. She was a known case of abdominal tuberculosis and had received anti-tubercular treatment for two months. Vital signs showed a blood pressure of 100/64 mm Hg, heart rate of 110 per minute, respiratory rate of 18 per minute, oxygen saturation of 96% on room air and a temperature of 100.5°F. She was cachectic with a weight of 28 kg which was less than 5th percentile for gender and age matched normal population. Physical examination revealed diffuse abdominal tenderness and rigidity. Her erect abdominal radiography showed gas under right dome of the diaphragm. Clinical diagnosis of perforation peritonitis was made and the patient was wheeled to the operating room for emergent laparotomy. Intraoperatively, a 3 cm x 2 cm single ileal perforation was found 1 foot proximal to the ileocecal junction, with unhealthy bowel margins and multiple mesenteric lymphadenopathy. Resection of perforated bowel segment with proximal ileostomy and mucus fistula of distal ileum was performed. Later, histopathology report confirmed tubercular etiology showing caseous necrosis in mesenteric lymph nodes and epithelioid granuloma in ulcer edge biopsy. Two days after the surgery, the patient was started on oral feeds and was accepting meals orally subsequently. Ten days following surgery, the patient had multiple episodes of bilious vomiting with stoma output decreasing to less than 100 mL per day. Her abdomen remained flat with no increase in bowel sounds. Abdominal radiography showed dilated stomach with no air fluid levels. Oral feeds were stopped and a nasogastric tube was placed for drainage of gastric contents. A contrast enhanced computed tomography (CECT) abdomen was done to determine the cause of obstruction which revealed a decreased aortomesenteric angle of 15° (Figure ).\nAs a result of this decreased angle, an abrupt collapse of the third part of the duodenum was seen along with proximal duodenal and stomach dilation (Figure ).\nBased on the clinical features and CECT findings, diagnosis of SMA syndrome was made and the patient was managed conservatively. The patient was not tolerating enteral feeds and no relief of postprandial abdominal pain was noticed even on changing to left lateral decubitus or prone position. Considering the patient’s nutritional condition, total parenteral nutrition (TPN) was started. Anti-tubercular drugs were continued with oral sips. She was started on 1500 kcal/day (50 kcal/kg/day), taking care of refeeding syndrome and was gradually titrated up to provide maximal caloric support. Monitoring of weight, blood glucose, electrolytes, liver function test, and lipid profile was done to prevent any TPN complications. A good clinical response was seen after starting TPN. The patient’s weight gradually increased to 34 kg in three weeks and gradually the stoma output increased and nasogastric drainage decreased. TPN was tapered off gradually with concurrent starting of oral feeds. On discharge, the patient was advised a high caloric and protein diet with small frequent meals. A follow-up CECT was done after six weeks from discharge which showed an increase in aortomesenteric angle from 15° to 21° (Figure ) with the patient being asymptomatic and a total weight gain of 12 kg post-surgery. |
We report a case of a 75 year old woman addressed to Allergology Department of our hospital in January 2014 for recurrent episodes of angioedema since the age of 66, with progressively increased severity and frequency. It was first considered to be induced by treatment with angiotensin-converting-enzyme inhibitors (ACEI) for mild hypertension, but she continued to have angioedema attacks for the next 6 years after discontinuation of ACE, with progressive aggravation during the last year. The previous multiple evaluations by many specialists in other hospitals did not succeed to give a clear diagnosis and treatment.\nThe patient had no relevant medical history and took no medication, except ACEI that was stopped some months after angioedema onset. No relation with possible allergic stimuli could be identified and she had no clinical manifestations between attacks. Angioedema was painful, not accompanied by urticaria or abdominal symptoms, located variably to neck, arms or buttocks, without facial involvement during 6 years. The attacks occurred at weeks or months intervals and usually lasted between 48 and 72 h, irrespective of corticosteroids and antihistamines treatment usually administered. The frequency of attacks had progressively increased from one at 2–3 months intervals to almost weekly during the last year before presentation. The last angioedema attack, determining emergency hospitalization in September 2014, was more severe and prolonged, accompanied for the first time by laryngeal edema and respiratory symptoms. The extended medical evaluation, including complete blood tests for inflammation, allergy, autoimmunity and cancer, were all negative (Table ). Full body CT scan and bone marrow examination were normal. No criteria for lymphoproliferative, mieloproliferative or autoimmune diseases could be found. Measurement of C1 inhibitor (C1INH) in plasma showed significantly decreased level at all measurements, with low activity ranging from 58 to 4% and constantly low C4 (Fig. ). Complement fraction C1q plasma level was measured twice and had normal value. Genetic tests were not performed, given the patient advanced age and lack of family history of angioedema, which are against HAE. A spontaneous mutation in SERPING 1 gene is noticed in up to 25% cases of HAE without family history, but we considered this probability very low in our case, due to late onset of angioedema.\nTreatment of angioedema attacks before hospitalization consisted of antihistamines and systemic corticosteroids, which proved to be ineffective. Since no pathogenic therapy with C1INH concentrate, antagonists of bradikinin receptors (icatibant) or selective inhibitor of plasma kallikrein (ecallantide) was available in 2014, we first initiated daily prophylactic treatment with tranexamic acid for 3 months, with no amelioration. During the more severe attack with laryngeal edema, in September 2014, we have switched to attenuated androgen danazol, given 400 mg the initial dose, reduced to 200 mg daily after 1 week and then to 100 mg daily. The clinical evolution was very good, no angioedema attack occurred since the introduction of danazol. C1INH and C4 plasma levels increased after 2 weeks of treatment and became normal after 1 month. After some months, the patient decided herself to discontinue danazol for short time, in order to check effects and to taper the minimal dose. Serum C1-INH and C4 plasma levels were measured after 2 and 4 weeks and showed significant lower levels, but no angioedema attack occurred during this period of time. She therefore restarted danazol prophylactic therapy at a minimum of 50 mg dose daily, ongoing after 3 years. The patient was closely monitored during the next 3 years, with complete clinical and laboratory control twice a year. We took into consideration the possible side effects of danazol, mainly dislipidemia, haematological and liver malignancies and any other complications or concomitant diseases. The clinical evaluation was very good, except two episodes of pulmonary cysts infection, remitted with broad spectrum antibiotherapy, which could not be related to danazol treatment. The clinical outcome of angioedema after 3 years of danazol treatment is very good, with no attacks or other related symptoms. No clinical or laboratory sign of any disease that could induce C1 INH deficiency occurred. No relevant side effects of danazol were noticed. The patient has an improved quality of life due to therapeutic compliance and general management plan. |
Our patient was a 12-year-old female who presented to us with “poor cosmesis” of her face, decreased vision, and an inability to close her left eye since birth. Initially, the parents noticed that the child had multiple episodes of recurrent watering in her left eye during the first five months of her life. But later on, they clearly noticed that she could not close her left eye and had an unusual facial appearance while smiling or crying.\nThe mother of the patient was 28 years of age at the time of birth of the patient. The mother did not have any history of antenatal infection or fever. She had not used any medications or supplements during pregnancy as well. The patient was born by normal vaginal delivery without any complications. There were three siblings of the patient who were all healthy without any medical illness or any similar abnormality in the family.\nOphthalmic examination of the patient revealed a best corrected visual acuity (BCVA) of 20/20 and 20/60 in her right and left eyes, respectively (Table ). The cause of her decreased vision in her left eye turned out to be anisometropic amblyopia, due to asymmetric hyperopia and astigmatism in her eyes. The patient gave a history of patching her right eye for one year for amblyopia treatment at the age of 10 years, but mentioned that it did not significantly improve her vision.