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A 27-year-old male was admitted to hospital emergency due to 4 h of chest pain in 13 January 2016. In fact, he had suffered from fatigue after activity for more than 10 days and felt chest tightness and chest pain for 4 days before admission. The patient, a taekwondo trainee, had a healthy body, and his family history was unremarkable. Emergency aortic computed tomographic (CT) scan showed a thickened aortic wall, bilateral pleural effusion and pericardial effusion (Figs. a and b). Cardiac colour ultrasound suggested aortic hematoma or dissection (Fig. ). On the basis of the condition and the results of auxiliary examination, the formation of aortic dissection should be considered. The next day, after excluding surgical contraindications, the patient was performed an emergency operation. Cardiac surgeons underwent ascending aortic replacement and aortic arch replacement. They developed postoperative comprehensive treatment measures, including anti-infection, adjustment of cardiac function, nutritional nerve and symptomatic supportive treatment, and the patient recovered well and was discharged after 15 days. The cause of aortic dissection was unclear, although the surgeons extracted arterial tissues and pericardial tissues and sent them for pathological examination. In fact, postoperative pathology of the aortic wall tissue showed aortitis, such as visible necrosis, granulation tissue hyperplasia and a large number of acute and chronic inflammatory cell infiltration (Figs. b, c and d). However, the surgeons and the pathologist at the time did not consider the cause of aortitis or aortic dissection due to GPA because of a lack of understanding of GPA-induced aortitis or aortic dissection. This event is one of the starting points of our study. We want to attract the attention of clinicians. GPA is also a common cause of aortitis and even aortic dissection.\nOn the first day of 2017, the patient was re-examined for aortic CT because of a 6-day fever and right chest pain. He showed a good prognosis in terms of aortic dissection after a review of aortic CT, but we found significantly increasing lesions in his lung, such as flake density increased shadow, less clear boundary, visible cavity and bronchial meteorology (Fig. c). The patient had no renal insufficiency and sinusitis but had eye damage with scleritis. The next day, the patient was readmitted to our hospital. At first, we considered pulmonary infection because of the combination of fever, haemogram, pulmonary shadow and cavitary lesions. Hence, we administered moxifloxacin. After 12 days, we reviewed chest CT again and found that the lung lesions became significantly heavier than before (Fig. d). Moreover, the patient still had intermittent fever, and the infection treatment was ineffective. At the same time, the patient developed conjunctivitis in the left eye and pain in the finger joints. Thus, we started to suspect pulmonary infection. We checked connective tissue disease-related indicators, such as ANCA, anti-nuclear antibody and immune indicators. The proteinase 3 (PR3)-ANCA (c-ANCA) level was 180 IU/mL, and the MPO-ANCA (p-ANCA) level was 10 IU/mL. Rheumatoid factor and anti-O experiments were positive, and anti-nuclear antibody spectrum was negative. We then performed a bronchoscopy, including brush biopsy, bronchoalveolar lavage and transbronchial lung biopsy. The pathological results suggested inflammatory cell infiltration, cellulose exudation and necrosis in clamped lung tissues (Fig. a). However, these pathological changes may be insufficient to diagnose GPA. We therefore carefully re-examined the pathological findings of the patient’s aorta and pericardium one year ago. Surgical biopsies obtained from the aorta and pericardium tissue showed that the epithelioid cells and multinucleated giant cells formed a granuloma (Fig. b), small vasculitis (Fig. c and Additional file : Figure S1 and Additional file : Figure S2) existed in the aorta and cellulose exudation and inflammatory granulation tissue hyperplasia were present in the pericardium (Fig. d).Combining with the two pathological results, clinical manifestations and laboratory tests, we invited a radiologist and a pathologist to perform a multidisciplinary discussion in the initial diagnostic assessment of the patient with suspected GPA. Finally, the diagnosis of GPA was established. Then, immunosuppressive therapy with i.v. steroids (methylprednisolone 40 mg twice daily) and cyclophosphamide 125 mg·day− 1 was initiated. The patient is currently followed up with the above treatment programs. We also adjusted the treatment program according to the patient’s disease progression. At present, the patient recovers well and is in stable condition. |
A 9-year-old boy was hospitalized in our cardiology department for repeated episodes of paroxysmal tachycardia at 180 bpm (Fig. ). Propranolol was ineffective to control arrhythmia. Because of frequent episodes of tachycardia the family accepted electrophysiological study and ablation.\nThe ECG during sinus rhythm showed no sign if pre-excitation. Echocardiography showed normal values for the age of 9 years (Fig. ).\nDuring the electrophysiological study we mechanically induced PSVT. The coronary sinus catheter confirmed an orthodromic reentrant tachycardia using a left lateral accessory pathway (Fig. ). Our protocol is to perform electrophysiological study with zero or near-zero irradiation in children under 14. We used the NAVX Ensite Velocity system provided by Saint Jude medical (Fig. ). The impedance-based system permits introduction of catheters through the femoral or jugular veins and inside the heart chambers without the use of fluoroscopy. The system delivers a small amplitude electrical signal between surface electrode patches. A catheter that is introduced in the electrical field has a clear position and can be visualized inside the heart chamber. The 3D geometry of the cardiac structure can be thus created. Using the patch surface electrodes from the abdomen and thigh we first created the image of the right common femoral vein, than the right iliac vein, inferior and superior vena cava. Using the previously created contours we placed the catheters inside the right atrium, right ventricle and coronary sinus.\nAfter catheter placement at the His level and right ventricular apex, we punctured the right femoral artery and introduced the ablation catheter retrogradely through the aortic valve at the level of the mitral ring. We reconstructed the descendant, ascendant aorta and the aortic arch and precisely localized the mitral annulus (Fig. ). During tachycardia we mapped the mitral ring and we found a spot where the ventricular and atrial electrograms were fused (the blue dot, Fig. ). Radiofrequency application at this spot stopped the tachycardia (Fig. ) which became uninducible. During ventricular stimulation there was no retrograde conduction towards the atrium. At 12-month follow-up the boy had no arrhythmia recurrence. No medication was needed. |
A 71-year-old man was referred to our department with a diagnosis of squamous cell carcinoma of the lung, clinical stage IA. Computed tomography showed a pulmonary tumor 2 cm in diameter in the left lower lobe and a right aortic arch (Figure\n, A and B). Three-dimensional chest computed tomography revealed that three arteries originated from the ascending aorta in order of the left common carotid artery, the right common carotid artery, and the right subclavian artery (Figure\n, C). Moreover, it showed that the left subclavian artery branched from the descending aorta and that the origin of the left subclavian artery had dilated into a cystic form, known as a Kommerell diverticulum (Figure\n, D). There were no abnormalities of the pulmonary arteries, veins, or bronchi (Figure\n, E). The lengths of the left and right bronchi were 4.8 and 1.2 cm, respectively. It is normal size for adult male. The anatomy of the brachiocephalic veins and the superior vena cava was normal. They located anterior to the trachea and posterior to the ascending aorta. The azygos vein drained into the superior vena cava as usual. Echocardiography revealed no cardiac abnormalities.\nThe patient underwent a VATS left lower lobectomy and mediastinal lymph node dissection. All procedures were performed with 3-cm anterior and 2-cm posterior access ports in the 5th and 6th intercostal spaces without rib splitting. There was no aortic arch or descending aorta in the left thoracic cavity, but the esophagus. There was no anomaly in the location and branching of the pulmonary arteries, veins, bronchi, and the lobulation of the lungs. A left lower lobectomy was performed in the usual manner. The upper mediastinal lymph node dissection followed the lower mediastinal dissection. First, we identified the vagus nerve at the level of the left pulmonary artery. When we proceeded with the dissection in the cranial direction, the arterial ligament was found between the left subclavian artery and the left pulmonary artery. It measured 4.3 cm length and 0.4 cm in width. The RLN, which branched from the vagus nerve, was recurrent around the left subclavian artery (Figure\n, A and B). A Kommerell diverticulum was found at the origin of the left subclavian artery. The patient was discharged without any complications such as hoarseness. |
A 4-year-old previously healthy boy had several (seven) episodes of colicky abdominal pain and loose stools over ten months. These episodes were never associated with fever but have had loss of appetite and infrequent vomiting. He had perianal discomfort both in the day and night. His mother had observed the passage of live worms with faeces on all these occasions, and she emphasized that it was more than 500 in number. During this period, no one else in the family had an illness similar to food poisoning, gastroenteritis, or worm infestation.\nHe was first seen by a general practitioner and treated for a common intestinal helminthiasis with mebendazole 100 mg twice daily for three days without obtaining laboratory evidence. After that, the child had been seen five times by general practitioners before the first author saw him. Stool investigations were done on two occasions and were reported as normal. It was not easy to get the treatment history due to retrograde exploration. The child had been treated with various anthelmintics and antibiotics. His mother could recall that he had been given both anthelmintics and antibiotics in a combination in few occasions. He had been treated with mebendazole 100 mg twice daily for three days in two occasions, albendazole 400 mg single dose in two occasions, and once treated by two tablets of pyrantel pamoate (250 mg). Whenever those episodes were predicted as food poisoning or infective gastroenteritis, the child had received either cephalexin or metronidazole along with zinc and probiotics for 3–5 days. Usually, the child became symptom-free in 6-7 days regardless of the treatment modality, but the reappearance of the symptoms in few weeks were inevitable.\nThe first author has seen the child in his sixth episode. The child complained of a gush of offensive loose stools with worms and abdominal cramps. A sample of stool has been sent for amoeba, ova, and cysts but became negative. Fecal culture was negative for bacteria. However, the child was treated empirically for helminthic infection using 400 mg albendazole single dose and repeated the same dose of treatment after a week. All family members were treated with the same regimen and instructed to follow hygienic measures. The child was brought to the same clinic in few weeks due to reappearance of similar symptoms. This occasion, the faeces were frothy and greenish. The parents have taken all possible hygienic measures to prevent the recurrences by introducing regular hand washing, cutting nails short etc. However, it did not prevent him getting recurrences.\nHe was from a semiurban area. In his garden, there were many guava trees bearing fruits. He had the habit of eating ripe guava. Upon the authors' request, the parents collected a fecal sample and guava fruit with larvae (). The parents succeeded in taking video-clips of live worms on faeces and fruits.\nThe second author examined all samples at the Department of Parasitology, Faculty of Medicine, University of Ruhuna. Live specimens were found in faeces and fruits. Faeces was examined using saline, iodine wet mounts, and Kato-Katz. It was negative for amoeba, ova, and cysts.\nLive wormy specimens were submerged in hot water (∼95°C) for 30 seconds and then in 95% ethanol before examining them under a dissecting microscope. The average length of larvae was 7 mm.\nPictorial key of Centers for Disease Control and Prevention, USA, was referred for larval identification (). All specimens collected did not have a definite, hard, sclerotized head capsule. The body was smooth and had short spines but no long lateral processes. Posterior spiracles were not on peg-like tubercles (). Body was tapered posteriorly but did not extend into a tail-like process. Dissected posterior spiracle showed peritreme with three distinct slits which were positioned straight (). The dorsal arm of cephaloskeleton is longer than the ventral arm and posterior spiracles with incomplete peritreme (Figures and ).\nSpiracle slits point towards opening in peritreme (). Prothoracic spiracles had 15 openings (). Posterior spiracle button is indistinct, and wall of slits had swellings (). Tracheal trunks were not pigmented (). Larvae have been identified as Cochliomyia macellaria. All larvae collected from faeces and fruit were morphologically similar.\nThe parents were instructed to avoid possible exposure to eating fruits (guava) from their garden. The child was reviewed in 3 and 6 months. He was symptom-free, and larvae have ceased appearing in the faeces. |
A 66-year-old female with a history of mucinous adenocarcinoma of the cervix presented to the neurosurgery outpatient clinic for evaluation of a sellar mass found during workup of adrenal insufficiency and hypothyroidism. The patient did not have unusual headaches or vision problems. Three months prior to discovery of the sellar mass, she was diagnosed with stage IIb mucinous adenocarcinoma of the cervix and was treated with chemotherapy. At the time of neurosurgery clinic presentation, she was neurologically intact, including full visual fields. Laboratory work-up demonstrated pituitary insufficiency with central hypothyroidism.\nThe initial magnetic resonance imaging (MRI) revealed a 1.8 × 1.1 cm contrast-enhancing mass within the sella, with extension to the suprasellar cistern and optic chiasm abutment. Preoperative imaging obtained the following month in preparation for surgery demonstrated that the mass had grown to 2.2 × 1.5 cm ().\nThe patient underwent an endoscopic endonasal approach for resection of the intradural sellar mass. Intraoperative findings demonstrated a very firm, infiltrative, vascular mass with dense adherence to surrounding structures, including the dura, medical cavernous walls, and diaphragma. Intraoperative frozen section pathology was consistent with metastatic carcinoma. The tumor was debulked until normal appearing pituitary tissue was identified and the margin of tumor adherence to the diaphragma was reached. Postoperatively, the patient did well without new hormonal deficiencies or vision problems. A subtotal resection (>80%) was achieved (). Gross histology and immunohistochemical staining ultimately confirmed the diagnosis of metastatic mucinous adenocarcinoma of the cervix (Figures and ).\nThe patient's immediate postoperative course was unremarkable. Given the diagnosis and intraoperative/postoperative findings of subtotal resection, adjuvant chemoradiation therapy was encouraged but the patient refused additional treatment. She was discharged home two days after surgery. She developed decreased left eye visual acuity and ptosis one week after surgery. A CT of the head at this time did not show any intracranial hemorrhage and a repeat MRI showed new enhancement suggestive of tumor recurrence within the sellar and suprasellar regions. The patient was started on steroids but declined any further treatment, including repeat surgery. Her ophthalmic symptoms ultimately progressed to a complete left cranial nerve III palsy four weeks after surgery. Follow-up MR imaging at five weeks after surgery revealed significant progression of the tumor to 2.9 × 2.4 cm with significant suprasellar extension (). Though a computed tomography scan of the chest, abdomen, and pelvis at this time demonstrated no new neoplastic burden, a radiotracer bone scan demonstrated likely new metastatic lesions in the skull, bilateral humeri, bilateral acetabula, bilateral femurs, and the lumbosacral vertebrae. After further discussion with her gynecologic oncologist and radiation oncology, the patient again refused pursuing any further treatment, including palliative radiation or systemic therapies and elected to pursue home hospice. The patient died approximately two months after surgery. |
A 60-year-old diabetic man took one 50 mg tablet of Sildenafil in the evening for 2 consecutive days without any effects and he was unable to have intercourse. On the third day he discontinued antiglycaemic medications, took another 50 mg tablet and engaged in sexual activity. Sixteen hours later he noted sudden decrease of vision in both eyes with a pronounced worsening in the right eye. He was admitted in a nearby hospital where a CT brain and a chest radiography were performed. They were unremarkable as well as complete blood count and the erythrocyte sedimentation rate. His medical history was significant for noninsulin-dependent diabetes for 7 months and his medication was metformin. He was discharged from hospital with a new prescription of 100 mg daily aspirin.\nSeven days after the onset of symptoms, the patient was hospitalized at our department. Visual acuity was 20/63 right eye (OD) and 20/32 left eye (OS). On fundus examination, optic disc edema and peripapillary nerve fiber layer hemorrhages were disclosed in both eyes (). Serous macular detachment was present in OD () whereas peripapillary cotton wool spots were found in OS. No evidence of diabetic retinopathy was noted. Blood pressure was within normal limit during the admission period. Humphrey visual field testing showed superior altitudinal and central defects OD and inferior altitudinal defect OS (). A fluorescein angiogram showed late leakage in the optic disc of both eyes (). Symptoms of giant cell arteritis were not present and no relative afferent pupillary defect was detected. A diagnosis of bilateral NAION was made and he was treated for three days with methylprednisolone 1 g/d intravenously, followed by oral prednisone 75 mg/d. The prednisone dose was tapered and discontinued over one month. Two weeks after the last steroid i.v. administration, visual acuity had increased to 20/50 OD and 20/20 OS. Optic disc edema, sub-retinal fluid and serous macular detachment resolved in OD and optic disc edema improved in OS. No sign of dye leakage of the optic disc was found in eyes and visual field testing disclosed altitudinal defects in both eyes. Three months later, visual acuity was stable and the optic disc was pale in OD. |
In this report we present an interesting case of hypertrophic nonunion of humeral shaft fracture in a 14-year-old boy. In the first instance the X-ray picture appeared to mimic an enchondroma.\nHe was a fully independent, medically fit and well, right-handed, and a nonsmoker who presented to the outpatient department with a deformity of the left arm. He had history of a minor fall about 1.5 years back followed by pain in left arm. He had visited a local bonesetter who gave him a massage and local bandage. The bandage was removed after 3 weeks. The pain had settled; however the patient was unable to lift weight with his left upper limb and had complained of increasing deformity in the proximal arm. He also complained of difficulty to carry out daily activities with his left upper limb.\nOn examination, he had a good range of motion in his shoulder and had an intact neurovascular status. While examining his arm, a completely asymptomatic mass was readily palpable over the middle left humerus with a detectable motion in the midhumerus.\nPlain radiographs demonstrated a suspicious solitary, diaphyseal humeral lesion, sparing the cortices, and surrounding soft tissues with an abundance of bone formation (). The CT revealed a lesion with exuberant bone formation at the site of deformity ().\nThe patient was planned for surgery after preanesthetic checkup. Intraoperatively the site was exposed and a sample was collected for histopathological examination. The excess callus was trimmed, and after freshening of the lesion site and clearing of the marrow, an intramedullary Rush nail was passed with an entry point at the lesser tuberosity (). Postoperatively the patient had an intact neurovascular status.\nThe histopathology report was suggestive of callus, with features suggestive of osteoid formation, with no features of malignancy ().\nThe patient was given a U slab postoperatively for 14 days; on the 14th postoperative day the slab was removed and sutures were cut. The wound was healthy with no suture line redness or discharge. A shoulder spica was applied for 6 weeks thereafter. The plain radiograph taken after 8 weeks of surgery showed features of union. At the end of 8 weeks postoperatively the spica was removed and the patient was advised careful physiotherapy of the elbow and shoulder.\nWith regular follow-up and physiotherapy, at the end of 5 months of follow-up, the patient had a completely united fracture () with good range of motion at the shoulder and the elbow (). |
This report is of a 73-year-old male patient who presented to the surgical out patient department (OPD) with voiding difficulty for 8 months duration. He also gave history of a right groin swelling which gradually increased in size. He emphasized that he could empty his bladder completely, only if he pressed this swelling and swelling reduced in size on voiding but the swelling never disappeared completely. There was history suggestive of chronic obstructive pulmonary disease (COPD). There was no history of constipation. Patent underwent left herniorrhaphy about 15 years back. On examination, his general condition was unremarkable. His hernia was right sided, complete, direct inguinal, irreducible and was with no signs of inflammation []. With the age and voiding difficulty in consideration, benign prostatic hypertrophy (BPH) was made as the differential diagnosis. Also, BPH along with COPD was thought to have caused the hernia. As the patient could void without difficulty after manipulation of the hernial sac, BPH was ruled out. Rectal examination and prostate specific antigen (PSA) were normal.\nA retrograde cystogram was performed using water soluble contrast. It showed part of the urinary bladder in the pelvis and part in the hernial sac extending up to the base of scrotum with feeding tube seen in situ and conforming its wide mouth and continuity with bladder seen in suprapubic area. The cystogram was diagnostic of bladder herniation []. Patient was also subjected to ultrasonograpy to rule out BPH which was reported to be normal but ultrasonograpy showed continuity of groin swelling with bladder.\nOperative procedure included a right inguinal incision extended across the groin swelling medially. Urinary bladder formed the contents of the hernia []. The hernial sac not opened but urinary bladder was confirmed intra operatively by filling the bladder with normal saline with Foleys catheter in situ. The hernial sac containing urinary bladder was reduced. There were no intraoperative complications. No resection of the bladder was performed but only hernioplasty was carried out. The patient remained clinically asymptomatic, without voiding difficulty after surgery and recovered well. |