\nThe patient had lagophthalmos in her left eye. On complete closure of her left eye, the patient had a space measuring 3 mm between her upper and lower eyelids. The corneal sensitivity was normal, symmetric, and intact in both the eyes. Slit-lamp examination revealed no corneal subepithelial punctate erosions, abrasions or any other sign of exposure keratopathy in the affected eye. The patient exhibited normal Bell’s phenomenon in both of her eyes. The extraocular motility testing of the patient showed a normal central position of the eyes in primary gaze. However, there was a left lateral rectus palsy leading to an absent abduction in the left eye (Figure ). The rest of the movements in all gaze positions were intact. The patient did not have diplopia or ocular pain in any of the gaze positions.\nCranial nerve examination showed left facial palsy (Figure ). When asked to crease up the forehead, no furrows were observed on the left side. She could not keep her left eye closed against resistance, and there was deviation of the angle of her mouth to the right side when asked to reveal her teeth. When asked to close her eyes, there was an incomplete closure of the left eye but due to normal Bell’s phenomenon, both the eyes rolled up-and-out, leaving no part of cornea exposed. When asked to protrude her tongue, there was deviation of the tongue to the left side, pointing towards left hypoglossal nerve palsy (Figure ). Rest of the central nervous system (CNS) and cranial nerve examinations were normal. Her hearing was intact as well. She did not have any other abnormality in cardiovascular, gastrointestinal, urinary, and reproductive systems.\nThe child had associated musculoskeletal deformities as well. She had syndactyly in one of her hands, and brachydactyly in both. She had already been operated to release her syndactyly at the age of six years (Figures -).\nHer various laboratory investigations revealed a healthy profile. The x-rays of her hands revealed a rudimentary middle-phalanx in most of her fingers, while an absent distal-phalanx in the little finger of her left hand was leading to brachydactyly. The magnetic resonance imaging (MRI) of her brain also showed normal cranial nerve complexes with no signs of cranial nerve nuclei hypoplasia and no other gross CNS pathology. |
A 21-year-old Japanese male presented at the outpatient department with sporadic nausea, abdominal discomfort, and worsening headache. Cysto-peritoneal (CP) and VP shunts were implanted 11 months after birth by another doctor for the treatment of hydrocephalus due to a suprasellar arachnoid cyst. His VP shunt had been replaced twice by the age of 13 years. At the time of last surgery, the CP cranial tube was removed while the extra-cranial tube was left in place because it passed through the mediastinal space rather than the subcutaneous tissue and adhered to the surrounding organs. During this course of treatment, the patient had been diagnosed with slit ventricle syndrome but had no current restrictions on daily activity except for mild mental retardation. He had no remarkable medical history to suggest systemic abnormalities or risk factors for infection and had not traveled to foreign countries.\nNausea was reported for 2 months and abdominal pain for 1 month before admission. A few days before presentation, the patient developed fever and mild consciousness disturbance. Initial physical examination revealed a fever of 38.9°C, restlessness, and significant abdominal distension but no meningeal irritation or focal signs of neurological dysfunction. White blood cell count was 10,900/mm3, and serum C-reactive protein level was 31.8 mg/dl. Chest X-ray was negative but brain and abdominal computed tomography (CT) scans revealed enlarge lateral ventricles, a huge peritoneal cyst surrounding the distal end of the peritoneal tube, and fluid in the sheath surrounding the peritoneal tube, indicating possible shunt blockage and a concomitant increase in intracranial pressure (ICP) []. In contrast, chest CT revealed no abnormalities in bilateral lung fields or subcutaneous tissues of the chest. The tentative diagnosis was shunt malfunction resulting from some undefined peritoneal abnormality.\nContinuous external drainage was established to remove CSF from the lateral ventricles and peritoneal cyst. The CSF was clear and the fluid in the abdominal cyst was xanthochromatic. Chemical screening of CSF was negative. The cyst fluid contained 1495 cells/mm3 (69 monocytes and 1426 polycytes/mm3) and 1.4 g/dl total protein. Antibiotic therapies for abdominal infection were empirically administered and bacterial cultures analyzed, but no causative pathogen was identified.\nPreoperative clinical symptoms immediately improved after surgery to insert an external drainage system. However, the cyst fluid gradually recollected and signs of mild inflammation remained despite antibiotic therapy. In the second stage of treatment, transcutaneous drainage of the cyst under CT guidance was performed; however, again cyst fluid recollected []. In the third stage of treatment, laparotomy was performed, which revealed no membrane surrounding the abdominal cyst (defining it as a pseudocyst), no malignancies within the omentum, and no ascites within the abdominal cavity.\nThroughout the course of treatment, all cytological examinations for malignancies were negative and bacterial cultures did not reveal a causative pathogen. Two months after initial surgery, inflammation of his left knee joint became apparent. The fluid taken from the joint was xanthochromatic, similar to the pseudocyst fluid, and Mycobacterium tuberculosis was detected in a culture from the fluid. Thus, the patient was diagnosed with an abdominal pseudocyst associated with TBP and successfully treated by anti-TB agents following insertion of a ventriculo-pleural shunt []. At follow-up 2 years after treatment, he again presented with signs of inflammation, and the abscess around the CP shunt was strongly suspected as the cause. He was again treated with anti-TB agents, and all signs of inflammation subsided after the removal of the previous CP shunt tube by thoracoscopic surgery. |
Mr P was a married man in his 40s with one child. He had served in the UK army and was deployed to the Bosnian conflict in the mid-1990s. During his deployment he was stationed at a morgue that had been set up to aid the identification of bodies recovered from a mass burial site. He described how the morgue contained a large number of bodies in various states of decomposition, with varying degrees of physical trauma. He reported re-experiencing fragmented intrusive images of the morgue and daily emotionally distressing nightmares of which he could not recall the content. He experienced high levels of anxiety that triggered his dissociative symptoms. These appeared to function as a coping strategy to avoid distressing emotions connected to his memory of the morgue. Other avoidance symptoms included feeling detached from people around him and feeling emotionally numb. Mr P avoided family occasions and busy places which could trigger his hyper-arousal symptoms and had never spoken to anyone about his experiences. He reported coping historically through the use of alcohol to block out his emotions. His avoidance behaviours had maintained his PTSD symptoms.\nAt the start of therapy sessions, Mr P was reluctant to talk about his past experiences stating he only felt able to discuss vague details about his intrusive memories. Mr P attempted to use physical objects such as stones and leaves, or his e-cigarette, as grounding strategies in the clinic room. However, he also reported feeling unable to tolerate any emotions connected to the events and found it difficult to identify peri-traumatic cognitions. Mr P appeared to experience intrusions of one particular fragmented image and could not recall any other contextual information from before or after this moment.\nIn both cases Mr C and Mr P experienced difficulties in engaging with traditional methods of imaginal reliving and prolonged exposure, due to high levels of avoidance and low thresholds for dissociation, and subsequently had difficulties accessing their cognitions for updating. For both clients the prior use of multiple grounding methods in the therapy room were unsuccessful. They experienced their traumatic memories as fragmented images with a sense of ‘nowness’ (), which indicated a distinct lack of contextualization.\nAn adaptive approach was then trialled to address these hindering factors. This involved three stages: (1) walking the client through the imaginal scene outdoors to address dissociation, (2) viewing the imaginal scene from multiple perspectives to facilitate contextualization of the memory and (3) identifying and reappraising the cognitions with frequently used approaches in trauma-focused therapy. |