A 23-year-old female was admitted to the emergency room because of abdominal pain with dyspnea. Last year, she presented a renal arterial embolism requiring a thrombectomy. The exploration of this renal infarction uncovered a dilated cardiomyopathy, initially presumed of viral origin, with heart failure requiring an implantable cardioverter defibrillator. When she was urgently readmitted, a computerized tomography (CT) scan revealed acute cholecystitis and an antibiotic therapy was begun. Given the cardiological history and the presence of dyspnea with elevated BNP, she was transferred to the cardiology ward. There, she presented a sudden right hemiparesis the next day with right hemianopsia and aphasia. Since implantable cardioverter defibrillators contraindicate magnetic resonance imaging (MRI), an emergency CT scan was performed and showed an acute left carotid T occlusion and a long-standing right internal carotid occlusion (Fig. ). The patient was transferred to the stroke unit for systemic thrombolysis and thrombectomy. Throughout the procedure, middle cerebral artery recanalization was obtained, but a clot was dislodged in the ipsilateral anterior cerebral artery during the procedure (Fig. ). After a short hospitalization in the intensive care unit for respiratory difficulties due to acute pulmonary edema, the patient was readdressed to the stroke unit where the neurological examination was normal.\nSupposing a cardioembolic origin of the stroke, due to dilated cardiomyopathy, a heparin treatment was started. The remainder of the exhaustive etiological assessment was not contributory. No biological thrombophilia was found. Despite an effective anticoagulation (unfractionated heparin with anti-Xa activity in the target), the patient presented an embolic complication in the form of acute ischemia of the left lower limb which was successfully treated surgically. In addition, several episodes of sustained ventricular tachycardia requiring two internal electric shocks by the implantable defibrillator were recorded.\nAn echocardiographic evaluation showed a dilated left ventricular heart disease, high ventricular pressures with ejection fraction of 22%, an overall severe hypokinesia, and the presence of left ventricular crypts with a typical aspect of “spongy myocardium.” A diagnosis of left ventricular noncompaction cardiomyopathy (LVNC) was suspected and it was confirmed when the patient was transferred to the cardiology unit due to the results of the cardiac MRI and cardiac CT scan performed during her last year's hospitalization (Fig. ). In the cardiology ward, 9 episodes of ventricular tachycardia causing syncope were recorded. The hospital stay was complicated by cardiogenic shock, requiring the patient's transfer to the transplant center where a heart transplant was done. |
A 13-year-old male patient was referred to the Department of Paedodontics and Preventive Dentistry with the chief complaint of pain in the right side of the face, right-sided headache, and painful jaw opening for the last 5 months.\nThe patient gave a history of extraction of the maxillary deciduous second molar 5 months back, reportedly without the administration of local anesthesia. The parents reported that the child was left psychologically traumatized by the experience and that following the dental extraction, began suffering from continuous pain on the right side of the face. Two months after the tooth extraction, the parents noticed a brownish patch developing on the right cheek region of the patient. The patient also gave a history of difficulty in mouth opening since 3 months and reduced oral intake of food with subsequent weight loss of 4–5 kg during the period. The patient visited several local dentists and general medical practitioners for the management of the pain and was prescribed various drugs which included analgesics, muscle relaxants, broad spectrum antibiotics, and mouth washes but showed no improvement in symptoms. The parents also reported that the child started being chronically anxious and refused to go to school and withdrew from his social circle. Patient demonstrated tense cooperative behavior on presentation (Frankl's behavior rating: negative).\nExtraoral examination revealed hyperpigmentation of the skin overlying the right cheek region. The brown pigmented lesion measured about 4 cm × 3 cm in size, with no sinus tract opening or pus discharge. () There was tenderness over the right parotid and masseteric region. Examination of the TMJ revealed no abnormality. There were however no definite trigger zones. Intraoral examination of the patient revealed a permanent dentition, generalized extrinsic stains, and chronic generalized marginal gingivitis Ellis Class II fracture of 21. Tooth number 15 had erupted and no associated soft or hard tissue abnormalities were detected intraorally ().\nThe orthopantomogram revealed no discernible dental anomalies (). The child was referred to the Department of Radiodiagnosis and imaging for ultrasound of right cheek and Department of Neurology for neurological evaluation. As the brownish skin discoloration appeared to be superficial and dermatophytic in nature, the patient was referred to the Department of Dermatology for opinion. No sonological abnormality was detected on the ultrasound. The Department of Neurology provisionally diagnosed him to be suffering from pathology of the maxillary division of trigeminal nerve secondary to the dental extraction and prescribed Tegretol and Naproxen tablets, after which he reported some but not complete relief from the facial pain. The child also continued to be anxious and had difficulty in opening the mouth. The patient reported that despite being on pain medication, he continued to experience pain which was moderate to severe in intensity, continuous in nature, and radiated to the temporal region.\nBased on the history and clinical examination of the patient, a diagnosis of chronic atypical facial pain, psychogenic in origin, and pityriasis versicolor resulting in cutaneous hyperpigmentation was established.\nThe parents and the patient were given informed reassurance, a psychological technique involving empathic explanation of the disease status, thereby alleviating a great of anxiety. During the follow-up appointments, the patient received positive reinforcement and was managed with tender loving care (TLC) approach. Oral prophylaxis and L.C composite build-up for 21 was done. For the skin lesion, the patient was prescribed Ketoconazole Soap (2%) following the use of which there was complete resolution of the brownish pigmentation of the skin of the right cheek within a few days. With the resolution of the skin discoloration, the patient reported significant improvement in symptoms despite not taking any pain medication (Figs and ). |
A 70-year-old male was referred to our hospital complaining of left flank pain. At physical examination, nothing relevant was observed and laboratory findings were within normal limits. A CT examination of the abdomen and the pelvis was performed with a multidetector scanner, before and after contrast media administration. Portal phase images showed a large amount of solid tissue in the left perirenal space, infiltrating the renal capsule and the main renal vessels; the tissue did not show significant contrast enhancement. Similar findings were detected also in the right perirenal space (). CT images also revealed a partial stenosis of the common bile duct, with intrahepatic bile duct ectasia owing to hypervascular eccentric tissue (). Hence, a diagnostic integration with endoscopic retrograde cholangiopancreatography was performed to exclude an intraductal proliferation. A biopsy was also performed in the left perirenal space; the pathological samples were composed of connective and adipose tissues, revealing the histological features of a DT or abdominal fibromatosis and also showing immunohistochemical markers typical of muscular tissues, such as actine. The tumour was considered unresectable and medical therapy was started with tamoxifen (20 mg die–1); after an episode of thrombophlebitis, the patient asked to suspend tamoxifen and accepted a new therapeutic regimen (docetaxel 75 mg m−2 every 3 weeks); unfortunately, even this treatment was discontinued after only 4 weeks owing to neuropathy. A new CT examination was then performed to assess the results of the treatment. Unexpectedly, on venous phase images, the perirenal tissue showed a remarkable reduction on the left side and had almost disappeared on the right side (). The peribiliary tissue had equally decreased in size and thickness (). The treatment was then discontinued in accordance with the patient’s decision. 1 year later, both the left perirenal and peribiliary tissues demonstrated no progression and remained clinically stable on off-treatment; however, new tissue was visible surrounding the right renal pelvis and the calices (). The same therapeutic regimens (tamoxifen and docetaxel) were proposed to the patient based on the previous good response; unfortunately, the patient refused any treatment. |
A 62-year-old female with no past medical history presented to the emergency department after a one-month history of progressive left-sided numbness, weakness, and unsteady gait. She had also experienced multiple falls and bladder incontinence over the past two weeks. She complained of frequent throbbing headaches in the occipital area that were not relieved by analgesics. The patient denied fever, chills, confusion, visual changes, or seizures. She had not sought any medical attention prior to this point and was not on any medication. She was of Portuguese origin and was married with two adult children. She had previously worked as a salesperson. She had never smoked and had no history of illicit drug or alcohol use. She denied recent travel, sick contacts, or exposure to wildlife. Family history was unremarkable.\nOn examination, she was alert and oriented to person, time, and place. Some slight left-sided tongue deviation as well as decreased motor strength in the left upper and lower extremities was noted, including a mild drift of the left arm. Deep tendon reflexes were found to be normal. She had diminished light touch on the left side. Pupils were equal and reactive and extraocular movements were fully intact. Blood pressure was 145/75 mm Hg. Her lab work on admission was found to be normal, including a white blood cell count of 9000 cells/μL. Her electrocardiogram showed normal sinus rhythm. Computed tomography (CT) scan of the head outlined an area of diminished attenuation in the high right parietal lobe that was suspicious for edema. No midline shift or mass lesions were noted.\nMagnetic resonance imaging (MRI) was performed to better characterize the CT findings (). Multiple abnormal areas were seen throughout the periventricular and subcortical white matter of the bilateral cerebral hemispheres, including the right parietal lobe region. No enhancement was noted in these areas, making malignancy less likely. The ventricles were normal in size and configuration. Carotid Doppler ultrasounds did not show significant stenosis. A transesophageal echocardiogram showed a normal ejection fraction and no thrombi, with the presence of a patent foramen ovale with a right-to-left shunt. The patient's condition did not show any improvement over the following five days and a follow-up brain MRI performed at this time showed decreased perfusion of the right parietal area in comparison to the contralateral side. Based on her CT scan, MRI, echocardiogram, and neurological exam, acute on superimposed chronic infarcts was suspected and the patient was transferred to the inpatient rehabilitation unit ten days following admission and placed on an antiplatelet agent.\nDuring rehabilitation, her condition worsened. At day 15 of her hospitalization, she became unable to walk due to worsening weakness of her left lower extremity and experienced high fever. She also complained of occasional vertigo. Her thinking became disorganized with diminished attention. No rigidity, myoclonus, or cogwheeling was noted. The patient's leukocyte count was elevated to 18,900 cells/μL, with 67% lymphocytes. Urine and blood cultures returned negative. Anti-nuclear antibodies and rheumatoid factor were negative. HIV status was negative on ELISA and polymerase chain reaction (PCR) testing. A CT of the abdomen and chest revealed only mild hepatosplenomegaly. Follow-up brain MRI showed a 0.5 centimeter increase in the diameter of the right parietal lesion.\nDue to lack of clear diagnosis, biopsy of the right parietal lesion was performed, in addition to analysis of the cerebrospinal fluid (CSF). This showed demyelinating macrophages in addition to enlarged bizarre-shaped cells with nuclear inclusions that stained positive for antibodies against simian virus 40 (SV40). SV40 immunochemistry is known to cross-react with the JCV []. A diagnosis of PML was made based upon these findings. This was confirmed by positive PCR and in situ hybridization results for JCV from CSF samples were sent to the National Institute of Health. She was started on a mefloquine trial, which had previously shown some success in inhibiting JCV replication. Unfortunately, she did not show any improvement while on mefloquine and continued experiencing a decline in mental function over the following four months. She ultimately became comatose and died. |
A 49-year-old male patient visited our clinic in December 2015 with a chief complaint of swelling of gums in the lower jaw. An informed consent was obtained from the patient for taking a detailed case history and treatment planning.\nThe patient had been a smoker (4–5 cigarettes per day) for the past 20 years. He was also using alcohol frequently. He was hypertensive and was under medication (nifedipine) for the past 5 years. He was also under the care of the Nephrology unit of a local hospital for interstitial nephritis for the past 5 years.\nHis dental history recorded periodic oral prophylaxis visits. He stated that he had noticed a small swelling on the gums on the inner surface of the lower anterior teeth for 3 months. He visited a local dentist who prescribed an antibiotic ointment (Metronidazole gel) for topical application. However, the swelling continued to increase in size and so he decided to consult a specialist and reported to our clinic. The patient did not have any pain which was the reason for the delay in asking for a second opinion about the swelling.\nPeriodontal evaluation showed that he had chronic generalized periodontitis. The lingual gingiva of mandibular anterior teeth revealed an exophytic growth of size 4 cm × 3 cm, firm in consistency and sessile. The surface of the lesion was irregular, with areas of ulceration []. Deep periodontal pockets of 6–8 mm [lingual aspect] and mobility of mandibular anterior teeth [incisors] were recorded. Bleeding on probing was present.\nExtraoral examination revealed palpable, firm, mobile, and nodular submandibular lymph nodes on the left side. The patient was initially advised to take an intraoral periapical radiograph. Radiographic evaluation showed extensive bone loss in relation to tooth number 41, 31, 32, and 33 [].\nBased on the patient's history, clinical and radiographic evaluation a provisional diagnosis of pyogenic granuloma was made with a differential diagnosis of SCC.\nNonsurgical periodontal therapy was done and chlorhexidine mouthwash was prescribed twice daily for 1 week. However, no change in the gingival enlargement was observed at 1 week recall. The case was discussed with his consultant nephrologist and an excisional biopsy of the lesion was done.\nHistopathological examination showed hyperplastic hyperparakeratinized stratified squamous epithelium with features of dysplasia. The surface epithelium showed breach in the continuity of the basement membrane. The underlying connective tissue was densely collagenous and showed numerous islands of neoplastic epithelial cells and abundant keratin pearl formation. Moderate amount of chronic inflammatory infiltrate was also seen composed of lymphocytes, plasma cells, and neutrophils [Figure and ]. Based on these histopathological findings, a diagnosis of well differentiated SCC was made.\nA cone beam computed tomography scan was done to assess the extent of alveolar bone involvement. Severe bone loss was seen in relation to teeth number 31, 32, 33, and 41 extending up to the apical third giving a floating teeth appearance [].\nTo assess the extent of lymph node involvement an ultrasonogram of the head and neck was done. It revealed the presence of a small benign appearing subcutaneous nodule (4.6 mm × 3.1 mm) with central cystic degeneration in the right posterior triangle. A small submandibular lymph nodes measuring 2.8 mm in short axis and a few, small, level two, lymph nodes measuring 2–3 mm which appear reactive were noted on the left side. Echo texture of submandibular glands appeared normal. Bilateral thyroid gland appeared normal.\nThe patient was immediately referred to Regional Cancer Centre, Trivandrum (a comprehensive cancer hospital) for expert management. A wide excision of the lesion with margins was done followed by bilateral functional neck dissection of regional lymph nodes. This was followed by fractionated radiotherapy sessions. The patient is being followed up for the past 8 months and no signs of recurrence have been detected. |
In our hospital, a 52-year-old male patient was hospitalized who had paralyses of the upper and lower limbs, mutism, respiratory disorder, and dysphasia. The patient had entered a Korean oriental medical clinic with dizziness and dysarthria 5 months prior to the hospitalization. He showed a sudden progress of altered mental status, quadriplegia, for 3 days. Due to these symptoms, he was transferred to the ER at a general hospital. While hospitalized in the general hospital, in tests using Magnetic Resonance Imaging (MRI) scan and angiographical inspection he was diagnosed with infarctions of cerebella, pons, midbrain, corpus callsum, of the left occipital lobe and the right thalamus caused by bilateral vertebral and basilar artery occlusions. At ICU, he received acute phase treatments (e.g. tracheostomy and respirator, anticoagulation therapy, etc.). After these treatments, he was in a stable condition, receiving hospital treatments for approximately 4 months. In order to receive comprehensive rehabilitation therapy, he entered our hospital approximately 5 months later after he showed the previously mentioned symptoms. When showing these symptoms 5 years ago, he was diagnosed with diabetes and high blood pressure and he was then administered drugs continuously. He had no particular family history of these diseases.\nAfter he was admitted into our hospital, he received a neurological examination. The test showed that he was in a conscious state. However, his cognitive and perceptual abilities could not be determined due to the difficulty in communicating with him. The cranial nerve examination showed a voluntary minute tilt of the right side of his head. He could move his eyeballs in limited perpendicular and horizontal directions and blink his eyes. Also, bilateral facial paralyses were observed. He could not project his voice nor could he swallow anything. He could not move his tongue and the gag reflex did not occur.\nThe muscular strength of the upper and lower limbs measured by Manual Muscle Testing (MMT) is grade zero. The spinal reflex of the limbs increased. The muscle tone assessment showed that the right upper and lower limbs and the left limbs were in Steps 2 and 3 respectively. The patient's Korean modified Barthel index (K-MBI) was Grade 0/Grade 100 because he needed the carer's absolute help when performing all physical activities and activities of daily life.\nThe patient did not make an attempt to express himself. After he entered our hospital, he was trained to express "Yes" or "No" by using a certain number of eye blinks. At the Rehabilitation Center attached to our hospital, he received alternative communication training to express himself more concretely and actively, by using a software program related to the alternative input device.\nWe fitted glasses on the side of his left eye by attaching these glasses to SCATIR (Self-Calibrating Auditory Tone Infrared) Switch (TASH, Roseville USA), which detects an infrared reflection stimulated by physical activities because he could not perform activities other than eye blinks (). Further, Switch Interface Pro5.0 (Don Johnston, Illinois, USA) was used, which delivers the input signal sent by the switch, operated when the patient blinks his eyes, to software programs and NeoSpeech Yumi (NextUp Technologies LLC, North Carolina, USA), a voice output software program ().\nAccording to the situation, he received training in the position of sitting on a bed or a wheelchair 40 minutes a day for 3 weeks. During the first week, the software programs Evaluware (Tobii Assistive Technology, Massachusetts, USA), Northern Grid For Learning activity (Northern Grid, Tyne and Wear, UK), and Sim tech single switch software for preschoolers (Don Johnston, Illinois, USA), etc., were used to improve the accuracy and speed of eye blinks, (). This would encourage the patient to correctly click at regular time intervals (). For 2 weeks after the first week, he received training in the use of Clickey2.0 (Seong-Pil Kim, Seoul, Korea), which enables the relevant person not only to use all functions of a keyboard only by clicking but also to output the chosen expression (i.e. a target word or sentence automatically scanned after its input operation) into a voice ().\nThe three-week training enabled the patient to express simple words (e.g. "bada (sea)" and his name by the combination of words using functions of the keyboard. Further, it enabled him to deliver some of his ideas and feelings by outputting the selected sentences (e.g. "I love you," "When am I leaving the hospital?" etc.) into a voice with an audio glossary function. |
A 50-year-old male presented with complaints of chronic, intermittent abdominal pain. The patient had a 20-year history of mild-to-moderate amount of alcohol consumption. There was no history of jaundice, fever or weight loss. His laboratory tests revealed no significant abnormal findings. Serum amylase and lipase, and tumour markers [CA 19-9, carcinoembryonic antigen (CEA)] were within normal limits. The patient was subjected to a CT examination. On unenhanced CT image, a poorly defined soft tissue mass was seen in the pancreaticoduodenal (PD) groove with a hypodense cystic lesion within the mass (). On post-contrast study, the lesion showed minimal enhancement in the portal venous phase ( and ), but delayed imaging at 2–3 min showed mild persistent enhancement of the lesion compared with the pancreatic parenchyma that was consistent with scar tissue () and a non-enhancing cystic lesion within the lesion. The lesion showed poorly defined fat planes with the adjacent second part of the duodenum on its right side and pancreatic head on the left side. Mild thickening of the wall of the second part of the duodenum adjacent to the lesion with variable luminal narrowing was noted. The common bile and pancreatic ducts appeared grossly normal. The pancreatic body and tail were normal. In order to clearly delineate the ductal system and the periampullary region, and to know the extension of the lesion, the patient was also subjected to an MRI examination. Axial two-dimensional fast imaging employing steady-state acquisition () sequences showed soft tissue signal intensity lesion in the PD groove with a cystic lesion within the lesion and the aforementioned CT scan findings. MR cholangiopancreatography sequence () revealed smooth and regular tapering of the pancreatic and common bile ducts. The gallbladder was distended and the cystic duct was normal. Incidentally, a few simple cysts (Bosniak 1) were noted in both the kidneys. The patient was advised further surgical intervention but he refused and was managed with conservative treatment. At present, the patient is asymptomatic. |
A 28-year-old female with previously treated tubercular lymphadenitis presented with left flank pain and gross painless hematuria of 15 days duration. On evaluation with contrast-enhanced computed tomography (CECT) scan of the abdomen and pelvis there was a 4.4 cm × 2.9 cm × 4.4 cm heterogeneous enhancing mass in the interpolar region of left kidney []. She underwent left open radical nephrectomy via flank incision and the histopathology specimen revealed clear cell carcinoma Fuhrman grade II confined to the kidney (T1BN0M0). She recovered well from surgery and was on regular 3 monthly follow-up with clinical examination, serum chemistries and ultrasonography when she noticed gross painless hematuria 2 years after radical nephrectomy. CECT was normal and urine cytology was inconclusive. On cystoscopy bloody efflux was seen from left ureteric orifice and retrograde pyelogram and ureteroscopy identified a mass lesion in residual ureteric stump. Using a Gibson's incision the ureteric stump and cuff of bladder were excised and there was a 1 cm × 0.5 cm clear cell cancer Fuhrman grade II similar to primary RCC extending up to submucosa and deep muscles were free. One year after the resection, she presented with a 2 cm × 2 cm firm mobile nodule in left iliac fossa at the margin of Gibson incision []. Fine-needle aspiration cytology revealed clear cell cancer and it was widely excised and was localized to subcutaneous tissue. Final histopathology was clear cell cancer Fuhrman grade II similar to primary tumor. She was kept on close surveillance and was asymptomatic for 2 years when she again developed gross painless hematuria with passage of clots. CECT showed two small lesions on the posterior wall of urinary bladder as the only abnormality []. On cystoscopy, there was a solitary 1 cm × 1 cm pedunculated growth in midline over interureteric bar with overlying calcification, which was completely resected and deep muscle and random bladder biopsies taken. On histopathology, the tumor was RCC Fuhrman grade II, deep muscle was not involved and random bladder biopsies showed normal urothelium. Currently, 11 years postnephrectomy, with 3 histologically documented recurrences and 5 years after the last recurrence she is disease and symptom free with normal chest and abdominal computed tomography scan and continues on 1 yearly regular follow-up. |
A 14-year-old otherwise healthy Caucasian girl presented to the pediatric emergency department complaining of a swelling under her tongue. Although the swelling started insidiously approximately four weeks prior to presentation, the mass had expanded over a three-hour period. The patient was seen by her pediatrician a few weeks ago when the lesion was smaller; she was treated at that time with a course of oral antibiotics (the patient's mother could not recall which antibiotic was prescribed) for possible infection. Nevertheless, the lesion did not improve and the patient was referred to an otolaryngologist for further evaluation. However, because the lesion expanded rapidly, the patient presented acutely to the emergency department for evaluation.\nUpon arrival, the patient appeared anxious but in no distress. Prior to arrival, the patient reported she was in her normal state of health and ate a normal dinner. Shortly thereafter she noticed an increased fullness under her tongue in which the ranula expanded from about 0.5 cm in diameter to its current state of approximately 5 cm in diameter; moreover, the expanding mass was impairing her ability to swallow and speak. The patient noted the mass was painful which worsened when she tried to open her mouth.\nThe patient denied fever, chills, weight loss, diet changes, cold and flu-like symptoms, shortness of breath, chest or neck pain, or recent trauma to the area. The patient's past medical, surgical, family, and social history were noncontributory. The patient had stable vital signs and she was afebrile with a pulse oximetry reading of 99% in room air. On exam, the patient was noted to have a large bluish intraoral swelling under the tongue which was most prominent on the right side and extended to the left side of the floor of her mouth. Her tongue was elevated superiorly due of the size of the lesion and she has difficulty speaking clearly. There was swelling noted in the submandibular area which was fluctuant and tender to palpation. There was no erythema or induration of the neck and no cervical lymphadenopathy was appreciated.\nA computed tomography scan (CT) of the patient's face and neck with intravenous (IV) contrast revealed a large fluid collection in the right sublingual space () extending through the right mylohyoid muscle and crossing midline anterior to the left submandibular space measuring 7.1 cm × 3.3 cm × 2.8 cm (66 cm3). There was no enhancement with IV contrast; however compression of the trachea due to mass effect was evident ().\nThe patient was given methylprednisolone 125 mg IV, morphine 2 mg IV, and clindamycin 900 mg IV and admitted to the hospital out of concern for potential airway compromise as well as inability to maintain oral intake due to progressively worsening swelling of the oral mass. While still in the emergency department, the lesion continued to swell and the decision was made to drain the ranula at bedside. After initial needle aspiration, an incision and drainage was performed which produced approximately 80 mL of viscous fluid consistent in appearance with saliva. Fluid culture ultimately grew Haemophilus influenzae. The patient was started on a 7-day course of oral clindamycin and discharged home the next day without further sequelae.\nThe patient was later seen by an otolaryngologist who initially opted for conservative management; however, the lesion has since recurred and surgery or sclerotherapy is planned. |
A 27-year-old male was admitted to hospital emergency due to 4 h of chest pain in 13 January 2016. In fact, he had suffered from fatigue after activity for more than 10 days and felt chest tightness and chest pain for 4 days before admission. The patient, a taekwondo trainee, had a healthy body, and his family history was unremarkable. Emergency aortic computed tomographic (CT) scan showed a thickened aortic wall, bilateral pleural effusion and pericardial effusion (Figs. a and b). Cardiac colour ultrasound suggested aortic hematoma or dissection (Fig. ). On the basis of the condition and the results of auxiliary examination, the formation of aortic dissection should be considered. The next day, after excluding surgical contraindications, the patient was performed an emergency operation. Cardiac surgeons underwent ascending aortic replacement and aortic arch replacement. They developed postoperative comprehensive treatment measures, including anti-infection, adjustment of cardiac function, nutritional nerve and symptomatic supportive treatment, and the patient recovered well and was discharged after 15 days. The cause of aortic dissection was unclear, although the surgeons extracted arterial tissues and pericardial tissues and sent them for pathological examination. In fact, postoperative pathology of the aortic wall tissue showed aortitis, such as visible necrosis, granulation tissue hyperplasia and a large number of acute and chronic inflammatory cell infiltration (Figs. b, c and d). However, the surgeons and the pathologist at the time did not consider the cause of aortitis or aortic dissection due to GPA because of a lack of understanding of GPA-induced aortitis or aortic dissection. This event is one of the starting points of our study. We want to attract the attention of clinicians. GPA is also a common cause of aortitis and even aortic dissection.\nOn the first day of 2017, the patient was re-examined for aortic CT because of a 6-day fever and right chest pain. He showed a good prognosis in terms of aortic dissection after a review of aortic CT, but we found significantly increasing lesions in his lung, such as flake density increased shadow, less clear boundary, visible cavity and bronchial meteorology (Fig. c). The patient had no renal insufficiency and sinusitis but had eye damage with scleritis. The next day, the patient was readmitted to our hospital. At first, we considered pulmonary infection because of the combination of fever, haemogram, pulmonary shadow and cavitary lesions. Hence, we administered moxifloxacin. After 12 days, we reviewed chest CT again and found that the lung lesions became significantly heavier than before (Fig. d). Moreover, the patient still had intermittent fever, and the infection treatment was ineffective. At the same time, the patient developed conjunctivitis in the left eye and pain in the finger joints. Thus, we started to suspect pulmonary infection. We checked connective tissue disease-related indicators, such as ANCA, anti-nuclear antibody and immune indicators. The proteinase 3 (PR3)-ANCA (c-ANCA) level was 180 IU/mL, and the MPO-ANCA (p-ANCA) level was 10 IU/mL. Rheumatoid factor and anti-O experiments were positive, and anti-nuclear antibody spectrum was negative. We then performed a bronchoscopy, including brush biopsy, bronchoalveolar lavage and transbronchial lung biopsy. The pathological results suggested inflammatory cell infiltration, cellulose exudation and necrosis in clamped lung tissues (Fig. a). However, these pathological changes may be insufficient to diagnose GPA. We therefore carefully re-examined the pathological findings of the patient’s aorta and pericardium one year ago. Surgical biopsies obtained from the aorta and pericardium tissue showed that the epithelioid cells and multinucleated giant cells formed a granuloma (Fig. b), small vasculitis (Fig. c and Additional file : Figure S1 and Additional file : Figure S2) existed in the aorta and cellulose exudation and inflammatory granulation tissue hyperplasia were present in the pericardium (Fig. d).Combining with the two pathological results, clinical manifestations and laboratory tests, we invited a radiologist and a pathologist to perform a multidisciplinary discussion in the initial diagnostic assessment of the patient with suspected GPA. Finally, the diagnosis of GPA was established. Then, immunosuppressive therapy with i.v. steroids (methylprednisolone 40 mg twice daily) and cyclophosphamide 125 mg·day− 1 was initiated. The patient is currently followed up with the above treatment programs. We also adjusted the treatment program according to the patient’s disease progression. At present, the patient recovers well and is in stable condition. |
The patient is a 60-year-old male with a history of metastatic lung cancer to the lung, liver, and brain. The patient presented to an outside hospital's ER due to rapidly progressive right foot pain, swelling, and a black area on right lower extremity. About a week earlier, he had scratched the anterolateral part of his ankle while mowing his lawn. He subsequently worked on his pool which was known to be dirty with black mold. The patient was found to have a quarter-sized lesion on right lower extremity. Then he began feeling more sick and went to a local hospital. That same night, the patient was transferred to Moffitt Cancer Center from over 100 miles away, despite his blood pressure being in the 70s. On admission, the patient appeared to be in sepsis and was immediately transferred to ICU to start hemodynamic support. The patient's past medical history includes a progressive cancer despite frontline chemotherapy that he last received 2 months earlier. He was to be enrolled in a clinical trial when screening evaluation found a new brain metastasis and had since received whole-brain radiation therapy. Medications included dexamethasone 4 mg twice a day for the past 3 weeks and Xarelto for history of pulmonary embolus.\nThe patient was afebrile with a temperature of 97.8 F. Pulse was 89 and respiratory rate was 23. Of note, the patient was hypotensive with a blood pressure of 72/50. He was alert and oriented and in considerable distress. On physical exam, the patient was found to have warm extremities with bilateral pitting edema. His right foot was erythematous and tender on palpation. His wound had gotten worse as there was purplish discoloration in a 10 cm area with possible necrosis of skin. No crepitus was felt. Also, the patient had thrush and no lymphadenopathy with no other skin lesions noted. Lab results showed the following: negative urine nitrite and leukocyte esterase, WBC of 18.23 k/ul, neutro auto of 15.24 k/ul, prothrombin time of 17.9 s, APTT of 35.3 s, INR of 1.9, glucose of 47 mg/dl, BUN of 32 mg/dl, creatinine of 1.7 mg/dl, estimated GFR of 41 ml/min/1.73 m2, albumin of 2.5, total bilirubin of 4.9 mg/dl, alkaline phosphatase of 182 U/L, AST of 173 U/L, ALT of 130 U/L, and lactic acid of 4.8 mmol/L. His initial ABG after intubation showed pH of 7.08, pCO2 of 54, and pO2 of 62 on 100% O2. His chest X-ray showed what appeared to be acute respiratory distress syndrome.\nDuring initial resuscitation, the patient was treated with maximum levophed dose and systolic blood pressure in the low 100s was obtained. He then developed atrial flutter with heart rate in the 200s. After giving 5 mg of metoprolol and holding levophed, the patient converted to normal sinus rhythm. Systolic blood pressure again dropped into the 50s and the patient was again given levophed along with phenylephrine and vasopressin and blood pressure rose to the 70s. He needed to be intubated due to progressive hypoxia. The patient had an episode of aspiration with a 10-second period of O2 dropping to 30% during procedure. He eventually had successful intubation.\nFor empiric coverage, the patient received vancomycin, meropenem, and doxycycline. A diagnosis of septic shock with multiple organ failure secondary to necrotizing fasciitis was made and urgent surgery consult was placed. The patient was also found to be coagulopathic, in acute renal failure, acidotic, and in acute respiratory failure. Surgery agreed with the diagnosis and the patient was emergently sent to OR for irrigation and debridement, about 2 to 3 hours after transfer to our institution. In the OR, purulent material was found coming out from the fascia. The material was cultured. Full thickness skin necrosis was found throughout most of the anterolateral aspect of the leg. The entire skin was removed in excess of a 15 by 20 cm area with 1 cm of subcutaneous tissue. Thorough irrigation was done with over 6 liters of normal saline with bacitracin in two different compartments of the leg. Despite the extensive infection, the muscles appeared to be healthy. At the end of the procedure, the patient was stable and ultimately required less pressor drugs.\nDue to extensive coagulopathy, it was not deemed safe for the patient to go back to the OR, so surgery was unable to reevaluate wound the next day as planned. After correcting his coagulopathy, he was taken back to the OR on hospital day 4 for elevated lactic acid and possible ischemic bowel. During the procedure, the bowel appeared to be healthy; however, the liver was found to be replaced by tumor and physiologically the patient appeared cirrhotic. Wound cultures taken during initial debridement were found to grow nonlactose fermenting, oxidase-positive, Gram-negative rods. Cultures were later confirmed to grow Aeromonas hydrophila and Aeromonas caviae. The cultures showed sensitivity to ceftriaxone, ciprofloxacin, gentamicin, tobramycin, and piperacillin/tazobactam.\nDespite treatments, on hospital day 8, the patient developed severe subcutaneous emphysema with unclear cause. After discussion with the patient's family, the decision was made to perform terminal extubation, given the patient's underlying stage IV lung cancer and worsening clinical status. |
The case we present here is a 34-year-old female patient [] who was complaining about non-healing opening in the upper left front region and exposure of root of one tooth. The lesion started as a small swelling in the left maxillary anterior vestibular area in the region of canine eminence. The swelling ruptured and exuded the discharge. After few days, patient noticed the area where the swelling ruptured was not healing and instead, it was showing small aperture. The aperture went on increasing to the present size of 0.5 cm × 0.5 cm. Patient revealed the history of trauma in maxillary left front teeth region during field work 2 years back.\nExtraoral examination revealed a mild swelling in the left maxillary sinus area []. On intraoral examination [] oroantral communication was noticed in maxillary left anterior labial vestibule at mucogingival junction in the area of canine. The opening was around 0.5 cm × 0.5 cm in size exposing the apical third portion of root of canine. The dorsal surface of the tongue was coated with white leathery coat with central rhomboidal shape erythematous area []. No caries was detected in all maxillary left anterior teeth. On pulp vitality test maxillary left lateral incisor and canine was found to be non-vital.\nThe clinical diagnosis of OAF secondary to chronic periapical abscess in relation to maxillary left canine was established. The conventional approach was undertaken to close the fistula and access opening was created in maxillary left canine. Patient was followed up for observation of healing at closure of OAF and also to continue with root canal treatment of maxillary left canine. After 4 weeks of primary closure of OAF, still there were no signs of healing and instead the OAF was re-established as it was before commencing the treatment. Patient also complained of dull gnawing pain in the maxillary sinus area and more heaviness in the same region.\nLooking at non-healing chronic nature of OAF, the incisional biopsy from the margin of OAF was planned to seek histological changes in the region of OAF. On histopathology, the diagnosis of chronic maxillary sinusitis was given. Based on histopathological diagnosis, reclosure of OAF was planned. For chronic maxillary sinusitis antibiotics and nasal wash was prescribed. The patient was followed-up regularly to assess for healing at OAF and check for the status of chronic maxillary sinusitis. Patient reported with non-healing of OAF and even chronic maxillary sinusitis did not respond to treatment. This time to avoid surgical trauma, a cytological smear was prepared by swabbing the unhealed OAF.\nCytosmear stained with periodic acid Schiff stain, revealed a pale homogenous stroma, with abundant acute inflammatory cells. Numerous long thin filaments of candidal organism having budding yeast cells were seen scattered along with pseudohyphae []. Based on the cytological features, the diagnosis of OAF with superadded candidiasis was given. Patient finally received antifungal medication of nystatin suspensions followed by reclosure of oroantral communication. After about 5-6 weeks patient was completely all right and clinical healing was complete. There were no sign of candida organism on repeated cytological examination at the site of closure. |
A 35-year-old woman was referred with an 11-month history of gradually swelling of the right maxilla and buccal spaces began during pregnancy two years ago. Patient felt no facial pain or paresthesia.\nThere was no history of trauma to the facial bones, nasal obstruction, dental problem, epistaxis, drugs consumption or visual change.\nOn physical examination, facial asymmetry was found due to swelling of right maxilla without significant tenderness.\nNo other clinical or laboratory problems were found. Postpartum CT scan of the maxillofacial region revealed a large lytic expansile multi-septated mass along the right maxillary sinus and buccal region (60 × 52 × 45 mm) (). In this patient, surgical excision of maxillary lesion was performed and histopathology was compatible with a giant cell tumor (GCT) of bone. Biopsy revealed a benign process consisting of significant number of osteoclasts with little osteoblast cells and small component of fibrosis and osteoid formation without evidence of malignant features.\nFour months following surgery, gradual progressive swelling of the bed of tumor was recurred and revised pathological slices were compatible with GCT. Regarding patient recent paresthesia, repeated laboratory tests were performed. According to laboratory tests (elevation of serum calcium and parathyroid hormone), ultrasonography and radioisotope scan (Sestamibi) were considered. In ultrasonography, a 11 × 14 × 27 mm mixed hypo echo mass lesion was seen in posterior aspect of right thyroid lobe compatible with probable vascular parathyroid mass (). In sestamibi isotope scan, increased uptake in posterior aspect of right thyroid lobe suggested a right parathyroid adenoma or hyperplasia . Other bone survey was unremarkable.\nAfter diagnosis of parathyroid mass, surgical excision was performed. Pathological findings of the right parathyroid gland revealed an encapsulated mass with solid-cystic component. Microscopically, the mass had a nodular growth pattern. Cells organizing the mass had a uniform shape with rounded nucleoli and pale eosinophilic cytoplasm without evidence of fat within the cytoplasm (). Pathological and surgical findings were compatible and consistent with parathyroid hyperplasia.\nDiagnosis of brown tumor due to primary hyperparathyroidism related to parathyroid hyperplasia was achieved according to clinical and pathological findings. |
A 47-year-old female patient was referred to our emergency department where she presented with productive cough since 11 months, weight loss for 4 months, shortness of breath, and loss of appetite since last one month. In past history, she was treated as a case of pneumonia with empirical antibiotics at a nearby private teaching hospital where CXR revealed increased opacity. A CECT chest was done which showed left hilar lymphadenopathy with minimal left effusion with focal areas of consolidation (). As a result, empirical therapy was changed to broad spectrum antibiotics. She showed relative improvement for a period of 2 months, but then developed weakness of the left upper limb and lower limb with twitching over the face. MRI scan was done at the same private setup which revealed multiple rounded enhancing lesions scattered in the B/L cerebral and cerebellar hemispheres, midbrain, pons, and vermis. A repeat CECT chest done at our hospital showed solid mass in the left hilar region involving the left lower lobe. EBUS (endobronchial ultrasound) guided biopsy from the mediastinal lymph node and endobronchial lesion showed no evidence of malignancy. In view of the high clinical suspicion deterioration of the patient, radiotherapy was given for the space occupying lesions in the brain. As per metastatic workup, a bone scan was performed which showed increased osteoblastic activity in the left 8th and 9th rib anteriorly. A percutaneous lung biopsy was done which showed features suggestive of moderately differentiated squamous cell carcinoma. Chest X-ray was performed immediately after biopsy, and it revealed bilateral diffuse consolidation and cavitation (). However, sputum sample sent for KOH mount revealed weak, acid-fast, and filamentous bacilli resembling Nocardia spp. She was immediately started on trimethoprim-sulphamethoxazole, linezolid, and imipenem/cilastatin for suspected disseminated nocardiosis. But due to the deterioration of the type II respiratory failure, the condition of the patient worsened, and by the next day, she developed septic shock. She was put on a ventilator but succumbed to her illness despite the best of resuscitative efforts. A final diagnosis of disseminated nocardiosis was made. |