In this case, a 25-year-old male patient lost his maxillary left central incisor long before. From 2001, he had been treated with pre- and post-operative orthodontic therapy and gnathosurgery to improve his facial appearance. In this process, the maxillary left lateral incisor was moved on the position of maxillary left central incisor and the maxillary left canine was moved on the position of maxillary left lateral incisor. The patient wanted to improve esthetics of anterior dentition. First, the space between anterior dentitions was analyzed. Analysis showed asymmetry in anterior dentitions because of the absence of the maxillary left central incisor and size difference between maxillary central and lateral incisors. Analysis also showed lingual inclination of the left lateral incisor. The maxillary left lateral incisor was more yellowish than the maxillary right central incisor and incisal edge of the maxillary right central incisor was concave due to attrition ().\nPorcelain laminate veneer was planned to improve esthetics of the anterior dentitions through transforming the shape of the teeth with porcelain laminate veneer. The treatment plan was to transform the maxillary left lateral incisor into a central incisor and the maxillary left canine into a lateral incisor. The restoration of the maxillary right central incisor was also included into the plan for esthetic improvement. The maxillary central incisors are mesio-distally larger than the lateral incisors for about 2 mm, and it was necessary to do minor tooth rearrangement by orthodontic treatment. After the tooth rearrangement, the space of the maxillary left lateral incisor became equal to that of the maxillary right central incisor. In addition, cross-bite of the maxillary left lateral incisor was resolved ().\nDiagnostic wax-up was performed including the maxillary right central incisor, the maxillary left lateral incisor and maxillary left canine on the study cast (). Mockup is critical for fabrication of the provisional restorations, enabling the patient and clinician to evaluate esthetics, and to preview the anticipated result.\nAfter duplicating diagnostic wax-up cast, putty index (Exafine putty type, GC, Tokyo, Japan) was made for tooth preparations. Selective preparations were performed with the guide of putty index using tapered diamond burs (Shofu, Kyoto, Japan). The preparation margins were placed at the equi-gingival level for esthetics. Preparation margins and surfaces were finished with a low speed white stone bur (Shofu, Kyoto, Japan) under water spray (, ).\nProvisional restorations were fabricated by Luxatemp® (DMG, Hamburg, Germany) using putty attained from the diagnostic wax-up cast. After 20 seconds of spot etching (Scotchbond etchant, 3M ESPE, MN, USA) for 20 seconds at the labial surfaces, provisional restorations were set using flowable light-cured composite resin (AELITEFLO™, Bisco, IL, USA).\nA week later, provisional restorations were removed and abutments were cleaned with a rubber cup. Before the impression was taken, retraction cord (UltraPak #00; Ultradent, UT, USA) was packed in the gingival sulcus to exposure the preparation margin. The final impression was taken with polyvinylsiloxane impression materials (Exafine putty type; GC Corporation, Tokyo, Japan / Honigum Light; DMG, Hamburg, Germany) light body polyvinylsiloxane (Honigum Light, DMG, Hamburg, Germany) using 1-step impression technique.\nPorcelain laminate veneers were fabricated with IPS e.max Press system (Ivoclar-vivadent, Schaan, Liechtenstein). After IPS e.max Press copings were made, they were layered with glass ceramics (IPS e.max Ceram Powder; Ivoclar-vivadent, Schaan, Liechtenstein) and then finishing and glazing were done (). IPS e.max Press has high flexural strength (400 MPa) and esthetics. Also, the advantage of this method is needless of a refractory cast. Porcelain laminate veneers were cemented with the light-cured resin cement (Variolink II; Ivoclar-vivadent, Schaan, Liechtenstein). After a month from the delivery, as shown in and , the gingiva was healthy and the interproximal area was fully filled with interdental papilla. Patient was satisfied with the harmonious shape and contour of the restorations which also showed good function. |
A 51-year-old man with a medical history of end-stage renal disease and poorly controlled DM was admitted to the hospital after syncope with head trauma. His neurological and cardiac workups were negative and his physical examination was within normal limits except for a slightly tender and mildly swollen left knee, which was attributed to his fall. The patient went into septic shock and was started on norepinephrine for vasopressor support. Blood culture results were Streptococcus dysgalactiae positive, and he was started on ceftriaxone. However, the infection source remained unclear. As his left knee swelling had increased compared to baseline on admission, a diagnostic tap was performed that showed purulent fluid comprising 250,000 white blood cells, which were predominantly neutrophils. Our patient underwent surgical drainage in the operating room (OR), where 100 mL of purulent fluid was drained from his left knee; however, he remained in a state of sepsis and was continued on vasopressors. We undertook a computed tomography (CT) scan of the left knee as shown in Figures , , which showed infection extending into the popliteal fossa, along with compression of the popliteal artery and vein. He underwent further surgery to drain 500 mL of purulent fluid from the popliteal fossa.\nCultured samples taken from the knee joint positively confirmed Streptococcus, which was consistent with his blood sample results. Given the extent of the infection, and because he was in septic shock with high-grade bacteremia, we administered a synergistic dose of gentamycin, and management was guided by infectious disease consultants.\nPostoperatively, his condition improved, vasopressors were weaned, and a transthoracic echocardiogram was performed to rule out infective endocarditis, the results of which were found to be vegetation negative. Therefore, we did not proceed to perform transesophageal echocardiography as the treatment duration was four weeks. He remained on ceftriaxone for four weeks, along with a synergetic dose of gentamycin with dialysis, and he was followed up in an outpatient clinic and showed good progress. |
A 23-year-old man sprained his left ankle while playing football 3 years previously. He complained of left ankle swelling and chronic pain. His ankle X-rays were unremarkable. Following this initial presentation, his symptoms gradually improved, but he continued to note intermittent resting pain at night that was unrelated to activity. This night pain could become severe but was relieved by NSAIDs. One year after his injury, the patient presented for treatment because of recurring left ankle pain and swelling. X-rays showed sclerotic lesions in the neck of the talus (Fig. ). Magnetic resonance imaging (MRI) of his left ankle showed oedema in the talar bone marrow, left ankle joint effusion, and periarticular soft tissue swelling (Fig. ). He was diagnosed with ankle traumatic arthritis and underwent ankle arthroscopy at a local hospital. We referred to the patient’s record at the local hospital. The attending doctor performed arthroscopic debridement of the left ankle joint and found free small pieces of broken bone and osteophytes in the ankle joint during the surgery. However, no lesion was detected on the articular surface of the talus. We speculated that this may have been related to the limited arthroscopic field of view as well as the diagnostic level of the physician and his or her the lack of experience in arthroscopic techniques. The patient obtained short-term relief of pain for 3 months after the surgery because the arthroscopic debridement reduced inflammation around the joint. Additionally, long-term bed rest after surgery helped to temporarily control the symptoms. However, unless the nidus is completely removed in cases of OO, the symptoms gradually worsen after a short period of relief. The patient’s pain reappeared 3 months later, with more frequent night pain than before surgery. Postoperative MRI exhibited widespread oedema in the talar bone marrow (Fig. ). The attending doctor provided supportive treatment such as ice, compression, physical therapy, and oral NSAIDs to ease the pain. During the following 2 years, the patient’s pain continued but remained unassociated with activity. When the patient presented to our hospital, he could hardly stand or walk. His left ankle joint was swollen with a slightly increased local skin temperature. His foot was diffusely tender, especially at the anterolateral talus, without exacerbation upon palpation of the anterior talofibular ligament (ATFL). The ankle anterior drawer test was negative. A typical OO nidus was demonstrated in the talus on X-ray (Fig. ), CT (Fig. ), and MRI (Fig. ). The patient subsequently underwent nidus excision and artificial bone grafting (Fig. ). Postoperative pathologic examination confirmed that the excised tumour was a 1.5- × 1.5- × 1.0-cm OO (Fig. ). The patient’s pain and swelling disappeared after the treatment. He was able to walk normally when he presented for follow-up 3 months later. |
A 60-year-old woman presented with grade 3 (morbid) obesity, body mass index (BMI) 40.02 kg/m2, body weight 108 kg, and height 164 cm. Patient-reported history included venous insufficiency and two operations for varicose veins in the lower limbs. The patient underwent a hysterectomy due to giant sarcomas, and a history of two childbirths by natural labor. She was not treated for other systemic diseases. The first rhinorrhea occurred approximately 3 years before. Initially, her symptoms were believed to be due to chronic allergic rhinitis. The patient experienced more frequent upper respiratory tract infections accompanied by pronounced general symptoms, an elevated body temperature of 38–39°C, fatigue, muscle and joint pains, altered mood and meningeal irritation. The patient was referred to a laryngology department at her place of residence where CT and MRI diagnostics revealed a clival bone defect of 9 × 7 mm and 8 × 5 × 6 mm, which was subsequently diagnosed and differentiated as cerebrocranial hernia. Nasal cerebrospinal fluid leak was confirmed in biochemical investigations. The patient was hospitalized in our clinic in July 2014 due to sinogenic orbital complications that manifested as swelling and congestion of the frontal orbital segment and irritation of cerebrospinal meninges. Imaging diagnostic scans confirmed a bone defect within the posterior clival framework that was larger compared to earlier scans. Fluid leaks occurred at several-day intervals. Therefore, the patient declined consent for the procedure and underwent consultations at other neurosurgery clinics. She received no guarantees of the potential endoscopic management of the leak. An intracranial approach was proposed. The patient was referred to our clinic for the second time due to increasing headaches, intense, continuous rhinorrhea, irritation of cerebrospinal meninges with inflammatory orbital complications and oral herpes of the upper lip. Follow-up imaging scans confirmed enlargement of the defect within the posterior clival framework to the size of 16 × 9 × 4 mm with a suspected meningocerebral hernia (–). In CT and MRI performed three times before surgery, pathology, especially hydrocephalus, in the central nervous system was excluded by the radiologist (, ).\nAn intranasal transsphenoid endoscopic exploration of the skull base defect and fluid leak closure procedure was performed using DigiPointer (Collin) neuronavigation with neurosurgical assistance. The procedure started with a complete bilateral sphenoethmoidectomy and radical resection of the vomer. This step allowed the surgeons to use the two nostrils – four hands operating technique. The next step identified the Turkish saddle, optic nerve, and carotid arteries on both sides within the sphenoid sinus. An extensive defect in the bone framework of the left internal carotid artery and exposed meninx within the sphenoid sinus were revealed, as observed previously in CT scans. The anterior and middle portions of the clivus were exposed between both carotid arteries. Resection of the barrier between both sides of the sphenoid sinus facilitated visualization of the bone defect within the posteroinferior part of the left sphenoid sinus and the posterior wall of the superior and middle clivus (–). The bony edges of the defect framework and the critical structure, in this case the basal artery, were identified during the procedure (, ). The artery protruded into the clival defect, and it was shifted 2 mm to the right of the medial line, as observed in the MRI scan. The bony edges of the defect were carefully dissected, and fragments of the cerebrospinal meninges were located intracranially with much difficulty (–). The skull base defect was repaired using fascia lata, septal nasal cartilage, autogenic fat, vastus lateralis muscle, fast-binding adhesive (fibrin glue) and a Tisseel TachoSil fibrin sealant patch.\nHigh-pressure cerebrospinal fluid leak and local bleeding were encountered intraoperatively despite hypotension. Lumbar constant drainage (φ17G) into the L3–4 space was used intraoperatively. Postoperative measurement of CSF pressures with a lumbar drain showed a mean of 30.0 cm H2O. The patient was awakened from anesthesia 3 days after the procedure. A follow-up head CT scan 7 days after surgery revealed no intracranial complications or neurological defects. The patient has remained free of clinical features of CSF leaks postoperatively. Perioperative vancomycin, metronidazole and meropenem were used for 14 days because Pseudomonas aeruginosa and Klebsiella pneumoniae bacilli were detected in microbial investigations. Lumbar drainage was used 1 to 7 days after surgery. CSF loss levels varied around approximately 150 ml per day, except for day 3 when the patient was transferred from the ICU to the increased postoperative supervision room, and the total leak volume was 350 ml. The patient was in limited neurological contact and required medications to improve intracranial flow on this day. The patient has recovered without complications, and she was discharged 14 days after the surgery. |
Patient 1 was a white 64-year-old male elementary school teacher, and patient 2 was a white 58-year-old male farmer. Both patients were referred to the colorectal surgery clinic for APR surgery by an oncologist following the diagnosis of rectal cancer. The initial presentation was constipation for patient 1 and severe weight loss for patient 2. Both patients had severe constipation and both reported pain on defecation and rectorrhagia. There was no history of prior surgeries, allergic reaction to drugs, psychological diseases, and smoking. A mass was found in the digital rectal examination in both patients, and they both underwent neoadjuvant chemoradiotherapy to shrink the tumor size.\nPathologic studies revealed rectal adenocarcinoma. Endoanal sonographic study showed distal rectal mass with involvement of external anal sphincter in both patients. There was no metastasis in patients. All options alongside the new method were described to the patients and informed written consent was obtained.\nThe patients were positioned in a semi-lithotomy position and received general anesthesia. Ceftriaxone and metronidazole were administered intravenously at the induction of anesthesia. The patients also received neomycin and metronidazole orally a day before surgery and underwent APR surgery. Inferior part of rectus abdominis muscle flap was used for reconstruction with the following technique (Alem Method).\nFor patient 1, the flap was taken from the inferior part of the rectus abdominis muscle and mobilized by lifting up the skin paddle, its underlying fat, and rectus muscle. The skin and anterior fascia were dissected from the bulk of the muscle (). The right rectus abdominis muscle was preferably selected to allow colostomy through the left rectus muscle. The inferior part of rectus abdominis muscle was cut just below the arcuate line and medial to the semilunar line (). The inferior epigastric artery was carefully dissected. Perforating vessels in anterior rectus sheath were also dissected to achieve minimal fascial resection to prevent abdominal hernias and allowing primary closure of the defect. The flap dissection was continued to the origin which is on the pubis (). The flap was then fashioned into the perineum (). The skin and external rectus fascia was used to repair the abdominal incision, so the risk of an incisional hernia was minimized and the muscle itself was used to fill the defect in the pelvic floor. Multiplelayer sutures with Vicryl 2-0 were used to place the flap without any tensions (). The defect created by the rectus muscle excision on the abdominal wall was closed and repaired primarily with minimal tension using nylon. To reduce the risk of infection, no mesh was used.\nThe same procedure was done for patient 2, but in both sides, which resulted in a bilateral inferior rectus abdominis muscle flap reconstruction, since the pelvic cavity of patient 2 could not be covered by a unilateral flap. The colostomy in this patient was placed on the left side of the abdomen ().\nAfter the surgery, 5000 units of heparin was administered twice a day subcutaneously.\nThis procedure was done on a human in this study for the first time, and an attending colorectal surgeon with more than 6 years of experience performed the procedure.\nThe follow-up visits were scheduled for 1 week, 1 month, 3 months, 6 months, and 12 months after the surgery in the colorectal surgery clinic.\nThe duration of surgery was 124 minutes for patient 1 and 142 minutes for patient 2. After the surgery, colostomy functioned properly in both patients. The length of hospital stay for patient 1 was 5 days and for patient 2 was 4 days. Both patients started walking and taking liquid food 1 day and 2 days after the surgery, respectively.\nThe patients were instructed about the ostomy care and other postabdominal surgery cares (heavy lifting and abdominal binder).\nNo significant blood loss was observed during the surgery. Patient 1 had an abdominal wound infection, with mucopurulent discharge, which was treated conservatively with orally administered antibiotics, wound washing, and changing clothes twice a day (Clavian-Dindo grade I). There was no specific complication in patient 2 after the surgery and before discharge. In the follow-up period, there were no reported or observed complications in the patients. No fatal outcomes were reported in the 12-month follow-up period. Obstruction, wound infection, abdominal hernia, abscess formation, flap loss, cellulitis, prolonged healing, pulmonary thromboembolism, deep vein thrombosis, parastomal hernias, and urinary tract infection were not observed. |