Mrs. S is a 28 year old married female who is an illiterate housewife. This patient came to our outpatient department with symptoms of decreased sleep and appetite and lack of interest in her usual household chores. The patient complained that her liver was "putrefying" and her heart was "altogether absent". She also reported that when she walked she could not feel her body and reported that her "stomach was missing". She was 6 months into a pregnancy with her first child.\nHer problems had started after she had left her joint family subsequent to frequent quarrels with her in-laws and had started to live along with her husband in a small rented accommodation. Initially she developed loss of sleep and palpitations and a feeling of loneliness which progressed to loss of interest in her surroundings and her daily household work. She began to have feelings of guilt about herself and felt that she had done something wrong in leaving the joint family. The patient's husband took her to a doctor who described her symptoms as 'vague', diagnosed her as having 'weakness' and prescribed multivitamin tablets. Over the next 6 weeks the patient's symptoms failed to improve. At that point, she consulted another doctor who, after an initial two weeks of unsuccessful treatment for her 'vague' abdominal complaints (missing stomach and putrefying liver), referred her for a surgical consultation. After examining the patient and carrying out various investigations, including ultrasonography and endoscopy, the surgeon continued the previous treatment and added an antibiotic for a week. Within the next 2 weeks the patient visited a faith healer and a practitioner of the Indian system of medicine. After this the patient consulted yet another doctor who referred her for a psychiatric consultation. In the meantime 3 months had passed since she had developed her symptoms. She had previously made an unsuccessful attempt at suicide by taking organophosphorus poison for which treatment included medications and observation. She had been advised to seek psychiatric consultation on discharge but had not complied. One month after the suicide attempt she had again thought of suicide but did not act on these thoughts because of her "fear of God".\nThe patient was dressed appropriately and was well groomed. Her speech was coherent and relevant. Her complaints included insomnia, a general loss of interest, lack of concentration and a feeling of hopelessness. She was found tohave depressed mood but would occasionally smile while answering questions related to her symptoms because the "doctors wouldn't believe her". She had partial insight into her symptoms. She was not concerned about her pregnancy and had ambivalent feelings of love and hate towards the unborn child. There was no disorder of content or possession of thought. Physical examination was unremarkable except the patient was anaemic with haemoglobin of 8.5 mg/dl. Other routine investigations, including thyroid function tests, were normal. A CT scan or an MRI was advised by a consultant to rule out any organic problem. The CT was not done because of her pregnancy and the MRI could not be done because of financial reasons. A DSM IV [] diagnosis of Major Depressive Disorder with Nihilistic Delusions was made.\nECT was suggested but the patient refused because of the social stigma associated with the procedure. Other possibilities were explored. Initially, the patient was reluctant to take drugs because of her pregnancy. After persistence and a few counselling sessions the patient agreed. The patient could not tolerate fluoxetine 20 mg because of persistent nausea. Escitalopram caused similar problems. Both drugs were discontinued within a week. Tricyclic antidepressants were not prescribed because of the side effects. The patient was put on mirtazapine 15 mg and haloperidol 5 mg. Both of these were increased to 30 mg and 10 mg, respectively, over the next five days. The patient showed remarkable recovery within the next 4 weeks. MADRAS [] scores came down to 18 from an initial score of 44. Over weeks four to eight the patient's mood and delusions improved. The improvement was sustained for four months and, at the time of writing, was in remission. |
A 27-year-old male patient presented at our hospital in September 2008. He was suffering from a sudden onset of headache that had awoken him from sleep and increased with eye movements. He also had vision loss in his left eye. A neurological examination identified only bilateral papilledema, and a visual field examination demonstrated bilateral (more prominent on the right side) peripheral narrowing. He had no known systemic disease and no drug use, except for amoxicillin one week prior to his presentation.\nAll tests performed to determine the symptom etiology were negative. His cranial magnetic resonance imaging (MRI) was within normal limits. Cranial MRI venography demonstrated that the left transverse sinus and sigmoid sinus were hypoplastic, and the right side was dominant (MRI venography) (). The patient did not benefit from medical treatment. He underwent daily lumbar punctures for one week. The opening cerebrospinal fluid (CSF) pressure was above 40 cm H2O. The patient's headaches improved, but his vision loss remained constant.\nThe patient was diagnosed with PTC. We took him into surgery and implanted a lumbo-peritoneal shunt in January 2009. The operation was carried out under general anesthesia in the left lateral decubitus position, with bilateral lower limbs in the bent position. The L2-3 intervertebral space was selected as the point of puncture, through which a 1-cm opening was made with a sharp knife. A modified spinal puncture needle was inserted 5-7 cm along the midline. When an obvious breakthrough feeling was noted, this indicated that the needle was penetrating the spinal dura mater, at which point the needle core was withdrawn until the needle had been inserted through the subarachnoid cavity, with CSF flowing. Meanwhile, the shunt tube was inserted 7-9 cm into the subarachnoid cavity through the spinal puncture needle, and when CSF flowed through the shunt tube, the spinal puncture needle was pulled out. The shunt tube was tunneled to 2 cm lateral to the umbilicus on the lower right abdominal wall. This was penetrated with the puncture needle towards the lower and inward direction. When an obvious breakthrough feeling was noted, the needle core was withdrawn and the abdominal end of the shunt tube was inserted 20 cm via the puncture needle. Sutures were applied to fix the shunt valve and shunt tube in place, which aided in the avoidance of shifting or movement. Finally, the incision was sutured. Four hours after the operation, he developed urinary and fecal incontinence with paraparesis (1/5). Lumbar MRI identified an intradural hematoma at the level of L2-L3 (, ), and the patient was taken back into surgery. Firstly, L2-L3 total laminectomy was performed. A dural defect was detected on the needle's entrance zone into the dura. Subsequently, the dura was opened. An arterial injury caused by the needle at the inner side of the dura was observed. The intradural hematoma was removed totally (, ). During removal of his shunt, a CSF fistula occurred, and dural repair was performed. The family members of the patient refused a second shunt implantation. Physical therapy was started because of the lower extremity weakness. Two months after the operation, the patient's muscle strength had increased to 3/5. A clean intermittent catheterization was performed for urinary incontinence. At the final neurological examination of the patient, he had light perception in his right eye, paraparesis (3/5) and urinary incontinence. |
A 73-year-old white man presented to our emergency department with a 3-day history of left lower extremity swelling and acute-onset shortness of breath. On evaluation, he was tachycardic with a pulse of 113, hypertensive with a systolic blood pressure of 130-170 mmHg, and demonstrated poor oxygen saturation of 81% on room air. He was supported with continuous positive airway pressure (CPAP) and supplemental oxygen while a computed tomography angiogram (CTA) was obtained, which revealed a saddle PE (Fig. ).\nTissue plasminogen activator (tPA) was administered and he was started on a heparin infusion and admitted to our intensive care unit (ICU) for management. He remained on the heparin infusion for 3 days, during which he continuously improved and was eventually weaned to 3 L oxygen via nasal cannula. On hospital day 2, he was transferred to intermediate level of care. Per hematology recommendations, he would have to be on indefinite anticoagulation due to the massive PE he had sustained, the source of which was a left lower extremity popliteal deep vein thrombosis (DVT). The plan was to transition him from the heparin infusion to enoxaparin twice per day with hematology follow-up in 1 month.\nOn the day of discharge, however, he had sudden onset of right leg numbness and weakness below the level of his hip. He had previously been working with physical therapy and had been able to walk 200 feet with the assistance of a walker during each session. A physical examination revealed decreased sensation to light touch, 2/5 strength in right hip flexion and right knee extension and flexion, and loss of right patellar reflex. Left leg physical examination was normal at that time.\nAn emergent head computed tomography (CT) scan was ordered due to concern for a possible stroke, and neurology was consulted. The head CT was negative for infarction or hemorrhage. Neurology was concerned for spinal cord infarction versus hematoma and recommended emergent magnetic resonance imaging (MRI) of his thoracic and lumbar spine. The MRI revealed a left psoas hematoma (Fig. ). A CT of his pelvis performed the same day also showed a right psoas and iliacus hematoma. Due to these findings, hematology recommended discontinuing enoxaparin and reverting to a low intensity heparin infusion, as well as placement of an inferior vena cava (IVC) filter. The following day his left leg began exhibiting the same symptoms as his right leg. There was concern regarding the risk of progressive and irreversible nerve damage due to compression if the hematomas were not promptly drained. IR was consulted and advised that if the drainage were to occur while our patient was on anticoagulation, the risk of rebleeding into the retroperitoneum would be high and potentially nullify any benefit from drainage. Our patient would also be at risk of hemodynamic instability if the current hematomas were acting as tamponades against further bleeding. An additional complicating factor was the risk of further thrombosis due to the presenting saddle PE. Hematology was consulted for recommendations on pausing anticoagulation, but they were hesitant to offer a timeframe as there was no established safe period to enable this type of procedure to take place. Eventually, a window period of pausing the heparin infusion for 3 hours pre-procedure and up to 6 hours post-procedure was decided upon in the event that our patient agreed to have the drainage performed.\nThroughout this sequence of events, our patient and his wife were aware of the plans and considerations on how to proceed. They were informed of the recommendations and concerns made by neurology, hematology, and IR, as well as the risks and benefits of intervention versus non-intervention.\nAfter speaking with his wife, our patient decided to undergo the procedure. The low intensity heparin infusion was stopped 3 hours beforehand and the IR team then performed drainage of the right retroperitoneal hematoma, placing two pigtail catheters in our patient’s right flank (Fig. ). In total, the hematoma was drained of 215 milliliters of blood, 10 milliliters of which were drained during the procedure itself. The left psoas hematoma was not found to be amenable to drainage.\nOur patient tolerated the procedure well, and the heparin infusion was restarted 6 hours after it was completed. A repeat neurological examination demonstrated improved lower extremity strength bilaterally as well as the return of sensation to light touch. Hip flexion improved to 3/5 bilaterally, and knee flexion and extension improved to 4/5 bilaterally. Deep tendon reflexes remained absent. Four days later, the pigtail catheters were removed. His recovery was complicated by anemia requiring blood transfusions totaling 4 units of packed red blood cells (PRBC). Other sources of potential bleeding were evaluated and not found. A repeat CT on hospital day 10 (Fig. ) showed a stable right-sided hematoma, and our patient did not experience any further neurologic deficits. He was transitioned again from the heparin infusion to enoxaparin after 3 more days. His hemoglobin and hematocrit remained stable. During this time, he worked with physical therapy, who recommended discharge to a skilled nursing facility where his strength began to improve somewhat. Follow-up was scheduled with neurology and hematology. On hospital day 18, he was safely discharged. |
A 59-year-old man with a past medical history of nonischemic cardiomyopathy who initially underwent OHT in 1994 (biatrial anastomosis) was referred for pacemaker lead revision. His posttransplant course had been complicated by transplant vasculopathy, and he ultimately required a second heart transplant in 2002 (bicaval anastomosis). He also developed ESRD and underwent deceased donor kidney transplantation in 2004. He developed ehrlichiosis in 2011 in addition to cryptococcal pneumonia and histoplasmosis requiring chronic treatment with antifungals. In 2013, he had syncope leading to a subarachnoid hemorrhage and was diagnosed with sinus node dysfunction in the setting of intermittent sinus bradycardia to less than 20 beats per minute. He underwent dual chamber pacemaker placement in 2013 (Medtronic ADDRL1) with a Medtronic 5076 lead in the ventricular position and a Medtronic 5592 lead placed in the right atrial appendage after an active fixation lead was deemed to be unstable.\nHe was admitted for volume overload three years later, and pacemaker interrogation revealed undersensing on the atrial channel due to a gradual P wave amplitude decrease from 4.7 mV at implant to ~0.4 mV, leading to asynchronous ventricular pacing and failure to recognize atrial arrhythmias. No change in lead position was detectable on chest X-ray. An atrial lead addition was planned. However, the left subclavian vein was occluded. He underwent extraction of the atrial lead to obtain venous access. A 12 French Spectranetics SLS II laser sheath was advanced over the lead, and minimal application of laser energy was used to free adhesions. Countertraction using a snare was also employed from the femoral vein. The lead was removed, and subclavian access was retained. A Medtronic 3830 lead was implanted in the right atrium. The patient tolerated the procedure well, and he had no complications within the next 30 days. However, he was admitted with cryptogenic encephalopathy two months later which was thought to be at least partially related to subclinical cirrhosis. He was ultimately discharged to inpatient hospice and died shortly thereafter. |
A 40-year-old male patient came to the hospital emergency after a gunshot lesion in the cervical region. He was conscious, hemodynamically stable, and without signs of active bleeding or cervical spine injuries. Physical examination showed significant edema in the region of the mandibular angle, trismus, restriction of mandibular movements, absence of rhinorrhea or epistaxis, and soft tissue injury compatible with the bullet entrance orifice in the right posterior cervical region without clinical signs of exit bullet orifice.\nComputed tomography showed a comminuted fracture of the coronary and mandibular right ascending branches associated with ipsilateral zygomatic-orbital fracture (Fig. ) and the presence of artifacts compatible with the firearm projectile, suggesting an upward trajectory toward the face (Figs. ,).\nAfter physical and imaging evaluation, vascular surgery and neurosurgery teams opted for conservative treatment. However, the maxillofacial surgery team indicated surgical removal of the bone fragments due to the restrictions of the mandibular movements and removal of the fragments of the projectile due to discomfort and superficialisation in the genic region.\nOn the third day after trauma, under general anesthesia, removal of the bone fragments was initiated by intraoral access in the ascending ramus of the mandible, which evolved intraoperatively with an intense arterial bleeding, incompatible with the surgical procedure. Local compression maneuvers were performed using compresses, attempts to pinch with instruments after local exploration and the use of hemostatics, but they were not enough to contain the bleeding. After failure, it was decided to submit the patient to angiography of the external carotid artery.\nThe examination was performed by percutaneous puncture of the right femoral artery and selective catheterization of the external carotid artery and internal maxillary artery, which verified the presence of an PA (Fig. ) with indication of emergency embolization procedure. Through the catheter, the embolization was performed from the installation of 02 micro-platinum springs until the complete arterial occlusion and consequent end of the blood flow of the PA (Fig. ). The selective angiography of the left internal maxillary artery was then performed in different projections to rule out another possible source of bleeding and the possibility of compensatory revascularization.\nThe patient was transferred and remained under observation for 12 hours in the intensive care unit. A new angiography was performed after 24 hours for control, confirming the complete resolution of the PA. The patient was submitted to a second surgery 72 hours after the hemorrhagic episode when a large part of the bone fragments were removed by intraoral access. Also the palpable and superficial portion of the projectile located in the genic region by infraorbital access was removed.\nThe patient was hemodynamically stable, with no complaints and was discharged after 48 hours, without postoperative bleeding recurrences. He had no more complications after 8 months of follow-up. |
The patient is a 12-month-old girl with a double outlet right ventricle (DORV), non-committed ventricular septal defects (perimembranous VSD and anterior muscular VSD), aorta (Ao) positioned to the right and posterior to the pulmonary artery (PA) and transposition physiology. The patient was initially palliated with an MPA band to restrict pulmonary blood flow at 16 days of age, followed by an atrial septostomy at 4 months due to low systemic oxygen saturation (SO2 75%). Six months later her case was presented at the medical multi-disciplinary team meeting for planning of further surgical procedures. The two main drawbacks for biventricular repair were the possibly undeveloped left ventricle, and the long distance between the aorta and the VSD, which rendered either a Rastelli or arterial switch procedure more difficult. These conclusions were formed through review of a cardiac MRI. Therefore, a cardiac CT was indicated in order to define the locations of the VSDs and their relationship with the great vessels such that a surgical approach could be established. 3D models were requested to gather extra insight into the cardiovascular anatomy, and particularly to complement the imaging data with regards to the position and size of the VSDs and the position of the coronary arteries. Review of the models enabled a better understanding of the anatomy of the patient and it was felt that a Rastelli was not a viable option due to the long distance between the Ao and the VSD. Therefore, an arterial switch procedure was chosen as a the first option. However, the latter was highly challenging due to the high risk of damaging the chordaes of the tricuspid valve while enlarging the VSD to connect the left ventricle to the PA.\nThe patient ultimately became more symptomatic for shortness of breath, and the saturation levels dropped from 85% to <75%, secondary to the progression of VSD restriction and an increase in LV pressures to suprasystemic levels. An additional model was made, aiming to emphasize the tricuspid apparatus (leaflets and chordaes) and their proximity with the VSDs. This helped to improve the spatial visualization of these structures, thereby pre-empting potential complications such as tricuspid valve damage or rupture of tricuspid cordes and enabled the planning of the potential strategies needed to decrease the risk of complications.\nDuring the subsequent operation, after opening the pulmonary artery, it was felt that the muscular VSD could be enlarged and, after extensive sub-pulmonary fibrotic and muscular resection, the VSD could be committed to the pulmonary outflow. Furthermore, even though the pre-operative discussion highlighted concerns about the position of the coronary arteries, it was felt that there was a potential for their reimplantation after a more aggressive mobilization. Therefore, an arterial switch operation was performed without the LeCompte maneuver. The muscular VSD was enlarged and baffled into the neo aortic root and extensive muscle resection was performed underneath the pulmonary valve. The very small perimembraneous VSD was closed with a single stich through the tricuspid valve. The postoperative echocardiography showed an excellent repair with no acceleration through both the left and right outflow tracts and the patient was discharged home after 8 days. The patient was followed up 1 month later and showed peripheral SO2 of 100%, was asymptomatic for breathlessness and there were no changes on the echocardiogram. |
A 70-year-old woman presented with metastatic colorectal adenocarcinoma. She had a primary hemicolectomy in 2013 with local recurrence and secondary surgery and liver metastases treated with radiofrequency ablation in 2016. A computed tomography (CT) scan of her chest revealed the progression of her disease via multiple lung metastases in March 2017. The patient had a history of deep vein thrombosis three months earlier (right calf) and received rivaroxaban with good clinical and ultrasound outcome. She was admitted to our hospital for TIVAP insertion and polychemotherapy initiation.\nSurgery was performed under local anesthesia (April 2017). Initially, the right subclavian blind cannulation by the surgeon was ineffective. An experienced anesthesiologist was invited into the operating room, but multiple ultrasound-guided cannulation attempts failed bilaterally. The patient was transferred to the radiology department, and a chest CT scan revealed subclavian vein hypoplasia (vessel diameter was up to 4 mm). We decided to use right common femoral venous entry and discussed the situation with the patient. The patient provided additional informed consent to proceed. A catheter was inserted via femoral access without complications. In the operating room, the venous catheter was replaced by tubing from the port system kit.\nCatheter tip positioning was performed using transabdominal ultrasound guidance (with a convex probe). The capsule was implanted subcutaneously on the front lateral side of the right hip. Postoperatively, a whole-body CT scan allowed us to assess the TIVAP position and identify possible complications. Technical success was confirmed, and the patient was discharged from the surgery department. Anticoagulation therapy with rivaroxaban was prolonged to avoid a thrombosis recurrence.\nAs a routine practice, we include all patients with implanted ports in the observational venous thromboembolism monitoring program. This patient visited our hospital for follow-up evaluations one, three, and six months after surgery (May, July, October 2017, respectively). A physical examination showed no signs of venous thrombosis or port system occlusion.\nVascular ultrasound B-mode and color Doppler examination were performed (Figures , , ). No ultrasound signs of thrombosis, fibrin deposits, catheter occlusion, or perforation were detected. A whole-body CT scan (Figure ) revealed adequate device position. The patient is currently receiving polychemotherapy in an outpatient setting, and the port system is in use weekly without difficulties (last control - October 2017, next visit estimated in April 2018). |
A 38-year-old female patient came with a chief complaint of difficulty standing up from squatting position since 2 years ago. The patient also suffered heaviness and numbness from her hips that radiated to both of her knees and ankles. The symptoms worsened steadily in the past 4 months with both lower limbs getting weaker. Laboratory results came out normal, with no signs of infection or positive tumor markers. Radiological examination showed no apparent abnormalities as well. An MRI was obtained, and a tumor mass in the intradural region level of T10–T12 was found (). At that time, the patient was offered surgery, but she chose to undergo treatment with a bone setter. Around two months later, the patient returned to hospital with profound weakness on her lower extremity. Her physical examination revealed paresis from her thigh on both lower extremities grade 1-2/5 power in left and right lower limbs, respectively. Increased patellar reflexes were found on both limbs. Another MRI was performed and showed that the mass had grown to lumbar vertebrae L2, accompanied with worsening of the neurological statuses and impaired sensibility, as well as defecating and urinating problems (). From the history, spinal manipulation procedure was performed by a bone setter, although no specific techniques were available for review.\nA surgical procedure was proposed for exploration and decompression to the patient. The operation started by opening the lamina on T10–T12 levels, followed with laminectomy and hemostatic procedure to stop the bleeding, until the dura was exposed (). A dense mass from T10 to T12 was palpable from the dura layer. After we exposed the lamina, we observed that the dura was tense from touch, and a solid mass underneath the dura was palpable from T10 to L2. Intradural tumor excision was performed by a sharp 3 mm incision in the midline of the dura and then continued with blunt dissection, opening the dura layer to caudal L2 and to cranial T10. Dura was then parted with stay thread until the vessel-rich tumor mass was exposed. The tumor mass was excised, and the surgical field was contaminated by blood from hematomas from tumor vessels (). Bleeding was found to originate from the anterior part and posterior part of the cord, no bleeding source from the cerebrospinal fluid nor the subarachnoid space. The tumor mass was successfully evacuated with fragments of hematomas and necrotic tissues. The dura layer was then closed with a continuous suture. The tumor mass was fixed and transported for histopathology examination ().\nAt the time of discharge, the patient did not regain the function on her lower extremities (1-2 out of 5 on neurological motor examination). After six months of follow-up, some improvement on her lower extremity function was noted. Motor strength was returned to 3-4 out of 5, and the patient was able to ambulate using a walker. No improvement of her bowel and bladder symptoms was noted. |
We report the case of a 54-year-old male patient with a recent history of polytrauma (1 month before), and was referred to our institute because of an extended perineal and left gluteal defect of 18 × 25 cm. Following the high energy trauma, the patient presented hemodynamic instability on a pelvis open book fracture requiring emergency external fixator of the pelvis and radiological embolization of the left iliac artery for life-threatening bleeding. This led to multiple abscess in the perineal/gluteal region due to necrotic tissues, which finally required an abdominoperineal amputation, and an important loss of perineal and left gluteal soft tissue coverage (). Furthermore, he developed a bilateral DVT at the lower limbs with pulmonary embolism requiring inferior cava filter and therapeutic anticoagulation.\nThe reconstructive plan included a pedicled anterolateral thigh (ALT) flap extended to vastus lateralis (VL) muscle to obtain more bulk to fill the defect and cover the exposed left ischium. At surgery, patient had a body mass index of 23 kg/m2. Preoperative blood test showed hemoglobin levels at 8.3 g/dL and thrombocytes at 426 × 109/L. Therapeutic anticoagulation (intravenous heparin) was stopped 6 hours prior to surgery with normal prothrombin time (100%), and international normalized ratio []. With the patient in supine position the composite ALT-VL flap was harvested as previously described []. After flap raise, to provide fully access to the perineal and gluteal region, the patient was then positioned in high lithotomy position (). The pedicled ALT flap was transposed under the sartorius and the rectus femoris muscle to cover the perineal and gluteal area, passing posteriorly to the superficial femoral artery. The procedure lasted in total 5 hours with at least 2 hours in lithotomy position. Blood loss estimated was less than 500 mL and blood pressure was maintained stable during the whole surgery.\nTwo hours after surgery a pale and cold lower limb was noticed with no Doppler pulse at the foot, while flap showed normal appearance and appropriate capillary refill, speaking for an arterial occlusion distally to the depart of the lateral circumflex femoral artery. An emergency angio-computed tomography scan was performed, showing an 8 cm thrombus at left femoral artery (). An emergency Fogarty procedure was necessary to remove the clot and re-establish optimal flow to the limb. Postoperative outcomes were otherwise uneventful with effective flap coverage and no limb sequelae. |
A 61-year-old Black female with no significant past medical history was referred to our clinic for further management of severe primary open-angle glaucoma in both eyes. Prior to presentation at our clinic, the patient was placed on maximum medical therapy (500 mg of acetazolamide orally twice a day, brimonidine 0.2%-timolol 0.5% twice a day and latanoprost 0.005% at bedtime). She had undergone selective laser trabeculoplasty 6 years earlier. She presented to her local eye care provider for complaints of decreased vision without pain. During this visit, she was noted to have IOP readings of 45 mm Hg in the right eye and 35 mm Hg in the left. Her best corrected vision was found to be counting fingers at 3 feet in the right eye and 20/25 in the left. She was urgently referred to our clinic but was unable to follow up until 3 weeks later due to transportation issues.\nOn examination, her best corrected visual acuity in her right eye had decreased to no light perception, and her left eye remained at 20/25. Both eyes showed significantly elevated IOP – 46 mm Hg in the right eye and 30 mm Hg in the left. A dilated ophthalmoscopic exam was normal except for cupping of the nerves. The cup-to-disc ratio was found to be 0.99 in the right eye and 0.95 in the left. Given the recent visual loss in the right eye, we hoped to restore vision by performing an anterior chamber paracentesis to lower her baseline IOP while also administering maximal aqueous suppression therapy. This would allow the pharmacological reduction of aqueous humor formation and maintenance of a lower IOP until further surgical intervention could be performed in the coming week.\nThe patient was currently taking acetazolamide 250 mg PO BID from the referring provider. The anterior chamber paracentesis was performed and IOP immediately dropped to 6 mm Hg in the right eye. A drop of brimonidine and dorzolamide-timolol was then applied 5 min apart. No more than 5 min postoperatively, several new intraretinal blot hemorrhages appeared in the periphery of the right eye (Fig. ). No preretinal or subretinal hemorrhages were noted. No hemorrhages were appreciated in the left eye. IOP increased to 15 mm Hg 30 min after the procedure. Visual acuity in the right eye remained at no light perception.\nOne week later, the patient underwent implantation of an Ahmed glaucoma valve in the right eye and ab interno trabeculotomy with a trabectome in the left eye. On postoperative day 1 (7 days after paracentesis), fundoscopy revealed unchanged intraretinal hemorrhages in the inferior mid-periphery of the right eye (Fig. ). Visual acuity improved to light perception in the right eye and remained at 20/25 in the left. Eight weeks postoperatively, she developed a hypertensive phase and underwent cyclophotocoagulation. She developed choroidal effusions following this intervention. Twelve weeks postoperatively, peripheral ophthalmoscopic evaluation was repeated and the patient's choroidal detachments had resolved. Additionally, the retina was found to be normal without hemorrhage. More than 2 years following this presentation, the patient has undergone multiple surgeries but her vision has improved to finger counting with eccentric fixation. |
A 79-year-old man underwent ascending aortic replacement (AAR) because of an acute type A aortic dissection. Two years after the AAR, enhanced computed tomography (CT) revealed residual aortic dissection with a patent false lumen. The diameter of the aortic arch and descending aorta was 60 mm and 50 mm, respectively. We thus performed a TAR with the FET technique (J Graft Open Stent Grafts®, Japan Lifeline Inc., Tokyo, Japan). The FET diameter was selected to be about 110%–120% of the true lumen diameter. This was calculated from the circumference of the true lumen at the level of the descending aorta and was designated as the distal landing position. We determined the insertion length of the FET according to the intraoperative measurement using transesophageal echocardiography that was set at the aortic valve level of the descending aorta, and we inserted 90 mm in this case.\nEnhanced CT after the FET technique exhibited the expansion of the true lumen and thrombosis of the false lumen at the level of the aortic valve of the descending aorta (). The FET was not positioned at the straight portion of the descending aorta (); however, and we performed CT every half year. Three years after the FET technique, he was admitted to our hospital due to back pain. Enhanced CT revealed a new intimal tear just distal to the FET and rupture of the false lumen of the descending aorta (). We diagnosed the rupture of the false lumen caused by dSINE and performed an emergency thoracic endovascular aortic repair (TEVAR). One week after the TEVAR, he had a high fever, white blood cell count, and C-reactive protein (CRP) level. Enhanced CT showed good expansion of the stent graft but free air inside the false lumen (). Esophagoscopy revealed a fistula 40 cm from the incisor, and AEF was diagnosed. The administration of intravenous broad-spectrum antibiotics was commenced, and we performed an esophagectomy through the right thoracotomy. Two months after the esophagectomy, we performed descending aortic replacement using a rifampicin-soaked Dacron graft.\nAn intravenous antibiotic agent was administered for 20 days after the surgery, and oral antibiotics were administered until the CRP was <0.2 mg/dL. The patient underwent esophageal reconstruction 2 months later, and his postoperative course was uneventful. |
A 56-year-old male complained of mild intermittent diplopia. He also had swelling in both eyelids and proptosis that started 2 years ago, but he had not taken any special treatment (). He did not experience any symptoms of dry eyes or dry mouth. Exophthalmometric measurements were 21 mm in the right eye and 19 mm in the left eye. No limitation of motion was found in all extraocular muscles. His visual acuity, intraocular pressure, and fundus exams were normal. In his past medical history confirmed by his electronic medical records, both submandibular glands were enlarged five years ago. A fine needle aspiration biopsy done by an otolaryngologist revealed atypical lymphoid hyperplasia and periductal fibrosis, similar to sclerosing sialadenitis, but insufficient to diagnose KT (). Despite not receiving any special treatment, currently there have been no definite changes of signs. Thyroid function and antinuclear antibody test scores were within normal limits. The level of serum IgG was elevated to 2,770 mg/dL. Other serum levels of immunoglobulins, including IgA and IgM, were normal. Orbital computed tomography (CT) and magnetic resonance imaging (MRI) demonstrated bilateral lacrimal gland enlargement and thickening of extraocular muscles, including the right inferior rectus muscle and both lateral rectus muscles, with homogeneous enhancement (). Orbital lymphoma was highly suspected, so incisional biopsy of the right lacrimal mass was done ().\nLight microscopic examination of tissue stained with hematoxylin and eosin showed dense lymphocyte hyperplasia and lymphoid follicles. In the center of the hyperplastic lymphoid follicles, atrophic lacrimal ducts and periductal sclerosis with an onion-skin appearance around the ducts were observed. An atherosclerotic vascular change was also observed in the mass. Application of immunohistochemial stains revealed that the hyperplastic lymphoid tissues were composed of polyclonal lymphoid cells, positive for CD 20 and CD 79a in the center of the follicle, and positive for CD 3 and CD45RO around the follicles. This indicates reactive lymphoid tissue without malignant process (). Immunostaining for IgG4 also revealed numerous IgG4-positive plasma cells (). The possibility of malignant lymphoma, including extranodal marginal zone B-cell lymphoma (MALT lymphoma), was ruled out. In light of overall clinical characteristics, together with histological and immunohistochmical results, KT was determined to be a reasonable diagnosis.\nThe patient was administered oral prednisolone 30 mg/day for 1 week with a dose taper over the course of 3 weeks. The size of the orbital lesion and submandibular gland started to decrease slowly and the intermittent diplopia disappeared. Four months later, however, the disease recurred with upper eyelid swelling. Systemic steroid therapy was restarted with a dose of oral prednisolone 1 mg/kg/day, which was gradually tapered over the course of four months. This disease showed good steroid response, but tended to recur after steroid medication stopped. A third attack of the disease appeared 8 months after the last steroid treatment. Again the patient took high-dose systemic steroid medication, and after completion of the steroid-tapering schedule was administered azathioprine, a low-dose immunosuppressive agent. During the 1-year period of follow-up there was no recurrence of disease with the use of low-dose immunosuppressants. |
A 19-year-old young man status post repair of transposition of the great arteries (TGA) had a coarctation repair using a subclavian flap procedure at 3 months of age. During a recent follow-up exam at 18 years of age, he was noted to have mild upper extremity hypertension, good femoral pulses, and a mild discrepancy in his blood pressures and pulses with right arm greater than the left. He was placed on enalapril for his hypertension. Cardiac catheterization showed findings consistent with previous repair of his TGA, dilated aortic root with aortic incompetence, and no residual coarctation. A year later, he developed gradual onset of dizziness that was exacerbated by the use of his left arm. The patient also developed headaches, blurring of vision, confusion, and an acute syncopal episode while walking to school, which prompted admission and work-up. On examination, a grade 2-3/6 diastolic murmur was noted. His blood pressure in the right arm (127/72) was higher than the left (113/69) and the right arm pulse was stronger than left arm. Neurologic exam at baseline was unremarkable. The patient reported that his dizziness improved when the left brachial artery was compressed.\nBrain MRI with angiography showed no acute changes. Chest CT angiogram [] demonstrated the patient's anatomy status post subclavian flap, with the proximal left subclavian artery dissected away from the aorta and the left vertebral arising distally from the subclavian. shows the patient's carotid artery Doppler ultrasound, which demonstrates retrograde flow in the left vertebral artery consistent with left subclavian steal and continued retrograde flow in the left vertebral artery with left arm exercise. The patient underwent a hyperemia test with no significant change in his systolic blood pressure (SBP) in right arm (119 mmHg to 118 mmHg). However, his SBP dropped from 107 mm Hg to 102 mmHg in the left arm. shows the patient's aortic angiogram demonstrating prograde flow across the right common carotid, right subclavian, right vertebral artery, and left common carotid and delayed retrograde filling of the left vertebral artery to the distal left subclavian artery. There was no proximal left subclavian artery noted consistent with his earlier left subclavian flap angioplasty for repair of coarctation.\nThe patient underwent left common carotid-to-left subclavian bypass surgery using a 8-mm Dacron graft. The patient did well with complete resolution of his neurological symptoms following surgery. CT angiography of the chest [] showed a patent bypass between the left common carotid and left subclavian arteries. |
A 39-year-old East Asian man visited our hospital with a 5-year history of progressive dysphagia. At the age of 22, the patient had sustained C5–6 complex fracture/dislocation and complete cervical spinal cord injury at the C7 level due to a motor vehicle accident. No intracranial injury had been recorded. A halo traction was applied on the first day of his hospitalization as a temporary fixation, but definitive treatment was delayed due to severe respiratory distress, which required mechanical ventilation. He underwent anterior C5–6 corpectomy and fusion with iliac crest bone autograft without instrumentation 23 days after the admission. No bone morphologic protein was used. During the initial hospital stay, he underwent tracheostomy because of prolonged respiratory distress due to associated injuries. The tracheostomy site was complicated with methicillin-resistant Staphylococcus aureus (MRSA) infection, which was treated with antibiotics and repeated debridement. Since the time of injury, total non-oral nutrition had been continued for over 3 months, because of frequent aspiration and pain during swallowing due to inflammation of the tracheostomy site. No barium swallowing test was performed during the initial hospitalization. After swallowing rehabilitation, the patient could swallow liquid and solid food without aspiration. However, 12 years later, his dysphagia relapsed and gradually progressed. At the time of his 17-year visit, the patient aspirated frequently when he swallowed liquids or solids, to the extent that self-suctioning from the previous tracheostomy site was frequently required.\nComputed tomography (CT) scans of the cervical spine revealed almost complete resorption of the bone graft and a posterior shifted esophagus. The injured spinal columns were fused via the posterior and remaining anterior parts of the vertebrae. A bony bridge of heterotopic ossification was observed between the right posterior part of the cricoid cartilage and the right anterior tubercle of the C5 vertebra (Fig. ). A barium swallow study demonstrated significant barium aspiration into the airway and no laryngeal elevation (Fig. ) (see Video, Supplemental Digital Content ).\nThe patient underwent resection of the synostosis; the standard Smith-Peterson approach was utilized through the previous surgical scar. The resection was performed using a high-speed bar and small chisels. The ipsilateral omohyoid muscle (OM) was detached from the hyoid cartilage and the flap was inserted between the vertebral bone and cricoid cartilage to prevent recurrence. A laryngeal suspension procedure [] was added by otorhinolaryngologists. After surgery, his dysphagia resolved and he could swallow liquid and solids without aspiration. A follow-up barium swallow on the 10th postoperative day demonstrated improved laryngeal elevation and no aspiration (Fig. ) (see Video, Supplemental Digital Content ). The patient has had no dysphagia or recurrence 5 years after the surgery. |
This subject is a male, 54 years old, who was hospitalized in 2009 due to a headache that was diagnosed as a space-occupying lesion of the cerebellum via MRI examination. He was treated with a ventriculoperitoneal shunt and postoperative radiotherapy. In February 2011, he enrolled in our hospital due to dysfunction of the lower extremities. Contrast-enhanced MRI examination showed two large cysts in the bilateral cerebellum. Visible nodules with enhanced signals were found in the one cyst with a smaller size. Additionally, multiple nodular abnormalities were also found in the left cerebellum. Adjacent brain sulci were changed or disappeared due to enhanced pressure. The fourth ventricle showed unclear borders from the surrounding structures. Cerebellar tumour resection was performed, and pathologic analysis showed hemangioblastoma in the cerebellum (Fig. ). On October 30, 2014, an enhanced CT examination showed that multiple renal cysts occurred in both kidneys with unclear borders, and the arterial phase was significantly enhanced with uneven density. The largest cyst was located in the left kidney, and its size was approximately 5.8 cm × 7.9 cm. Both kidneys showed low signal density due to multiple cystic lesions but partial calcification (Fig. ). Multiple round water-like density shadows were observed in liver cirrhosis, with sharp edges but no enhancements. Considering the complications of kidney cancer, polycystic kidney disease and multiple liver cysts, the subject did not undergo further examination but underwent symptomatic treatments after discharge. In August 2017, the subject was enrolled in our hospital again due to dizziness and severe weight loss. CT examination of the cranial and thoracoabdominal regions showed multiple lacunar infarctions in the basal ganglia, bilateral softening lesions in the cerebellar hemispheres, multiple space-occupying lesions in both kidneys, bilateral polycystic kidney disease, multiple hepatic cysts and pulmonary nodules (Fig. ). The patient’s father was dead, the cause of death was unknown, his mother was alive, and the patient was married and had a daughter.\nPhysical examination: blood pressure 116/79 mmHg, consciousness, apparent anaemia, speech dysfunction. The left side of the temporal region had a visible mass with a size of approximately 0.5 cm × 2 cm × 3 cm that was red, hard, and fixed, with surface ulceration and no pus. The left occipital had old surgical scars. Limb muscle strength was at level 4. Laboratory tests: haemoglobin 81 g/L, glutamyl transpeptidase 352 U/L, albumin 30.6 g/L, serum sodium 128 mM, and creatinine 91 mol/L. The levels of tumour biomarkers were in the normal range. The patient was initially diagnosed with renal carcinoma that metastasized to the lungs. The patient and his relatives did not agree to perform further pathological diagnosis by renal biopsy. After a comprehensive analysis, it was thought that the patient might have VHL syndrome. Genomic DNA was extracted from the patient’s peripheral blood leukocytes using a DNA extraction kit, and VHL gene coding sequence analysis was performed by direct sequencing of PCR-amplified products using Sanger sequencing technology with an Applied Biosystems 3500 Dx Series Genetic Analyzer (ThermoFisher Scientific, USA) at Beijing Shengguzhi Medical Laboratory (Beijing, China) following the manufacturer’s instructions. It was found that a deletion mutation (Fig. ) occurred in the patient’s VHL gene at the c530-536delGACTGGA region in exon 3. This deletion caused a change in the amino acid at position 177 (Arg-177, Fig. ). Based on the results of genetic tests, the patient was diagnosed with VHL syndrome with renal carcinoma, bilateral lung metastases, multiple nephrotic cysts and multiple cysts of the liver, cerebellar hemangioblastoma and cystadenoma of the head (Fig. ). The patient’s conditions were informed to his family members, and a family medical census was proposed but was ultimately declined. The application of vascular endothelial growth factor inhibitors to treat the disease was also refused. Other symptomatic and supportive treatments for anaemia and hypoproteinaemia were also declined. On August 26, 2017, CT examinations showed that the renal tumours and lung metastatic lesions increased compared with previous conditions. The patient discontinued treatments and died in 2018. |
An 18-month-old female child reported to our outpatient department services with complaints of progressively increasing occipital cystic swelling since birth. The child was born as a result of cesarean section at term of nonconsanguineous parents. There was no family history of any congenital malformation or neural tube defect. The swelling used to increase in size with crying and coughing. On examination, a giant occipital swelling larger than the child's head was noted with a positive transillumination test. The child had delayed motor and social milestones. Noncontrast computed tomography head demonstrated a small defect (19 mm × 9.4 mm) in the occipital bone with a large swelling in the occipital region [].\nMagnetic resonance imaging of the brain revealed a giant cystic CSF intensity swelling of size 12 cm × 11 cm × 8 cm in the occipital region communicating with a posterior fossa cyst through a small defect in the occipital bone suggestive of giant OMC. Large posterior fossa with torcular-lambdoid inversion, vermin hypoplasia, and a posterior fossa cyst communicating with the fourth ventricle along with dilation of all the ventricles was also noticed suggestive of DWS [Figures and ]. A neuroadiological diagnosis of DWS with a giant meningocele was made; however, screening of spine was unremarkable. No antenatal radiological tests were performed. All hematological investigations were normal.\nThe child was taken for surgery under general anesthesia in prone position with a plan of meningocele repair followed by ventriculoperitoneal shunt surgery. As the large size of meningocele was causing difficulty in intubation and positioning, meningocele repair was planned followed by shunt surgery. The meningocele sac was slowly decompressed by draining around 800 ml of clear CSF. A linear incision was given over the sac, and the dural margins were defined all around. The meningocele sac was separated from the occipital bone defect. Finally, the dura was incised, and the meningocele sac entered. No neural elements were seen inside the sac suggestive of meningocele. Intraoperatively, communication between meningocele and posterior fossa cyst and fourth ventricle was visualized. Meningocele sac along with redundant dura was excised, and dural edges were approximated to achieve a primary watertight closure. The preexisting small occipital bone defect was repaired using autologous split calvarial graft to provide a good cosmetic result. The redundant skin was also excised and closed esthetically in layers. However, the child developed hypothermia, so shunt surgery was deferred to the next day; however, an external ventricular drain was placed and the next morning the child underwent shunt surgery [Figures and ]. |