A 61-year-old otherwise healthy Hispanic gentleman presented for consultation after being found to have prostate cancer on TRUS-guided biopsy by an outside provider in July 2017. Biopsy revealed Gleason 4 + 4 = 8 adenocarcinoma of the prostate involving 50% to 90% of 3 of 12 cores, Gleason 4 + 3 = 7 adenocarcinoma involving 20% to 85% of 4 of 12 cores, and high-grade prostatic intraepithelial neoplasia. He was referred to an outside hospital for surgical removal of his prostate. However, given his history of suprapubic prostatectomy for BPH in Nicaragua more than a decade prior, he was advised to undergo radiation therapy. The patient approached us for second opinion and underwent pelvic MRI, bone scan, and cystoscopy for further characterization of his disease. MRI revealed diffuse heterogeneous postcontrast enhancement of the prostate gland with no discrete mass. There was no abnormal lymph node enhancement and bone scan was negative for metastatic disease as well. On cystoscopy, the patient was noted to have mild prostatic enlargement with a defect from his prior simple prostatectomy as well as a scar at the dome of the bladder, suggesting prior transvesical prostatectomy. Preoperative prostate specific antigen (PSA) was 9.2 and sexual health inventory for men (SHIM) score was 23.\nOur patient underwent bilateral nerve sparing RALP in September 2017. Total operative time was 76 minutes. Blood loss was ∼25 mL. We used a transperitoneal four-arm approach with a 0° lens. Upon entry into the abdomen and colonic mobilization, care had to be taken while performing anterior bladder dissection because of adhesions present from the patient's prior open prostatectomy. After lysis of adhesions and dissection of the endopelvic fascia, the bladder neck was transected and the prostate was mobilized. A blunt posterior dissection was performed thereafter using PK scissors to minimize rectal damage. Dissecting planes at the bladder neck and the apex of the prostate were difficult and great caution had to be exercised to prevent positive margins or risk incontinence by not maintaining adequate urethral stump length. To that end, our modified maximal urethral length preservation (MULP) technique was employed to augment stump length. Bilateral neurovascular bundles were meticulously dissected away from the prostatic capsule using minimal thermal energy. Despite the inherently unfavorable anatomy, there were no operative complications and our patient was discharged on postoperative day 1 with an indwelling Foley catheter. He returned to clinic in 10 days for catheter removal. Pathology report returned back Gleason 3 + 3 = 6 adenocarcinoma of the prostate (downgraded) involving 25% of a 50 g prostate. Surgical margins and bilateral seminal vesicles were negative for cancer involvement. One pelvic lymph node was sampled and was negative for adenocarcinoma as well. Final staging of the disease was pT2pN0M0.\nIn December 2017, our patient returned for a 3-month follow-up appointment. At that time he endorsed perfect urinary continence with no pad or liner usage. In fact, he mentioned that he was fully continent as soon as 2 weeks after surgery. Our patient also stated that he was able to get and maintain erections strong enough for intercourse 1 month after surgery, with a postoperative SHIM score of 22. His PSA at the time of 3-month follow-up was <0.1 ng/mL. |
A 32-year-old Caucasian man, working as a professional welder, was admitted in our hospital in January 2004 after experiencing swelling and vague diffuse pain in his right shoulder for several weeks. The patient denied a history of trauma or previous fracture of the upper extremity in the humerus. His recent medical history was unremarkable and he denied any constitutional symptoms such as weight loss, fever, or malaise. He had been treated at our institution at the age of 11, in December 1982, due to an ossification on the proximal right humerus. The physical examination did not yield any evidence of either a palpable soft tissue mass or axillary adenopathy. The results of motor strength, distal pulses, and sensitive tests were normal. There was no alteration in the shoulder's range of motion and no other physically relevant findings.\nThe patient was pathologically diagnosed with a calcified hematoma with benign characteristics in November 1982. Both radiographs () and xerography () showed a regular calcified mass in the deltoid area that seemed to adhere to the bone surface but not fixed to the humerus, presenting a precise limit with the bone. According to the operative records, a round mass sized 2 cm × 1 cm was resected, but a small portion of the mass remained unresectable. The patient was followed up for 5 years after the operation, but no evidence of tumor growth or malignant changes were found. Therefore, the follow-up was stopped.\nIn January 2004, the patient sought a consultation for a growing mass with diffuse pain at the site of the previous surgery. Both plain radiographs and parosteal calcified images showed bony deformation (). Tc-99 Bone scintigraphic imaging showed an increased uptake of technetium-99m-methylene-diphosphonate in the proximal metaphyseal diaphyseal region of the right humerus but not in the rest of the skeleton (). Computerized tomography (CT) and magnetic resonance imaging (MRI) scans of the right upper extremity showed a large cortical lesion in the proximal diaphysis of the humerus with a significant cortical continuity and well-defined borders. No medullary extension was observed (). MRI revealed a hypodense mass with gadolinium uptake in all sequences. The posterior cortical involvement and a minimal medullary extension were also observed (). The results of further studies including chest radiograph, chest CT, complete blood cell count, and erythrocyte sedimentation rate were normal. The patient's liver and renal functions showed no significant alteration.\nBiopsies of the anteromedial and posterior exophytic lesions showed spindle-shaped fibroblastic cells with well-differentiated trabeculae of woven bone with an osteoblastic rim on its surface. The fibroblasts showed minimal cytological pleomorphism and small nucleoli. Focal islands of irregular primitive bone formation were visible between the lamellar trabeculae. No vascular invasion of the tumor was seen, and no dedifferentiated areas were identified (). Regarding the histological features, the lesion was identified as a grade I parosteal osteosarcoma of the humerus.\nA pathologist re-reviewed the tissue samples that had been examined in December 1982 and concluded that the initial lesion that had been diagnosed as calcified hematoma with benign characteristics should have been diagnosed as a POS (), which featured a high similarity with the current biopsy. Thus, 21 years after an incomplete resection of a POS, the patient showed a local recurrence with no systemic dissemination.\nA wide en bloc resection of the 2/3 proximal parts of the humerus was performed and a proximal humerus prosthesis (GRMS, Stryker®) was then implanted. On gross examination, the lesion was a firm exophytic polypoid bony mass attached to the cortex of the humerus by a broad base (). No adjuvant therapy was given because of the low-grade features of POS. During a 53-month follow-up after the tumor removal, the patient was free of symptoms, with no signs of tumor recurrences or metastasis (), and fully incorporated to a new professional activity. In terms of range of motion, the shoulder showed an abduction of 70° by tilting the scapula, an internal/external rotation of 75°/35°, and a flexion/extension of 80°/15°. |
A 51-year-old male presented to a private orthodontic practice for initial assessment. His medical history was unremarkable and he was not on any medication at that time. During intraoral examination, a 1 × 1 mm translucent nodule with a smooth round surface was noted. It was located in the attached gingiva between the lower left mandibular canine and first incisor. The nodule was asymptomatic, cystic in appearance, fluctuant, and noncompressible (). Pulp testing of his lower left canine and central incisor indicated normal pulp vitality. Radiographically, there was no finding suggestive of osseous involvement. The patient was unaware of the lesion prior to this assessment. Complementing his oral assessment, he also had bilateral agenesis of his lower lateral incisors and first premolars (). Based on the clinical and radiographic findings, the provisional diagnoses were salivary gland tumor or cystic lesion of the gingiva.\nDue to the size of the lesion, an excisional biopsy was performed under local anesthetic infiltration. An incision was made in the overlying mucosa and the lesion was completely dissected from the adjacent tissues. The specimen was fixed in 10% buffered formalin and sent to the Oral Pathology Laboratory of the São Leopoldo Mandic Dental Institute and Research Center for histopathologic examination. On macroscopic inspection, the material consisted of a nodular fragment of soft tissue, measuring 5 × 3 × 2 mm, with smooth surface, whitish coloration, and friable consistency. Histological sections revealed a cystic cavity lined by two or three layers of odontogenic epithelium (), exhibiting focal epithelial plates containing clear cells (). The histopathologic diagnosis was of a gingival cyst of the adult.\nThe sutures were removed 1 week postoperatively and the patient reported minimal discomfort. After one month, the patient returned for follow-up when complete resolution of the lesion was observed. The patient is currently under regular follow-up with satisfactory healing and no relapse for over 15 months. Written consent was obtained from the patient for publication of the images relating to his case. |