A 65-year-old male presented with a three month history of progressive left hip pain. His past medical history was noteworthy for a non-ischemic cardiomyopathy for which he underwent a heart transplant ten years prior to presentation. He had a history of osteoarthritis for which he had undergone bilateral hip replacements complicated by a left prosthetic hip infection with Cutibacterium acnes. His past history was also positive for rheumatoid arthritis, hypertension, and type 2 diabetes.\nThe first stage of his left hip revision after his first episode of prosthetic joint infection with C. acnes occurred pre-heart transplantation and he was treated with 6 weeks of vancomycin following hardware explantation. His second-stage hip revision occurred over a year later, following his heart transplant. His post-transplant course was otherwise uncomplicated, without any episodes of graft dysfunction, rejection, or infectious complications over the previous nine years. He was maintained on tacrolimus and mycophenolate mofetil since his transplant without recent corticosteroid exposure.\nWith regard to his present symptoms, along with the pain, he noted occasional night sweats. He reported receiving 2 hip injections for the pain (one into his hip joint and another into his trochanteric bursa, both on his left side) by his outpatient orthopedic doctor about 2 weeks prior to presentation that provided temporary relief. Sterile precautions were reportedly observed during in-clinic procedures. Following the injections, the patient noticed a progressive fluctuant area swelling of his left lateral thigh and hip. Aspiration of that area 4 days prior by his outpatient orthopedist yielded purulent-appearing fluid that was Gram stain negative for organisms, with no growth seen either on aerobic or anaerobic cultures. Three days prior to admission, he underwent hip arthrocentesis that showed cloudy fluid with 65,000 white blood cells per microliter and a differential of 95% polymorphonuclear neutrophils. Gram stain was negative, and no crystals were seen. Culture of the synovial fluid was later finalized as no growth. However, prosthetic joint infection was suspected and so the patient was admitted for surgery.\nThe patient was an active dog breeder (Yorkies, Maltese, and Morkies), with over 10 dogs at home at any one time, and had recently participated in the birth of several puppies. There was no history of illicit or injection drug use. He also noted being scratched on his legs by the dogs in the past, but could not recall any recent scratches or bites recently.\nOn admission, he was well appearing and comfortable at rest. All vital signs were normal and he was afebrile. He noted mild pain with passive and active movement of his left hip, however, he was able to ambulate. Oropharyngeal exam was unremarkable with good dentition. His bilateral shins had evidence of previously-healed scratches. On the lateral aspect of his thigh was a tender egg-sized swollen area of fluctuance and erythema, without increased warmth or obvious drainage or sinus tract.\nLaboratory evaluation was notable for a peripheral white blood cell count of 6300 per microliter. Erythrocyte sedimentation rate and C-reactive protein levels were 63 mm/hour and 5.4 mg/dL, respectively. Hip X-ray did not show loosening or disruption of his prosthesis. He did not receive any antibiotics prior to surgery. Two sets of pre-operative blood cultures were drawn and finalized as no growth. Medications at the time of presentation included methotrexate (2.5 mg daily), mycophenolate mofetil (250 mg twice daily), and tacrolimus (1 mg twice daily). His diabetes mellitus was well-controlled with dietary changes and his most recent hemoglobin A1c measurement was 6.2%.\nThe area of the suspected abscess the lateral thigh was incised and drained. The proximal prosthetic body, femoral head, and acetabular liner were all removed, the joint was irrigated and debrided, and hardware was replaced in a one-stage fashion. Vancomycin and tobramycin-containing beads were placed. Intraoperatively, frank purulence within the joint was not observed. Five separate tissue specimens were sent for bacterial, fungal, and mycobacterial culture. Post-operatively, vancomycin and ceftriaxone were begun empirically pending further culture data.\nApproximately 2-3 mm of each tissue submitted was embedded whole in fungal media. The remaining tissue from each site was ground in 1 mL Tryptic Soy Broth. Mycobacterial, fungal, and bacterial growth media were inoculated with ground tissue suspension. Gram, Acid Fast, and Calcofluor White stains were prepared from touch preps of intact tissue and ground tissue suspensions. All stains of tissue were negative for organisms. Bacterial cultures were incubated at 35 °C in a 5–10% CO2 incubator for aerobic cultures and in the BD BBL GasPak anaerobic pouch for anaerobic cultures. After four days of incubation, tissue cultures collected from the left hip (one of 5 total submitted) grew small, translucent, spreading colonies from the anaerobic blood agar incubated under anaerobic conditions. Gram stain of the colonies demonstrated spiral-shaped gram-negative rods (Fig. ). Matrix-assisted laser desorption time of flight (MALDI-TOF) mass spectrometry yielded and identification of Anaerobiospirillum succiniciproducens, which is currently unclaimed in the bioMerieux Vitek-MS database. Therefore, 16 s ribosomal RNA gene sequencing was performed, with the best matches using NCBI Blast Nucleotide Sequence Database to Anaerobiospirillum succiniciproduces and Anaerobiospirillum species with > 99% query coverage and identity. Ultimately, the laboratory report was released as “most closely resembles Anaerobiospirillum species; Identified by DNA sequencing.” 16S sequencing was not performed on any other samples including blood.\nMycophenolate was held temporarily in the setting of infection. Following identification of the organism, vancomycin was discontinued and ceftriaxone (2 g daily) was continued to complete 6 weeks total of antibiotic therapy. He tolerated treatment well to date, without evidence of recurrent infection.\nSaliva and perirectal anaerobic cultures of two of the patient’s dogs (both Maltese) were performed using flocked swabs submitted in anaerobic agar gel transport media, and cultured on standard anaerobic media. No organisms resembling Anaerobiospirillum spp. were isolated.\nFollow up 2 months later was unremarkable for relapse of infection, after completion of intravenous antibiotics,. |
A 61-year-old male patient presented to our institution with a complaint of pain in the right knee that had aggravated for the last three days. On the same knee, he underwent posterior cruciate ligament (PCL) reconstruction surgery due to trauma seven years prior and TKA due to rheumatoid arthritis four years previously. Although the patient suffered no difficulties with walking and performing daily living activities, he was admitted for three months in the recent year due to the worsening of the arthritis. He had been on methotrexate, one of the disease modifying antirheumatic drugs (DMARD) for one year. Pain, swelling, burning sensation, and limited range of motion were observed in the right knee during physical examination. The leukocyte count was 6,770/mm3, erythrocyte sedimentation rate was 46 mm/hr, and C-reactive protein was 44.55 mg/L. Plain radiographs revealed radiolucencies at the bone cement interface (), which led us to perform joint aspiration with a suspicion of deep infection. About 50 mL of purulent exudate was aspirated and SDSD was isolated from the culture, so surgical intervention was conducted. Pyogenic exudate and acute synovitis were noted during surgery. Femoral and tibial components could be removed without difficulty due to severe osteolysis. We did not remove the interference screws inserted in the previous PCL reconstruction because the insertion site in the medial femoral condyle was not affected by the infection. In addition, the screws were so firmly fixated that removal appeared impossible without special techniques and bone loss. Infiltration of more than ten multinuclear leukocytes per high power field was observed in the frozen biopsy samples. Accordingly, we performed a thorough debridement of the synovial sheath and inserted a block type cement spacer impregnated with a combination of three antibiotics (gentamycin 1 g, third generation cephalosporin 4 g, and vancomycin 4 g) to improve infection management (). A splint was applied to the knee in a 30° flexion after surgery. Histology showed acute inflammation () and the synovial fluid culture was positive for SDSD again. The SDSD cultured before and during surgery was resistant to oxacillin in the antibiotic sensitivity test. Accordingly, under the diagnosis of deep infection caused by SDSD resistant to oxacillin, vancomycin (2 g/day) was administered. In addition, third generation cephalosporin (2 g/day, ceftriaxone sodium) was used as a prophylactic agent to prevent other infections in the immunocompromised patient with an opportunistic infection. At seven weeks after surgery, the antibiotic treatment was discontinued because the leukocyte count, erythrocyte sedimentation rate, and C-reactive protein levels were normal and the swelling, burning sensation, and flare improved. The infection did not recur in the following two weeks, so revision TKA was performed. Synovial exudate was not noted during surgery and infiltration of one multinuclear leukocyte in the high power field was observed in the frozen biopsy samples (). Postoperatively, the burning sensation and flare continued and the C-reactive protein level increased to 53.09 mg/L. To prevent the recurrence of the SDSD infection and other infections that may be caused by the long-term hospitalization, vancomycin (2 g/day) and third generation cephalosporin (2 g/day, ceftriaxone sodium) were injected at the same time. The C-reactive protein was 23.88 mg/L at two weeks after surgery and 10.12 mg/L at four weeks after surgery and the symptoms improved. Therefore, the antibiotic treatment was replaced with oral administration of third generation cephalosporin (cefcapene pivoxil hydrochloride 300 mg/day) for the following six weeks. Blood tests were performed every seven days, which showed no abnormal findings. Currently, at six months after surgery, no pain, burning sensation, and swelling have been observed. From four months after surgery, heterotopic ossification in the anterior aspect of the distal femur was observed on plain radiographs. However, magnetic resonance imaging has not revealed abnormal findings such as soft tissue inflammation () and the range of motion has improved to 10° in flexion contracture and 75° in further flexion. |
A 21-year-old man with no significant past medical history was seen in his college student health department with complaints of bifrontal headaches and blurred vision in the left eye. He was noted to have 20/50 vision in his left eye. He denied any history of fever, chills or night sweats but did report a weight loss of 6 lbs. over the past month. He was referred to the emergency room (ER), and a magnetic resonance imaging (MRI) of his brain showed abnormal elevated T2 signal involving the left optic nerve along the leftward aspect of the chiasm in the left prechiasmatic and intracanalicular portions of the left optic nerve. He was treated with IV methylprednisolone for three days, followed by oral prednisone taper. Whereas his headache responded to the steroid treatment, his vision remained the same. An MRI of the cervical and thoracic spine was obtained to see the lesions of multiple sclerosis. This MRI showed a normal spinal cord with no findings consistent with multiple sclerosis. However, incidentally, we noted bilateral level five cervical adenopathy measuring approximately 3 cm and mediastinal and right hilar adenopathy also measuring up to 3 cm. A lumbar puncture was also done that showed glucose of 79 mg/dl, protein of 29 mg/dl, and 2 UL white blood cell (WBC). The results of his cytology examination were negative. A computed tomography (CT) of his neck and chest revealed extensive bilateral adenopathy in the lower neck, supraclavicular areas, right paratracheal region, right hilum, and an anterior mediastinal mass measuring 3.1 cm x 5.8 cm.\nThe patient was discharged from the hospital with an outpatient follow-up for a hematological evaluation. His positron emission tomography (PET) scan showed increased tracer activity associated with lymph nodes in the neck and chest, which was consistent with a clinical history of lymphoma. However, no tracer activity was identified below the diaphragm. He then underwent right cervical lymph node biopsy that showed large nodules encircled by fibrosis with scattered classic and lacunar Reed-Sternberg cells. The patient continued to have blurred vision that had remained the same in the initial outpatient follow-up. He underwent echocardiogram and pulmonary function testing and was started on ABVD (adriamycin, bleomycin,\nvinblastine, dacarbazine) chemotherapy with a plan for a PET scan after two cycles with a total of four cycles with involved-field radiation therapy (IFRT) depending on the chemotherapy response. Optic neuritis was a paraneoplastic phenomenon. The patient decided to receive chemotherapy at a different hospital and did not follow up with our clinic. Upon calling the patient later, he informed us that he was undergoing chemotherapy, and his symptoms of blurred vision have resolved completely. |
A 23-year-old man called for an ambulance due to a sudden onset of severe dyspnea. He had 1-year history of general malaise without a major change in his weight or other health problems. At age 13, he had been diagnosed with Asperger syndrome for which he received pharmacological treatment and psychiatric supervision. Otherwise, he was a healthy young man.\nAt admission, the patient showed clinical signs of acute severe left-sided congestive heart failure; a chest x-ray showed bilateral pleural effusions and a butterfly shadow. A 2-dimensional echocardiogram revealed that an intra-atrial tumor occupied the entire left atrial (LA) cavity and that it had prolapsed across the mitral valve without incarceration. The tumor had both solid and cystic components, and its stalk seemed to be extensively attached to the posterior LA wall. An enhanced computed tomography (CT) scan of the thorax also revealed a similar picture (Fig. ).\nThe patient underwent an emergent open-heart surgery under cardiopulmonary bypass. The LA was approached through a right-sided left atriotomy. The tumor occupied the entire LA cavity and extended to the entry of the right upper pulmonary vein (PV). Macroscopically, the tumor had three differentiated bodies with a shared stalk. One was a thin and fragile tumor widely attached to the posterior LA wall and extended from the entry of the right upper PV to the edge of the mitral annulus. The others’ stalk originated from this tumor. The second tumor was cyst-like with a smooth surface and it contained a bloody liquid with no thrombi. Its wall was very fragile and it was extracted as small pieces from the LA. We were unable to measure its length and width in vivo, but the pre and intra- operative echocardiography revealed that this balloon-like tumor contracted inside the LA and expanded into the left ventricular lumen through the mitral valve with each heartbeat (Fig. ). The third one was a solid, club-like tumor. Its stalk was about 1 cm, the head was about 3 cm in diameter, and its length was about 7 cm. This tumor herniated across the mitral valve during the entire cardiac cycle.\nThese tumors were grossly resected completely from the LA wall (Fig. ). The thin tumor was easily peeled off from the intima of the LA wall without apparent adhesion between the tumor and the outer layer of the LA wall except the stalk mentioned above. The stalk, which was about 2 cm in diameter and attached to the outer layer of the LA wall, was peeled off leaving the outer layer. Here, we intentionally avoided resecting all the layers of the LA wall because the thin tumor seemed to extend over most of the surface of the LA wall. We felt we would be unable to reconstruct the LA if we attempted to completely resect the tumor and the wall. The rest of the tumors were easily resected with the common stalk. In order to prevent a recurrence of the tumor, we applied cryoablation to the attached site.\nAfter the tumor resection, we identified a mitral valve annular enlargement with severe regurgitation, which was probably caused by the prolapse of the large tumor across the valve. We performed a mitral valve annuloplasty with a Physio II ring (Edwards Lifesciences Corp. Irvine, CA, USA) with satisfactory results. The surgery ended safely without major complications or need for transfusions.\nThe traditional histological examination with hematoxylin and eosin technique revealed a tumor composed of spindle-shaped cells with a predominantly myxoid background (Fig. ). Some tumor cells had round-shapes. Most of the tumor showed hypo to intermediate cellularity but focally the tumor contained a hypercellular area. Mitotic figures were found (4 or less mitoses per 50 high-power fields) and there was no necrosis.\nImmunohistochemical tests showed a weak immunoreactivity for calponin, and patchy positivity for CD34. It was negative for CD31, Factor VIII, desmin, myogenin, MyoD1, h-caldesmon, S-100 protein, CAM5.2 and AE1/AE3. The tumor cells showed focal myofibroblastic differentiation. Therefore, the final diagnosis was myxofibrosarcoma. The possibilites of angiosarcoma or leiomyosarcoma were ruled-out by the histological and immunohistochemical features.\nThe patient was discharged on post-operative day 10 without complications. A magnetic resonance imaging (MRI) of the brain and an enhanced whole body CT were negative for metastatic disease.\nUpon discharge, the patient was referred to a local cancer center where he later received radiotherapy for 2 weeks, as adjuvant therapy. His postoperative treatment was completed successfully, and 1-year later he remains asymptomatic without apparent recurrence. A transthoracic echocardiogram 14 months after the surgery was normal. |
The patient, a 30-year-old Tibetan man, was treated in the Lhasa Hospital for right tibiofibular fracture December 2012 (Fig. ). After the swelling subsided, he underwent right tibia fracture intramedullary internal fixation. Two weeks after the operation, the patient was able to walk with double crutches without load, and was able to walk without crutches and with some load 3 months after the operation. However, the patient complained of pain in his right lower extremity, especially when moving downhill or down stairs, and experienced a limping gait and obvious tenderness at the fracture end. Three years after originally presenting at Lhasa Hospital, the patient was experiencing persistent pain in the right lower extremity, and his daily activities were severely affected. Thus, he was re-diagnosed with nonunion of the right tibial fracture after internal fixation (Fig. ). The main causes of the nonunion of the fracture were considered to be the excessively thin intramedullary nails and unstable fixation of the fracture. The operation was repeated with replacement of the crude intramedullary nails and grafting of iliac bone (Fig. ). Three months after the operation, the tibial fracture had failed to heal, and further surgical treatment was performed. The proximal locking screw of the intramedullary nail was removed with an expectation to eliminate the stress shielding effect and to promote fracture healing by increasing the microdynamic force while retaining the static interlocking nail. Five months after the operation, the patient abandoned the crutches and was able to walk with a heavy load. Nevertheless, 2 years after the operation, active pain of the right leg persisted with some tenderness around the fracture, especially when moving downhill or down stairs. August 2018 the patient was admitted to our hospital. Physical examination revealed that two old incision healing scars, approximately 2 cm in length, were present at the proximal and distal ends of the right tibia. The middle section showed a scar from an approximately 10-cm incision; pigmentation was found around the incision and tenderness was experienced around the fracture. Imaging results revealed a nonunion of the right tibial fracture, and the admission diagnosis was nonunion of the right tibial fracture after surgery (Fig. ).\nTwo days after admission, “right tibial locking intramedullary nail removal, open reduction and porous tantalum metal plate fixation” was performed. After removal of intramedullary nails during the operation, nonunion of the tibial fracture was corrected and osteoporosis of the fracture end. Osteosclerosis of the fracture was observed, and the sclerotic bones as well as part of the hyperplastic epiphysis were removed; the marrow was reamed at the fracture to keep the medullary cavity open. The resected osteophytes were implanted into the fracture, followed by fixation with a porous tantalum plate.\nAfter the operation, the affected limbs were fixed with plaster. Ankle joint activity training was initiated on the first day after the operation, and knee joint activity training began 2 weeks after the operation. The patient was able to walk with double crutches without load 4 weeks after the operation and could perform normal activities 12 weeks after the operation without pain in the right limb. However, slight tenderness was still experienced around the fracture. Five months after the surgery, the right tibial fracture had healed, based on imaging examination, and the tenderness around the fracture had disappeared; the patient was able to work normally (Fig. ). |
We present the case of a 35-year-old woman who was brought to the emergency department by her spouse because she developed convulsion for the first time in her life. He reported that while the patient was having her breakfast, she experienced unusual visual and sensory sensations with tinnitus. She was not complaining of any pain or feeling abnormal heartbeats. Suddenly, the patient lost consciousness and fell to the ground. She was not aware of the surrounding. The patient had frothy secretions from the mouth, and her color turned blue. She had repetitive jerky movements of her upper and lower limbs. This episode lasted for two minutes and was terminated spontaneously. After that, the patient regained consciousness, but she was very tired and fell asleep. There was no history of difficulty in speech or weakness following the episode. She did not remember the event. There was no history of early morning headache, nausea, or vomiting.\nThe past medical history of the patient was remarkable for primary infertility due to polycystic ovarian syndrome for which the patient is on metformin 500 mg daily. She had a long-standing history of depression and anxiety for which she underwent frequent cognitive behavioral therapy sessions. She underwent elective laparoscopic cholecystectomy for recurrent episodes of biliary colic. The patient was a non-smoker and never consumed alcohol. She worked as a lecturer and research assistant. Her family history was significant for breast cancer and Parkinson's disease.\nUpon examination, the patient appeared tired. Her vital signs were within the normal limits. She had a heart rate of 90 bpm, respiratory rate of 14 bpm, temperature of 36.8℃, and maintained blood pressure. She was oriented to place, time, and person. There were no speech abnormalities. Neurological examination of the upper and lower limbs showed normal muscle tone and power. The reflexes were normal. She had a normal gait and coordination. Examination of other systems was unremarkable. Further, the initial laboratory investigations revealed no abnormalities (Table ).\nSince the patient had no previous history of epilepsy, a computed tomography scan of the head was performed to rule out any space-occupying lesion. The unenhanced scan demonstrated bilateral symmetrical calcification of the basal ganglia. No calcification was noted in the cerebellum or the cerebral cortex. Otherwise, no intracranial pathology was seen. Such findings conferred the diagnosis of Fahr disease (Figure ). The patient was discharged on carbamazepine to prevent further seizure episodes. After six months of follow-up, the patient did not experience further convulsion episodes. |