A 12-year-old boy was referred to pediatric clinic with abdominal pain lasting for one month. He was also complaining from nausea and vomiting during the week prior to his referral. On physical examination a huge abdominal mass mainly in the right side of the abdomen was detected. Abdominal ultrasonography showed a large solid/cystic, fluid filled mass measuring 17 × 17 cm in upper abdomen assumed to be originated from right kidney. In computed tomography with contrast the right kidney was not visualized, but a huge heterogeneous cystic, septated mass measuring 12 × 16 cm in the anatomic area of right kidney, extending upwardly to the diaphragm and liver and anteriorly to the pancreas, was noticed. Absence of right kidney was consistent with its agenesis. The patient was scheduled for laparotomy through which a huge tumoral mass with attachments to the diaphragm was removed. However, neither the right kidney nor the ureter was found during the operation. Macroscopic examination of the mass showed lobulated fragments of spherical tissues weighing about 630 gr with gelatinous and solid, hemorrhagic areas. Microscopically, a neoplasm consisting of serpentine wavy spindle cells with low cellularity in a myxoid stroma transforming to the hypercellular pleomorphic areas composed of atypical round to spindle cells with hyperchromatic, pleomorphic nuclei showing about more than 10 mitotic figures in 10 HPF was observed. The findings were suggestive of neurofibroma transforming to malignant peripheral nerve sheath tumor (MPNST), while no evidence of renal tissues was observed and margins of tissues were free of any tumoral cells (Figures and ).\nImmunohistochemical markers including smooth muscle actin, cytokeratin, CD34, and c-Kit were all negative except for protein S-100 and CD-57 which were positive; the markers all related to nerves and nerve sheath, respectively (Figures and ). Chest computed tomography, bone marrow aspiration, and bone scan as part of systemic evaluation of the patient yielded normal findings. The patient received 8 courses of chemotherapy with carboplatin, doxorubicin, and ifosfamide for MPNST. Imaging study of the abdomen was normal during the treatment. He is in complete remission with no evidence of tumor recurrence after 3 years of follow-up. |
A 48-year-old woman suffered from life-threatening injuries to head and chest caused by six pistol shots fired at close range in an attempted homicide. When the emergency physician arrived on scene, the patient presented with oral bleeding from a smashed mandible () and with lacerated tongue causing mechanical obstruction of the airway. Rapid sequence induction was performed with 3 mg midazolam and 50 mg S-ketamine. Despite pharyngeal suction, visualizing the vocal cords with conventional direct laryngoscopy was impossible. The patient had spontaneous breathing and the tracheal tube then was inserted where air bubbles arose from the bleeding tissue. Proper position was confirmed by capnography and volume-controlled mechanical ventilation was started. Pharyngeal packing with gauze was performed to achieve hemostasis. Crystalloids were administered, and anesthesia was maintained with 0.2 mg fentanyl and 10 mg midazolam. In the emergency department, tracheostomy was established and mechanical ventilation was continued by pressure-controlled ventilation. After clinical and radiologic evaluation, under general anesthesia conservative debridement of all devitalized tissues was performed by a multidisciplinary team comprising an ear, nose and throat (ENT) surgeon and a craniofacial surgeon. Following the trajectories of the missiles, one bullet was removed from the lower jaw (, ) and mandibular osteosynthesis was performed via buccal approach using miniature plates and screws []. Ten units of packed red cells were administered during the operation. The patient was transferred to the intensive care unit (ICU) and pressure support ventilation was continued for six days. Repeated chest x rays revealed minimal mediastinal hematoma and mild pleural effusion on the left side but no signs of pulmonary infection. Ten days later, with an approach via the right mastoid process, the ENT surgeon removed the planum of the mastoid with the help of an 8 mm driller, the mastoid showed multiple fractures and was filled with blood. Extended mastoidectomy was carried out. The inferior part of the mastoid had been destroyed by the bullet that had entered the posterior skull base and was removed with the help of a hook. The dura and the sinus had been destroyed in this part, and a 5-cm-long thrombus was removed from the right transverse sinus. The surgeon also repaired the dural tear, and the neurosurgeon established a lumbar decompressive drainage. The facialis nerve was not interrupted but partly the nerve fibers were exposed. Even though there were hematomas and massive swelling in the extratemporal course of the facial nerve, nerve continuity had been preserved so that there was a good chance of spontaneous regeneration. Thus, there was no indication for an interposition nerve graft for the facial nerve (). The patient recovered and could be discharged from ICU on day 14. She survived without neurological damage except for Bell’s palsy on the right side. The patient underwent several reconstructive interventions by the plastic surgeon including cross-face operations for residual paresis of the facial nerve. |
A 38-year-old man presented to the Department of Orthopedic Oncology of the First Affiliated Hospital of Nanchang University (Nanchang, China) with the chief complaint of a painless mass in the right lower limb that developed over a period of 16 years. The patient appeared projections without apparent inducement at the medial cutaneous right leg which was about 2 × 2 cm in size, fixed block, soft, clear boundary, all directions and passive activities were unlimited at the beginning. Physical examination did not reveal any abnormalities, and the results of routine laboratory tests, such as erythrocyte sedimentation rate, were within the normal range. Three years after the mass was first recognized, a biopsy conducted at the Jian City People’s Hospital, Jiangxi, China showed signs of NF. However, the patient did not report any obvious discomfort, and therefore, the physicians did not recommend further examination but only regular follow-up instead. By early 2012, the patient started to limp. The symptom relieved after walking for a long period; however, the patient subsequently experienced numbness over the skin of the right lower extremity. On July 9, 2013, the patient underwent debridement at a local hospital, wherein the pathology results showed inflammatory cell infiltrations along with ulcers. The treatment did not illicit a favorable response and the tumors were not completely removed. Multiple tumors extending from the right leg to the popliteal fossa, buttocks, and groin were observed, with the largest tumor measuring approximately 10 × 12 cm; knee function was acceptable and no localized tenderness was detected. On April 30, 2014, the patient underwent multiple suction lumpectomies. Postoperative pathological findings indicated multiple schwannomas. However, the patient was hospitalized again owing to a right thigh and groin mass that enlarged; the patient also reported discomfort and weakness in the right calf, and underwent multisite lumpectomy for the right lower limb. The intraoperative findings included irregular shaped masses in the nerves of the right hip, iliac spine, and right lower limb. The masses had a clear boundary, with no obvious signs of invasions to the surrounding tissue.\nComputed tomography (CT) revealed several unequally sized lobulated and cystic-solid masses below the right popliteal muscle, with the largest mass measuring 3.7 × 4.5 cm; the masses exhibited a homogeneous density and a clear border (). No bone hyperplasia or destruction or hardening of the right femur and tibia and fibula were detected; however, enhancement was detected at the edge, a part of the calcaneal bone, and formation of depression. Thus, a diagnosis of borderline or poorly differentiated malignant fibromatosis was established by a professional radiologist.