In 1960, a 12-year-old male was hospitalized in Shimane because a chest X-ray revealed multiple nodules in both lungs. Although he had no apparent symptoms, such as cough, high fever, or lethargy, he was assumed to have pulmonary miliary tuberculosis and received anti-tuberculosis therapy. Despite 2 years of therapy, the lung nodules continued growing, but he was doing well in general and developed no symptoms.\nHis family doctor doubted the clinical diagnosis of tuberculosis and consulted a specialist. The specialist recommended performing a 131I scintiscan to rule out the presence of metastatic lung tumors from thyroid cancer. In the 1960s, 131I scintiscans were not common and were only performed at one institution in Japan. The patient traveled to Tokyo to undergo the examination in 1962. After the examination, he returned to Shimane, and his medical treatment for tuberculosis was stopped because the 131I scintiscan had revealed that his pulmonary nodules were associated with thyroid cancer. Though his family doctor prescribed some medication, he quit taking the medicine, which caused him palpitation. In Japan patients were seldom informed that they had malignant tumors at that point of time; therefore, his father kept his son from being informed about his own disease. The patient grew up several decades without receiving any medication and led a typical life before his thyroid nodules were noted at a medical checkup. His thyroid carcinoma was resected at age 59 in 2006. Before that, multiple pulmonary nodules showed up many times in medical checkups including chest X-rays. However, further investigations were not performed because he knew that he had a history of multiple asymptomatic pulmonary nodules over a long time. Only one bronchoscopic examination that he underwent when he was 35 years old failed to confirm the diagnosis.\nSince the first diagnosis had been made in the early 1960s, the medical records were no longer available. However, old letters from his doctor passed to the patient's father and an old chest X-ray retained by his father that were kept in his old house were found incidentally in his father's room in 2008 and revealed the details about the patient's past history.\nAt age 58, in 2006, the patient's thyroid tumor and cervical lymphadenopathy were detected using ultrasonography at a health check screening. Total thyroidectomy and radical left neck dissection were performed, and the cancer was diagnosed as papillary carcinoma. X-ray and computed tomography (CT) of the chest revealed multiple fine nodules in both lungs (Fig. ). His serum thyroglobulin (Tg) level was 586 ng/mL even after the thyroidectomy; therefore, multiple pulmonary metastases from thyroid cancer were strongly suspected. However, it was necessary to clarify whether the miliary lung nodules were the metastatic disease from thyroid cancer or postinflammatory scars, because at that time he believed that he had a past history of miliary tuberculosis. A diagnostic 131I whole body scan (131I-WBS) was performed at Kyoto Prefectural University Hospital.\nAt the 131I-WBS, he was asymptomatic, and no pulmonary rale was audible on careful examination. After 2 weeks of L-thyroxine withdrawal and an iodine-free diet, 131I scintigraphy was performed 2 days following the administration of capsules totaling 111 MBq of 131I-NaI. Very intense accumulation was seen in both lungs, and small hot spots indicating metastatic lesions in the left supraclavicular region and in the thyroid bed remnant were found (Fig. ). Subsequently, 3,700 MBq of 131I was administered for treatment.\nAfter the patient had received RIT at age 58, the fine nodular pulmonary lesions showed only slight decrease, and his serum Tg level had dropped to between 150 and 250 ng/mL. Since the Tg level remained still quite high, additional 131I therapy was recommended, which the patient refused because he knew that he had carried the multiple pulmonary nodules for more than 45 years without suffering any symptoms, as he had discovered his father's secret letters that described the illness as having originated in the 1960s. He was convinced that the past detail-uncovered radioiodine administration in Tokyo had provided a dramatic decrease of his pulmonary nodules during several decades and expected a similar efficacy.\nIn 2008, his serum Tg was still high, and diagnostic 131I-WBS and 18F-fluorodeoxyglucose (FDG) positron emission tomography (PET)/CT were performed. 131I-WBS showed that the uptake in both of his lungs had markedly decreased and that the cervical lymph node lesions had disappeared. 18F-FDG accumulation was not seen in the lungs except for small hot spots in the relatively large nodules in his right lung base (Fig. ), which had not increased in size for 2 years. The follow-up did not include any aggressive treatment other than thyroid-stimulating hormone suppression.\nHowever, in 2011, the patient started developing shortness of breath, and X-ray radiography revealed right pleural effusion. CT revealed a pleural mass of about 5 cm in diameter in the right diaphragm. Aspiration examination of the effusion confirmed a diagnosis of anaplastic carcinoma. The right pleura was thickened and the nodules of the right lung base had increased, while the other fine nodules were unchanged compared with their sizes from his 2006 records. Systemic chemotherapy and drainage of the effusion with adhesion therapy were started. However, the pleural mass continued growing, and moreover bladder, multiple bone, and brain metastases developed. He eventually died 5 months after the diagnosis of anaplastic carcinoma. The time course of his disease is summarized in Table . |
A 73-year-old man visited an outside hospital with right chest pain lasting 2 weeks. A right pleural effusion was identified by chest plain film. Pleural fluid cytology was performed and the cytopathologic diagnosis was metastatic adenocarcinoma. The positron emission tomography-computed tomography revealed multifocal uptakes in soft tissue of the right thigh, multiple bones and lymph nodes at mediastinal, right inguinal and left supraclavicular area. He was provisionally diagnosed as having pulmonary adenocarcinoma with multiple metastases and was referred to our hospital.\nAn excisional biopsy of the right inguinal node and needle biopsy and cytology of the mediastinal lymph nodes guided by endobronchial ultrasound (EBUS) were performed. The histopathologic and cytopathologic diagnosis was metastatic angiosarcoma of the epithelioid type. The patient was started on adjuvant chemotherapy with taxol-cisplatin. During chemotherapy, the patient complained of back pain, and bone metastases at the right scapula and at the lumbar vertebra were diagnosed by bone scan and magnetic resonance imaging (MRI). The patient received 10 cycles of adjuvant radiotherapy for the spine metastasis. MRI scan of the thigh showed a persistent subcutaneous mass with ill-defined circumscription and heterogeneous contrast enhancement that was considered to be a soft tissue angiosarcoma without additional pathologic assessment. Chest computed tomography (CT) revealed a small spiculate nodule in the right apical lung, reduced right pleural effusion, and some enlarged lymph nodes in the right supraclavicular, upper paratracheal and hilar areas. Abdominal CT revealed multiple lesions, including masses in the spleen and both hepatic lobes, multiple enlarged lymph nodes at the inguinal, aortocaval and para-aortic areas, and ascites considered to be cancer peritonei. Neither thickening nor mass lesions in the gastrointestinal tract were identified, and no endoscopic evaluation was performed owing to the patient's refusal of the procedure. Several radiologic findings were suspicious for disease progression. The patient was alive at the time of writing, five months after pathologic and cytopathologic diagnosis, but he stopped adjuvant chemotherapy due to poor overall health.\nAn inguinal lymph node (4.5×4.5×3.3 cm) was excised and its cut surface revealed a solid mass, nearly replacing the entire lymph node. The lesion was grayish to whitish tan with hemorrhage. Microscopically, it was a high-grade malignant tumor with rudimentary vascular channel formation (). The major tumor cells were large and epithelioid, and arranged in a solid sheet and nest (). The nuclei were round, oval, indented or multilobulated with clumped chromatin and one to three prominent nucleoli. The features of the cytoplasm also varied from little to plump in amount, and from amphophilic to eosinophilic in color. The other minor tumor cell components were spindles with small and hyperchromatic nuclei and a small amount of eosinophilic cytoplasm. Vascular channel formation was identified in both components, although it was more predominant in the latter component, and focal in the former component. Mitotic figures were numerous throughout the tumor. Immunohistochemically, tumor cells were positive for vimentin and CD31 (), albeit with weak expression, and negative for other immunostaining, including CD34, factor VIII-related antigen, epithelial membrane antigen (EMA), cytokeratin AE1/AE3, cytokeratin 7, HBME1, and calretinin. The final histopathologic diagnosis of the lymph node was metastatic epithelioid angiosarcoma.\nBy EBUS-guided needle biopsy, the mediastinal lymph nodes were diagnosed as malignant, compatible with metastatic angiosarcoma. Tumor cells were arranged in individual cells, cords, loose clusters, or microacini with central lumens in a blood-rich background, which has been frequently found in other mediastinal lymph node biopsies guided by EBUS (). Tumor cells were epithelioid with round, irregularly indented or lobulated nuclei with prominent nucleoli and a scant amount of cytoplasm. Some tumor cells were plasmacytoid with intracytoplasmic amphophilic vacuoles (), perinuclear clearing (), or eosinophilic perinuclear condensation (). However, evident intracytoplasmic hemosiderin pigment or erythrophagocytosis was not identified. Immunohistochemically, tumor cells were positive for CD31 and vimentin ().\nCytologic examination of the mediastinal lymph nodes was also performed by EBUS-guided aspiration and it was prepared using liquid-based cytology (Thin Prep, Cytyc Corporation, Boxborough, MA, USA) by Papanicolaou stain. The cellularity was relatively low and tumor cells were individually scattered with a few small loosely cohesive clusters on a clean background. A few tumor cells were arranged in vague microacini, linear cords or tight small clusters (). The nuclear features ranged from a bland nucleus with round borders and fine chromatin to a pleomorphic nucleus with irregular borders and clumped chromatin. The nucleoli were often seen in multiples and appeared small, but distinct. Tumor cells occasionally exhibited binucleation or multinucleation () and few Reed-Sternberg cell-like cells with markedly irregular nuclei and prominent nucleoli were identified (). None of the tumor cells, even pleomorphic cells, exhibited nuclear hyperchromasia. There were no mitotic figures. The cytoplasm was scant to moderate in amount, and finely granular to densely glassy in texture. Many tumor cells had fine to prominent vacuoles, which were located inside the cytoplasm or protruded beyond the cellular outline (). Several intracytoplasmic vacuoles along the nuclei seemed to be fused, resulting in a band-like appearance (). Some tumor cells exhibited perinuclear clearing circumferentially or in section (). In the cytoplasm of a few tumor cells, there were condensed small blobs () or yellowish green, round bodies, which were considered degenerative RBCs (), but no definite intracytoplasmic hemosiderin deposits were identified. Some tumor cells had elongated cytoplasm and eccentrically located nuclei, creating a plasmacytoid appearance. A few tumor cells had a relatively long, elongated cytoplasmic projection, like a tail. In particular, the intracytoplasmic vacuoles and juxtanuclear condensations were frequent in plasmacytoid cells, where an occasional rhabdoid appearance was made by juxtanuclear condensation and eccentric nuclei. Immunocytochemically, tumor cells were positive for vimentin and negative for cytokeratin 7, pan-cytokeratin, EMA, and leukocyte common antigen. The cytopathologic diagnosis was malignancy compatible with metastatic angiosarcoma. |
A 58-year-old male patient with hyperlipidemia, and maintained on dual antiplatelet therapy (DAPT) due to coronary artery disease status and implantation of two stents, presented with a 4-h history of bilateral lower limb numbness. He was also a heavy smoker and had a history of stroke. He was initially sent to the local medical department where a computed tomography angiography (CTA) revealed pulmonary embolism with a massive thrombus straddling both the pulmonary arteries and aortoiliac artery occlusion with poor collateral recanalization to the lower limb arteries (Fig. ). He was immediately transferred to our center with symptoms of mild orthopnea and resting pain. In addition, cyanosis of the affected limbs was noted, and muscle strength in both the limbs regressed to grade 1. Since enoxaparin was given before transfer and echocardiography did not reveal right ventricle failure, he underwent an emergency thrombectomy for the aortoiliac artery occlusion. After the first operation to salvage the limbs, he was admitted to the intensive care unit for further treatment. On postoperative day 5, the general condition of both the legs improved, with the muscle strength improving to grade 4. He was then transferred to the general ward, and DAPT and enoxaparin were continued to be administered. Owing to the persistence of orthopnea and complaint of chest tightness with intermittent tachycardia, a CTA was performed again to further evaluate the pulmonary embolism on postoperative day 8 (Fig. ). The CTA results revealed that the thrombus was still lodged in the bilateral main pulmonary arteries. Fearing the possibility of a myocardial infarction, cardiac catheterization was performed, which did not reveal any intrastent restenosis or specific lesions in the coronary arteries. Owing to the failure of medical treatment, a staged pulmonary embolectomy with cardiac arrest using heart–lung-machine support was performed on postoperative day 13. During the surgery, intraoperative transesophageal echocardiography did not reveal a PFO or an existing right-to-left shunt. Postoperatively, he was closely monitored in the intensive care unit for 3 days and then transferred to the general ward for 10 days. A follow-up CTA was performed on postoperative day 18 (Fig. ), which revealed resolution of the thrombus in the pulmonary arteries. He was discharged on postoperative day 30 under DAPT and rivaroxaban prescription, without any in-hospital complications. |
An 18-year-old woman was referred with persistent bilateral erythema, warmth and burning pain over her face and ears. She started the symptoms spontaneously 3 years ago, which attacked several times a month and lasted from 2 to more than 10 hours, even days occasionally. She noticed that the episodes always started on the cheeks and then radiated rapidly over the adjacent areas including ears and forehead without any other associations including scales, sweating and headache. The most painful areas were the cheeks. The symptoms disappeared completely without any other facial lesions between episodes. She was diagnosed as seborrheic dermatitis and was treated with oral antihistamines, topical steroids and tacrolimus, and cool facial masque. The therapeutics seemed to be moderately beneficial, whereas cooling the lesion during the attack usually led to rapid pain relief. During the course, she found that the frequency and severity of the flares progressed gradually. She also noticed that warmth and movement could trigger the attacks or worsen the symptoms while cleaning her teeth, eating, drinking, brushing her hair, touching or rubbing the ears or neck movements had little influence on the attacks; and she preferred to stay in cold environment during the flares since the onset. For the past weeks, she was having constant attacking with nocturnal progress when lying down, which seriously disturbed her quality of life including sleep. The symptoms had poor response to the previously mentioned therapeutics except the cooling face masque. Because of intolerable pain, she had to spray cold water or use cold masque on the lesion all day long. The lesions had never involved the extremities. She had no prior history of migraine, episodic headaches or neck injury. Her family history was also unremarkable.\nOn physical examination, she was healthily appearing. During the flare, both ears including the entire helix and antihelix, the forehead and the face were evidently red and tender associated with warmth (Figure a-c). The most painful areas were the cheeks. The temperature on the cheek, forehead and axilla was 36.8 degrees centigrade, 36.7 degrees centigrade and 36.8 degrees centigrade, respectively during the attack. She had no swollen plaque over the involved areas and had no lesions on other parts including feet, hands, neck. Dermatoscope showed evidently dilated vessels inside the erythema during the episodes, which decreased markedly after application of ice over the erythema for about 5 minutes. The lesional redness and pain could be alleviated markedly and rapidly by topical application of ice, but resumed rapidly after the removal of ice. Her heart rate and blood pressure were normal during the attacking. Laboratory test for complete blood cell count, chemistry profiles, liver function tests, auto-immune antibodies, antistreptolysin O serology, HIV antibody and TPPA were either within normal limits or negative. The patient refused to take biopsy.\nThe lesions improved markedly after having been treated with oral gabapentin and indomethacin, and topical lidocaine compounds and cold masque for 3 days; but recurrence occurred 2 weeks after the quit of the treatment. She was then treated with aspirin 0.1, twice a day; paroxetine 10 mg daily, which resulted in complete disappearance of the symptoms after having been treated for 2 weeks. Without any treatment, the patient had been lesion free for 6 months and was still under follow-up. |
A 43-year-old right-handed African American female patient with past medical history of SCD presented to the ED for evaluation of new onset right-sided numbness and weakness 12 h prior to arrival. The patient reported symptoms which progressively worsened throughout the day and sought medical attention when she developed difficulty with her gait. She denied headache, loss of consciousness, and any other focal neurologic deficit. The patient reported an uneventful history of sickle cell anemia requiring a blood transfusion three years ago, but has otherwise been stable with no acute crisis. She was on no chronic pharmaceutical agents, except that she recently initiated megestrol acetate for appetite stimulation just three days prior to symptom onset. Comprehensive neurologic exam was unremarkable, except she had decreased pinprick and weakness on the right upper and lower extremities with associated pronator drift in the right upper extremity.\nOn admission, a complete blood count with differential revealed microcytic anemia with a hemoglobin level of 7.9, compared to her baseline of 8.0. Her peripheral blood smear demonstrated only 1+ sickle cells supporting that she was in a stable, nonexacerbated state of sickle cell anemia. The CT scan of the head demonstrated a focal area of cortical and white matter hypodensity in the lateral left frontal lobe consistent with acute ischemia. The patient was past the window period for consideration of thrombolytics, so tPA was not administered. Aspirin 325 mg was given for primary prevention in the ED and continued with Aspirin 81 mg for secondary stroke prevention. MRI without contrast illustrated several scattered punctate acute lacunar infarcts involving the bilateral hemispheres, suggestive of embolic infarcts (Figure ).\nThus, a cardiac etiology was evaluated via a trans-esophageal echocardiogram which confirmed a small PFO initially identified on a trans-thoracic echocardiogram with bubble study. The PFO was deemed too small to require surgical intervention. Further work up with magnetic resonance angiogram (MRA) demonstrated moderate to severe narrowing of the left anterior cerebral artery (ACA) at junction of the A2 and A3 segments (Figure ).\nDuring the hospital course, the patient returned to her neurologic baseline and was discharged home with aspirin, atorvastatin, and hydroxyurea therapy. She denied having any focal neurologic symptoms such as weakness or numbness on any of her extremities and was ambulating without difficulty. The patient was strongly advised on cessation of megestrol acetate, which could possibly be the culprit of her stroke as her symptoms started just three days after commencing treatment and she had remained otherwise asymptomatic from her SCD. |