\nMagnetic resonance imaging (MRI) showed multiple nodules of varying sizes, mass shadows, and border finishing at the hip, iliac spine, and right tibia and fibula; T2-weighted imaging showed a slightly higher signal intensity (), and T1-weighted imaging showed a low signal intensity (). Fat suppression sequence images showed a slightly higher intensity signal; the masses had uneven signals with the largest detected in the right popliteal fossa measuring approximately 7.0 × 6.1 cm2. The tibia and fibula signals were uniform. The tumor borders were well-defined. The soft tissue surrounding the masses demonstrated normal signal intensity. No enlarged lymph nodes or distant metastases were identified. The MRI findings suggested multiple neurofibromas. Therefore, the patient was diagnosed with NF1 and indicated for surgery to resect the tumors.\nFor the surgery, the patient was placed in a supine position, with disinfected and paved sterile drapes arranged around the right lower limb, iliac spine, groin, and right hip to expose the surgical field following successful epidural anesthesia. First, a medial approach was used to access the inner border of the femoral condyle via an approximately 8 cm long incision. The skin, subcutaneous tissue, superficial fascia, deep fascia, and popliteus were incised to isolate the tumor, which was almost entirely surrounded by a soft tissue capsule. The same process was followed for the masses located at the other sites until all masses were completely excised. The intraoperative findings included multiple irregular but oval masses in the nerves of the right hip, iliac spine, groin, popliteal fossa, calf, and foot. The masses had a clear boundary and no obvious invasion of the surrounding tissue. The nerve membranes were incised to completely remove the tumors.\nGross pathological examination revealed approximately 30 soft, smooth-surfaced, dark gray, oval nodules of varying sizes, with the largest measuring 7.0 × 6.5 × 4.5 cm3. A partially cut section showed a capsule diameter of 0.8 - 1.8 cm containing clear liquid (). Upon hematoxylin and eosin staining, the membrane-like manifestations were found to be mature connective tissue. The tumor cells were deeply stained and appeared to be spindle-shaped with oval nuclei as well as wavy or irregular in shape, existing in bundles or a swirl or fence-like arrangement. Portions of the tumors showed bleeding, cystic degeneration, and necrosis (). The neurofibroma component was composed of proliferating spindle cells containing bland cigar-shaped nuclei with inconspicuous nucleoli and an eosinophilic cytoplasm (). Immunohistochemically, the spindle cells were diffusely positive for S-100 protein (3+) and the Ki-67 labeling index was < 2%. Moreover, the tumor was partially positive for CD34, CD56, and CR but negative for NF. Taken together, the surgical findings, tumor location, and nodule pathology resulted in a diagnosis of primary NF1. There was no evidence of NF transformation.\nThe patient was discharged without any complications 1 week after the total excision surgery. At the 6-month follow-up, the patient was completely free from pain during daily activities and demonstrated normal range of motion in the right hip and knee joint. No evidence of recurrence was noted. This study has been approved by the Ethical Advisory Committee of The First Affiliated Hospital of Nanchang University Medical School (Nanchang, Jiangxi, China), and informed consent was obtained from the patient for publication of this case report and any accompanying images. |
A 42-year-old Hispanic woman with no known past medical history presented to our emergency department with the chief complaint of increasing fatigue and dizziness for 2 weeks and mechanical fall at home on day of presentation.\nOn physical examination, she was afebrile (36.1 °C), blood pressure was 107/47 mmHg with a mean arterial pressure of 69 mmHg, heart rate was 87 beats per minute (bpm), respiratory rate was 17 breaths per minute, and oxygen saturation was 100% on room air. Her height was 143 cm and weight was 45 kg (body mass index 22). She was fully alert and oriented to person, place, time, and situation without any neurological deficits and was speaking in clear, full sentences. She had marked pallor with pale mucous membranes and conjunctiva. She had no palpable lymphadenopathy. She was breathing comfortably on room air and displayed no signs of shortness of breath. Her cardiac examination was notable for a grade 2 systolic flow murmur. Her abdominal examination was unremarkable without palpable masses. On musculoskeletal examination, her extremities were thin, and her fingernails demonstrated koilonychia (Fig. ). She had full strength in lower and upper extremities bilaterally, even though she required assistance with ambulation secondary to weakness and used a wheelchair for mobility for 2 weeks prior to admission. She declined a pelvic examination. No bleeding was noted in any part of her physical examination.\nShe was admitted directly to the intensive care unit after her hemoglobin was found to be critically low at 1.4 g/dL on two consecutive measurements with an unclear etiology of blood loss at the time of presentation. Note that no intravenous fluids were administered prior to obtaining the hemoglobin levels. Upon collecting further history from the patient, she revealed that she has had a lifetime history of extremely heavy menstrual periods: Since menarche at the age of 10 years when her periods started, she has been having irregular menstruation, with periods occurring every 2–3 weeks, sometimes more often. She bled heavily for the entire 5–7 day duration of her periods; she quantified soaking at least seven heavy flow pads each day with bright red blood as well as large-sized blood clots. Since the age of 30 years, her periods had also become increasingly heavier, with intermittent bleeding in between cycles, stating that lately she bled for “half of the month.” She denied any other sources of bleeding.\nInitial laboratory data are summarized in Table . Her hemoglobin (Hgb) level was critically low at 1.4 g/dL on arrival, with a low mean corpuscular volume (MCV) of < 50.0 fL. Hematocrit was also critically low at 5.8%. Red blood cell distribution width (RDW) was elevated to 34.5%, and absolute reticulocyte count was elevated to 31 × 109/L. Iron panel results were consistent with iron deficiency anemia, showing a low serum iron level of 9 μg/dL, elevated total iron-binding capacity (TIBC) of 441 μg/dL, low Fe Sat of 2%, and low ferritin of 4 ng/mL. Vitamin B12, folate, hemolysis labs [lactate dehydrogenase (LDH), haptoglobin, bilirubin], and disseminated intravascular coagulation (DIC) labs [prothrombin time (PT), partial thromboplastin time (PTT), fibrinogen, d-dimer] were all unremarkable. Platelet count was 232,000/mm3. Peripheral smear showed erythrocytes with marked microcytosis, anisocytosis, and hypochromia (Fig. ). Of note, the patient did have a positive indirect antiglobulin test (IAT); however, she denied any history of pregnancy, prior transfusions, intravenous drug use, or intravenous immunoglobulin (IVIG). Her direct antiglobulin test (DAT) was negative.\nA transvaginal ultrasound and endometrial biopsy were offered, but the patient declined. Instead, a computed tomography (CT) abdomen and pelvis with contrast was performed, which showed a 3.5-cm mass protruding into the endometrium, favored to represent an intracavitary submucosal leiomyoma (Fig. ). Aside from her abnormal uterine bleeding (AUB), the patient was without any other significant personal history, family history, or lab abnormalities to explain her severe anemia.\nThe patient’s presenting symptoms of fatigue and dizziness are common and nonspecific symptoms with a wide range of etiologies. Based on her physical presentation—overall well-appearing nature with normal vital signs—as well as the duration of her symptoms, we focused our investigation on chronic subacute causes of fatigue and dizziness rather than acute medical causes. We initially considered a range of chronic medical conditions from cardiopulmonary to endocrinologic, metabolic, malignancy, rheumatologic, and neurological conditions, especially given her reported history of fall. However, once the patient’s lab work revealed a significantly abnormal complete blood count and iron panel, the direction of our workup shifted towards evaluating hematologic causes.\nWith such a critically low Hgb on presentation (1.4 g/dL), we evaluated for potential sources of blood loss and wanted to first rule out emergent, dangerous causes: the patient’s physical examination and reported history did not elicit any concern for traumatic hemorrhage or common gastrointestinal bleeding. She denied recent or current pregnancy. Her CT scan of abdomen and pelvis was unremarkable for any pathology other than a uterine fibroid. The microcytic nature of her anemia pointed away from nutritional deficiencies, and she lacked any other medical comorbidities such as alcohol use disorder, liver disease, or history of substance use. There was also no personal or family history of autoimmune disorders, and the patient denied any history of gastrointestinal or extraintestinal signs and/or symptoms concerning for absorptive disorders such as celiac disease. We also eliminated hemolytic causes of anemia as hemolysis labs were all normal. We considered the possibility of inherited or acquired bleeding disorders, but the patient denied any prior signs or symptoms of bleeding diatheses in her or her family. The patient’s reported history of menometrorrhagia led to the likely cause of her significant microcytic anemia as chronic blood loss from menstruation leading to iron deficiency.\nOver the course of her 4-day hospital stay, she was transfused 5 units of packed red blood cells and received 2 g of intravenous iron dextran. Hematology and Gynecology were consulted, and the patient was administered a medroxyprogesterone (150 mg) intramuscular injection on hospital day 2. On hospital day 4, she was discharged home with follow-up plans. Her hemoglobin and hematocrit on discharge were 8.1 g/dL and 24.3%, respectively. Her symptoms of fatigue and dizziness had resolved, and she was back to her normal baseline ambulatory and activity level. |
A 43-year-old right-hand-dominant male patient, with a history of axial SpA, bilateral sacroiliitis with positive HLA-B27 and possible history of uveitis, was referred in January 2020 with bilateral digital ischaemia, worse on the right side. Six weeks before that, he presented under the rheumatology services with a milder degree of skin changes affecting his fingers in the form of blisters. His working diagnosis was Beurger’s disease, in the context of a history of heavy smoking. He was advised to stop smoking and received a prescription for glyceryl trinitrate patches and calcium channel blocker tablets, along with arrangements of outpatient prostaglandin infusion sessions. At that time, the patient was seen in the outpatient department; he declined to wait for vascular assessment and subsequently did not attend a magnetic resonance angiogram appointment. He reported gradual progression of ischaemia over the course of 6 weeks despite vasodilator infusions and medical treatment and was referred to the oncall vascular team. He reported no previous history of arm claudication, cardiovascular disease or exposure to vibration tools. He was unemployed and used to be a van driver.\nOn examination, it was noticed that there was difficulty in recording blood pressure in the right arm, with a profoundly ischaemic hand in the form of fixed mottling of the thumb, index and middle fingers, whereas the ulnar side was pale, insensate and with reduced motor function. He was noticed to have a scar on the volar aspect of his wrist and thenar eminence from previous laceration at the age of 14 years, which necessitated reconstructive surgery in a different hospital. He had an easily palpable subclavian pulse with suspected dilatation, but no pulses below. He reported improvement in the left-hand blisters, and the left side had a normal full complement of pulses. All serological markers of vasculitis were negative. An urgent CT angiogram revealed the presence of bilateral cervical ribs, compression of the right subclavian artery in the abduction position and occlusion of the right brachial artery owing to thrombosis, with normal upper limb circulation on the left side ().\nHence, he underwent an emergency right cervical rib resection and brachial thrombectomy. The latter was successful in restoring pulsatile flow to the elbow level, but attempts at smaller vessel thrombectomy were not successful owing to the presence of chronic organized thrombus. The patient had an uneventful recovery, with a palpable brachial pulse and restoration of monophasic Doppler signal in the ulnar artery at the wrist level. The irreversibly ischaemic radial side of his hand became fully demarcated dry necrosis, whilst the ulnar side was preserved (). He also underwent left cervical rib resection 6 weeks later and is currently awaiting an attempt at reconstructive surgery of his right hand using a groin flap. |
A 45-year-old Caucasian woman presented to the emergency department of the University Hospital of Heraklion, Crete after a fall from a ladder. The patient occupied in everyday activities as a house wife and a farmer. There were no concomitant injuries. Thorough clinical examination and radiological imaging revealed a depression fracture of the lateral tibia plateau which was classified as Schatzker III/AO 41-B2 []. Radiological examination by a computed tomography (CT) scan was performed [], which showed the details and personality of this specific fracture to assist in decision making.\nEstimated the clinical and radiological findings as well as the needs of the patient, surgical treatment was decided.\nTaking into account that the position of the fracture was at the most posterior area of the lateral plateau and the difficulties from open reduction of the articular surface, closed reduction via a minimal incision arthroscopically and fluoroscopy assisted using balloon kyphoplasty instrumentation was performed []. After reduction of the articular surface confirmed by arthroscopy the bone defect was filled with calcium phosphate via injection []. The fracture was considered a stable one and no further support and fixation was needed.\nInitial immobilization was obtained with a log leg cast for 2 days. Afterwards, continuous passive motion (CPM) was started with flexion and extension as tolerable. The patient started to walk with crutches and no weight bearing. A knee cast with goniometer was placed permitting knee exercises in flexion and extension. The patient was discharged from the hospital on the 5th postoperative day in a very good condition.\nThe patient was examined 6, 12 and 24 weeks after the operation regarding the movements of the joint and the muscle strength. A CT scan was ordered at the 6 week follow-up for a detailed evaluation and imaging of the tibial plateau []. The clinical outcome was excellent at the 6, 12 and 24 weeks follow-up with full range of motion of the knee joint. The patient didn’t report any issues regarding her rehabilitation. At the 12 week follow up partially weight bearing was started as tolerable. At the 24 week follow-up full weight bearing was already achieved by the patient with no reporting problems. |
A 26-year-old primigravida with an uneventful prenatal course delivered a male child at full term by elective Cesarean section. The indication of the Cesarean section was a transverse lie. APGAR scores and the birth weight were within normal limits. Following delivery, it was noted that movements of the left shoulder in the infant were restricted; however, a complete neurological examination did not reveal any evidence of the brachial plexus injury. The child was initially treated by observation and a gentle range of motion and was referred to our hospital which happens to be a tertiary center for advanced care. He was first seen by us at 4.5 months of age. The mother admitted decreased movements of the left shoulder since child’s birth with inability of the upper limb to be brought to the side of the body.\nA physical examination revealed posturing of the left shoulder in 80° of abduction, neutral flexion, and extension and 30° internal rotation []. Adduction was not possible. There was tightness of the deltoid muscle which probably accounted for the abduction deformity. Passive and active movements at the shoulder were predominantly scapulothoracic. The remainder of the examination of the upper extremity revealed normal findings.\nA plain AP radiograph revealed an anteroinferior dislocation of humeral head with an absent physis []. An MRI scan of the left shoulder [] confirmed the above-mentioned findings and also demonstrated a hypoplastic scapula.\nA tight contracted deltoid muscle was preventing reduction by closed manipulation under general anesthesia and therefore open reduction by the standard deltopectoral approach was carried out. The age of the child at that time was around 6 months. Intraoperative findings included anteroinferior dislocation of the head along with scarring of the deltoid muscle which probably accounted for its tightness. A proximal release of the deltoid muscle along with the subscapularis release was done. The inferior joint capsule was surprisingly found to be thickened and tight, and required excision in order to achieve reduction. The glenoid was slightly shallow but there were no other bony abnormalities. Once reduction was achieved, the shoulder was found to adduct fully []. However, the reduction was found to be slightly unstable with the joint being dislocatable at 60° of abduction. A shoulder spica was applied postoperatively in 30° abduction, neutral flexion, and neutral rotation.\nThe postoperative course was uneventful. Spica removal was done at 6 weeks and passive shoulder movements were started subsequently. At the last follow-up at the age of 1.5 years, the child was able to actively flex shoulder to 100° with a maximum abduction of 90 and an external rotation of 25°. The child is able to keep the upper limb by the side of the body. However, active overhead abduction is still not possible and deltoid muscle stimulation and physiotherapy are being continued. |
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