A 65-year-old male presented with a three month history of progressive left hip pain. His past medical history was noteworthy for a non-ischemic cardiomyopathy for which he underwent a heart transplant ten years prior to presentation. He had a history of osteoarthritis for which he had undergone bilateral hip replacements complicated by a left prosthetic hip infection with Cutibacterium acnes. His past history was also positive for rheumatoid arthritis, hypertension, and type 2 diabetes.\nThe first stage of his left hip revision after his first episode of prosthetic joint infection with C. acnes occurred pre-heart transplantation and he was treated with 6 weeks of vancomycin following hardware explantation. His second-stage hip revision occurred over a year later, following his heart transplant. His post-transplant course was otherwise uncomplicated, without any episodes of graft dysfunction, rejection, or infectious complications over the previous nine years. He was maintained on tacrolimus and mycophenolate mofetil since his transplant without recent corticosteroid exposure.\nWith regard to his present symptoms, along with the pain, he noted occasional night sweats. He reported receiving 2 hip injections for the pain (one into his hip joint and another into his trochanteric bursa, both on his left side) by his outpatient orthopedic doctor about 2 weeks prior to presentation that provided temporary relief. Sterile precautions were reportedly observed during in-clinic procedures. Following the injections, the patient noticed a progressive fluctuant area swelling of his left lateral thigh and hip. Aspiration of that area 4 days prior by his outpatient orthopedist yielded purulent-appearing fluid that was Gram stain negative for organisms, with no growth seen either on aerobic or anaerobic cultures. Three days prior to admission, he underwent hip arthrocentesis that showed cloudy fluid with 65,000 white blood cells per microliter and a differential of 95% polymorphonuclear neutrophils. Gram stain was negative, and no crystals were seen. Culture of the synovial fluid was later finalized as no growth. However, prosthetic joint infection was suspected and so the patient was admitted for surgery.\nThe patient was an active dog breeder (Yorkies, Maltese, and Morkies), with over 10 dogs at home at any one time, and had recently participated in the birth of several puppies. There was no history of illicit or injection drug use. He also noted being scratched on his legs by the dogs in the past, but could not recall any recent scratches or bites recently.\nOn admission, he was well appearing and comfortable at rest. All vital signs were normal and he was afebrile. He noted mild pain with passive and active movement of his left hip, however, he was able to ambulate. Oropharyngeal exam was unremarkable with good dentition. His bilateral shins had evidence of previously-healed scratches. On the lateral aspect of his thigh was a tender egg-sized swollen area of fluctuance and erythema, without increased warmth or obvious drainage or sinus tract.\nLaboratory evaluation was notable for a peripheral white blood cell count of 6300 per microliter. Erythrocyte sedimentation rate and C-reactive protein levels were 63 mm/hour and 5.4 mg/dL, respectively. Hip X-ray did not show loosening or disruption of his prosthesis. He did not receive any antibiotics prior to surgery. Two sets of pre-operative blood cultures were drawn and finalized as no growth. Medications at the time of presentation included methotrexate (2.5 mg daily), mycophenolate mofetil (250 mg twice daily), and tacrolimus (1 mg twice daily). His diabetes mellitus was well-controlled with dietary changes and his most recent hemoglobin A1c measurement was 6.2%.\nThe area of the suspected abscess the lateral thigh was incised and drained. The proximal prosthetic body, femoral head, and acetabular liner were all removed, the joint was irrigated and debrided, and hardware was replaced in a one-stage fashion. Vancomycin and tobramycin-containing beads were placed. Intraoperatively, frank purulence within the joint was not observed. Five separate tissue specimens were sent for bacterial, fungal, and mycobacterial culture. Post-operatively, vancomycin and ceftriaxone were begun empirically pending further culture data.\nApproximately 2-3 mm of each tissue submitted was embedded whole in fungal media. The remaining tissue from each site was ground in 1 mL Tryptic Soy Broth. Mycobacterial, fungal, and bacterial growth media were inoculated with ground tissue suspension. Gram, Acid Fast, and Calcofluor White stains were prepared from touch preps of intact tissue and ground tissue suspensions. All stains of tissue were negative for organisms. Bacterial cultures were incubated at 35 °C in a 5–10% CO2 incubator for aerobic cultures and in the BD BBL GasPak anaerobic pouch for anaerobic cultures. After four days of incubation, tissue cultures collected from the left hip (one of 5 total submitted) grew small, translucent, spreading colonies from the anaerobic blood agar incubated under anaerobic conditions. Gram stain of the colonies demonstrated spiral-shaped gram-negative rods (Fig. ). Matrix-assisted laser desorption time of flight (MALDI-TOF) mass spectrometry yielded and identification of Anaerobiospirillum succiniciproducens, which is currently unclaimed in the bioMerieux Vitek-MS database. Therefore, 16 s ribosomal RNA gene sequencing was performed, with the best matches using NCBI Blast Nucleotide Sequence Database to Anaerobiospirillum succiniciproduces and Anaerobiospirillum species with > 99% query coverage and identity. Ultimately, the laboratory report was released as “most closely resembles Anaerobiospirillum species; Identified by DNA sequencing.” 16S sequencing was not performed on any other samples including blood.\nMycophenolate was held temporarily in the setting of infection. Following identification of the organism, vancomycin was discontinued and ceftriaxone (2 g daily) was continued to complete 6 weeks total of antibiotic therapy. He tolerated treatment well to date, without evidence of recurrent infection.\nSaliva and perirectal anaerobic cultures of two of the patient’s dogs (both Maltese) were performed using flocked swabs submitted in anaerobic agar gel transport media, and cultured on standard anaerobic media. No organisms resembling Anaerobiospirillum spp. were isolated.\nFollow up 2 months later was unremarkable for relapse of infection, after completion of intravenous antibiotics,. |
In June 2011, a 56-year-old male was referred to our department by head and neck surgeon in order to improve his upper denture retention and stability. The patient was diagnosed with a squamous cell carcinoma of the maxillary gingiva (T4N0M0) in May 2005 and underwent an extended left maxillectomy, an anterior and middle cranial base resection, a left ophthalmectomy, and a flap reconstruction using the rectus abdominis muscle were performed. On physical examination, a recessed deformation on the left side of his face could be seen because of the left ophthalmectomy. The function of the left levator palpebrae muscle was eliminated to the level of a slight elevation by using the frontal muscle. A metal plate was anchored to the inferior wall of orbit. The left ethmoid bone, inferior nasal turbinate, the maxilla, alisphenoid, medial and lateral pterygoid muscle were already excised during the mesh titanium plate reconstruction of the anterior wall from the maxillary orbital region. Intraorally, the left maxilla had been excised from the midline, with the rectus abdominis muscle flap sutured directly to the soft palate. The peripheral mucous membrane around the left upper lip was already scarred, without the oral vestibule, and the flap margin had moved along with the surrounding soft tissue. The 70 × 50 mm flap was sagging from its weight and was in contact with the mandibular molars, reducing the volume of the oral cavity unless dentures were worn. The maxilla was removed from the midline to the maxillary tuberosity, while the mandible was removed from the anterior border of the ramus to the coronoid process. Dead space was eliminated because the abdominal rectus muscle was placed from the anterior cranial base to the oral cavity during reconstruction (Fig. ). No expiratory leakage or food reflux was observed, and the rhinopharyngeal closure was maintained. Prior to performing surgery, there was no tumor recurrence or metastasis. The patient had a mouth opening of 43 mm, which we judged operable and then conducted the flap reduction and elevation under general anesthesia in Dec 2008. Informed consent was obtained from the patient’s parents prior to study initiation, and all procedures were performed in accordance with the Declaration of Helsinki.\nSurgical reconstruction was performed as follows:An incision was made from the buccal side of the sutured edge (scar) in the abdominal rectus muscle flap (Fig. ). We can conduct a vestibular extension at the same time by incising this position. The adipose tissue was peeled from the buccal side to slightly beyond the skin flap center while maintaining approximately 5 mm thickness. The adipose tissue was reduced using a radio knife (8 g) (Fig. ). When we reduce fat tissue, we must avoid perforating of the skin. The skin was incised directly above the zygomatic bone, with tissue separation (avoiding exposure of the plate) to enable easy visibility of the zygomatic bone. Subsequently, the subcutaneous tissue was peeled from the zygomatic bone to the oral cavity for tunneling. Three mini QUICKANCHOR® (Depuy Mitek Surgical Products, Inc. Raynham, MA, USA) anchors were placed in the zygomatic bone, and anchor sutures were drawn through the subcutaneous tissue to lift the skin flap. A modeling compound was used to shape the margin of the celluloid splint (Fig. ). The advantage of flap suspension using Mitek anchors is the simple operability, less anchor positioning limitation, and easier length adjustment of the thread for suspension, which lead to easier fixation of soft tissue without slackness as well as clinically sufficient strength for fixation of ligament and tissue. On the other hand, less than 4 mm thickness of the cortical bone for suture anchor fixation causes insufficient fixation, therefore, determining placing position on the bone for fixation is necessary. Consequently, due to the versatility, the position that is considered optimal for stronger fixation and more efficient suspension can be selected as the anchor placing position, while the periosteum, corium, and scar tissue that are thought the most suitable for maintaining the strength can be chosen for the suture thread. Regarding the anchor placing position in this case, we determined 3 positions on the zygomatic bone and sutured flap corium taking into consideration a complete maxillectomy had been completed, which resulted in being able to lift the flap outward and upward.\nPostoperatively, the color of the skin flap was normal without congestion or necrosis. The celluloid splint was removed 10 days after the surgery with no infection or necrosis observed in the skin flap. We can find only fat, scar tissue, not carcinoma in the reduced fat tissure. At 3 months postoperatively, epithelialization and scarring were observed on the border of the skin flap and buccal mucosa, with no wound opening. Next, a denture that was stabilized to the right residual teeth with a clasp made. This prosthesis had two double Akers cast clasps unilaterally to retain the prosthesis by the four remaining molars. The major connector used anteria paratal plate. The patient was quite satisfied to be able to masticate, form an alimentary bolus, and swallow without any teeth falling out. No re-sagging of the skin flap or wound infection was observed at 3 years postoperatively. Patient follow-up will be continued at our department (Fig. ). |
A 45 year-old Caucasian woman, with no significant past medical history, developed newonset headaches. She was initially treated conservatively with rest and analgesics over the course of several months. However, her symptoms gradually worsened and began to include nonspecific mental status changes such as forgetfulness, daytime somnolence and postural imbalance. She eventually sought care at a local emergency department where her review of symptoms at the time was negative for any cardiac or respiratory involvement. Physical examination revealed no abnormal findings in the chest on auscultation. During the course of her evaluation a magnetic resonance imaging scan revealed two brain lesions, one in the right frontal lobe measuring 2×1.7 cm with extensive edema and another in the left parietal lobe measuring 0.8×0.6 cm on the axial T1-weighted images (, yellow and red arrow, respectively). She underwent a stereotactic biopsy of the right frontal lobe lesion that revealed a spindle cell sarcoma with highgrade features of necrosis, increased mitoses, and nuclear pleomorphism.\nSubsequent staging computed tomography scans of the chest, abdomen and pelvis were not able to identify a primary tumor site. However, a PET scan revealed a FDG-avid in the left atrium. Further evaluation with a transesophageal echocardiogram showed a 2.5×1.8 cm left atrial mass at the base of the posterior mitral valve leaflet with mobile fronds (, yellow circle). An MRI of the chest further demonstrated that this mass was irregular and extended into the appendage and down the anterolateral mitral annulus without interfering with mitral valve function. Shortly thereafter, she underwent stereotactic radiosurgery for the two cerebral metastases present that was later followed by an excision of the left atrial mass with a pericardial patch reconstruction of the atrium and pulmonary veins. Intraoperatively, the tumor was found to obliterate the left atrial appendage and extended into the mitral valve annulus (, yellow arrow). Unfortunately, the mass proved to be larger than radiographic imaging had predicted making the goal of achieving clear margins unattainable, as a small rim of residual tumor was left behind in the atrioventricular groove. The final pathology confirmed a 5.5×5.1×2.1 cm high grade undifferentiated cardiac sarcoma.\nDuring the subsequent month as she was recovering from her surgery, she began to have severe headaches again and an MRI of the brain revealed several new lesions. Moreover, a repeat CT of the chest also showed interval growth of the mass within the left atrium as compared to post-surgical CT studies. She underwent palliative whole brain radiation therapy but continued to experience headaches, visual disturbances, and numbness in the upper extremities. She eventually transitioned to palliative care in view of the rapid progression of disease in the brain. |
A 79-year-old male was brought to the emergency room (ER) of our hospital after falling and striking the occipital region of his head following intake of alcoholic beverages. Four years prior, he had undergone anterior cervical discectomy and fusion of C5/6 at our hospital for degenerative cervical spine disease. He suffered no postoperative neurologic symptoms and was independent in his activities of daily living [Figure and ]. At arrival at the ER, he was lucid and found to be suffering from an occipital subcutaneous hematoma. He presented motor impairment at level C7 and below of manual muscle testing grade 1 and moderate loss of sensation from the trunk and peripheries of both upper limbs to the peripheries of both lower limbs (Frankel B). Emergency cervical computed tomography (CT) indicated severe anterior dislocation of C6/7, and magnetic resonance imaging (MRI) revealed severe spinal cord edema [Figure -].\nThe patient was brought to the operating room for emergency surgery. Under general anesthesia, the head of the patient was fixed with Mayfield in the supine position. Moreover, under the fluoroscopic guidance, we performed cervical traction slowly and carefully. The manipulative reduction was successfully performed, and the patient was placed in the prone position. We performed laminectomy of C5-T1 and posterior fusion of C6/7 [Figure -]. For the posterior fusion, we inserted a lateral mass screw into the facet joint of C6 and a pedicle screw into the vertebral body of C7, paying attention to the course of the vertebral artery on both sides []. Postoperative CT indicated that cervical alignment had improved, and MRI indicated that the spinal cord edema observed prior to surgery had been mitigated []. There were no problems during the patient's postoperative course, and he actively participated in rehabilitation after being transferred to a rehabilitation hospital. Three months after surgery, motor function and sensory impairment of the lower limbs had improved, and the patient was ambulatory upon discharge from hospital (Frankel D). |
We present the case of a 26-year-old male who is referred to the digestive consultation by two episodes of spontaneous paraesophageal abscess in an interval of 2 years.\nIt is a patient with no pathological history of interest that is presented in the Emergency Service for dysphagia for solids of 3 days of evolution that at the same time was suffering stabbing chest pain and fever of up to 38.8 °C in the last 24 h. In the last year the patient had already been in the Emergency Room (ER) twice for chest pain with non-altered complementary tests. The patient denies having any traumatic history or onset of symptomatology after food impaction. The physical examination shows no abnormality on a hemodynamically stable patient. It is performed a blood test showed a C reactive protein (CRP) 190 mg/L (Normal values 0–5 mg/L), and white blood cells 12,000/μL (Normal values 4000–10,000). For that reason it is decided to perform thoracic-abdominal computed tomography (CT), where a collection of 8 × 4 × 5 cm is displayed in the third inferior–posterior of the esophagus compatible with hematoma vs mediastinal abscess (Fig. ).\nThe surgery service is contacted and it is decided to choose the conservative treatment with broad-spectrum antibiotics and absolute diet. During the admission, a echocardiogram with normal results was performed, an esophagogram that does not present alterations and a gastroscopy, where a linear ulcer of 5 mm in distal third of esophagus with biopsy that shows granulation tissue was found.\nThe patient is discharged 7 days after, with the normalization of his analytical and clinical parameters, and showing a correct oral tolerance for later control in consultations.\nAn outpatient USE is requested 3 weeks later, after being discharged, where no paraesophageal collection is displayed. Gastroscopy was repeated where the esophageal ulcer is not visualized and biopsies are taken from the distal and proximal esophagus. In those biopsies, it is noticed an eosinophilic inflammatory infiltration of 40 eosinophils per field.\nThe patient does not attend any control, so no treatment is started.\nOne year later the patient returns to the emergency department with chest pain and dysphagia with same characteristics, and elevation of CRP and white blood cells. Again, a toraco-abdominal CT is performed, objectivizing mediastinal collection in the same location as 1 year before, with a size of 7 × 4 × 4 cm, compatible with abscess, which is retreated in a conservative manner with broad spectrum antibiotics. After 10 days, a CT control confirms resolution of the collection.\nAmbulatory gastroscopy is performed with biopsy-taking by objectivizing an eosinophilic inflammatory infiltrate compatible with eosinophilic esophagitis.\nThe patient denies dysphagia, chest pain, heartburn or any other clinic between episodes of mediastinal abscess.\nIt starts treatment with proton pump inhibitor in double doses during 8 weeks, persisting the eosinophilic inflammatory infiltrate in the biopsies. It is agreed a diet with the patient where two foods will be removed (milk and wheat), obtaining histological remission, and identifying the milk as the cause of the inflammation.\nAfter 2 years of follow-up, the patient maintains milk and derivatives restriction, and has not shown again any episodes of mediastinal abscess. |
A 65-year-old man referred to the emergency department for evaluation of the lower extremity swelling associated with pain for four days. Physical examination demonstrated an overweight man (BMI 29.6 kg/m2) with extensive pitting edema of the left lower limb from the groin to the knee joint with calf tenderness. Color Doppler ultrasound revealed an extensive DVT involving common iliac, external iliac and common femoral vein as well as superficial femoral down to popliteal vein. He was treated by bed rest, elevation with bandaging of left leg, 6000 IU of low molecular weight heparin subcutaneously twice a day and further evaluation was performed to find the underlying etiology. Investigations including hematological, immunological, biochemical, lipid profile, protein S and protein C were normal. Abdominal and pelvic ultrasound (US) showed incidental finding of severe left hydroureteronephrosis with almost lost of cortical thickness, for that abdominal and pelvic computed tomography (CT) scan revealed marked left-sided hydroureteronephrosis and an impacting stone measuring (18 × 10 × 10 mm) at the level of L5/S1 () with signs of DVT affecting left iliac and femoral vein below the above mentioned region (). Next day percutaneous nephrostomy was performed to decompress the hydronephrotic kidney. He was kept as an inpatient for one week under observation then after discharged home on oral anticoagulation in the form of rivaroxaban 20 mg daily. Six weeks later, color Doppler US showed complete recanalization of the superficial femoral, popliteal as well as the proximal segment of deep veins of the leg but common iliac, external iliac and common femoral veins and proximal superficial femoral vein were still partially thrombosed. Under spinal anesthesia, left ureterorenoscopy showed an impacted stone at the level of iliac vessel pulsation causing edema and external compression of the iliac vessels. Through pneumatic lithotripsy, the stone was fragmented and JJ stent inserted (). Next day the patient discharged home and continued on taking his antithrombotic treatment (rivaroxaban 20 mg). The JJ stent was removed 3 weeks later. Three months after that, Doppler US showed complete recanalization of iliac vessels. |
A 52-year-old Saudi woman presented to our outpatient dermatology clinic complaining of multiple painful bumps on her trunk and upper extremities, which had been present for the last 25 years. Recently, the lesions had become more painful to touch and increased in both size and number. She also had a history of uterine leiomyomatosis for which she underwent hysterectomy 17 years ago. A family history revealed that her mother, 2 sisters and 2 maternal aunts also had a history of multiple uterine leiomyomatosis.\nA physical examination revealed several smooth, nonmobile nodules that were reddish to brown in color, arranged in a cluster on her right arm (fig. ), left forearm and trunk. The nodules were both firm and tender to touch, ranging in diameter from 0.5 to 2 cm. The most prominent and painful nodules were removed and submitted for histological analysis, which showed a relatively well-circumscribed dermal proliferation of intersecting fascicles of spindle cells (fig. ). The cells exhibited cigar-shaped nuclei and growth in between collagen bundles. The findings were consistent with a diagnosis of benign dermal leiomyoma.\nBased on the patient's unique clinical presentation in conjunction with her surgical and family histories, a diagnosis of Reed's syndrome was confirmed. The patient was referred for genetic counseling and for evaluation of renal malignancy. Renal ultrasound and magnetic resonance imaging (MRI) were negative for renal pathology. No symptoms indicative of renal cell cancer were found.\nThe patient returned to the clinic after 5 years, complaining of more painful lesions.\nIn this instance, the patient's dermal lesions on her right arm were treated with liquid nitrogen cryotherapy. The therapy consisted of two 30-second freeze thaw cycles with a 1 mm margin. After 2 weeks of cryotherapy, the patient reported a decrease in the pain and size of the lesions (fig. ). One year after cryotherapy treatment, the patient reported nonrecurrence of the lesions, with absence of pain at the site of the lesions. |
The patient is a 70-year-old male who was found in a chair at home by emergency medical services after a call from a friend who did not have his usual contact with the patient for several days. When paramedics arrived, it appeared he had been in that position for some time, as he had been incontinent of both bowel and bladder. He was found to be non-verbal, responsive only to painful stimuli, and was able to move all extremities, giving him a Glasgow Coma Scale (GCS) of eight. Medical history obtained from the friend who called emergency services was largely incomplete but was positive for diabetes mellitus. The patient had no reported history of stroke. Upon arrival to the emergency department, the patient began to seize with right gaze deviation. Lorazepam was administered to control the seizure and an emergent intubation was performed to protect his airway. The patient was afebrile, and vital signs were within normal limits throughout the course of presentation and treatment. On physical exam, there was a mild left facial droop with 3 mm pupils that were equal and reactive to light. The rest of his exam was non-focal. Laboratory results demonstrated a leukocytosis of 20,720 per cubic millimeter of blood and mild electrolyte disturbances.\nA computed tomography (CT) scan performed in the emergency department showed a crescent-shaped hemorrhage with midline shift consistent with a subdural hemorrhage (Figure ). The CT was read by the radiologist as, “Subacute left subdural hematoma, 1.4 cm in width with midline shift from left to right of 0.7 cm. Left frontal encephalomalacia versus involving Q skin nick event.” The most likely differential diagnosis now included subdural hematoma vs. empyema. It was felt that the symptomatology did not overlap completely with subdural hemorrhage so magnetic resonance imaging (MRI) was performed. It showed a complex left-sided acute subdural fluid collection of moderate size with mass effect upon the left cerebral hemisphere and a 6.3 mm midline shift to the right (Figure ). It also showed a heterogeneous area at the frontal tip of the left frontal lobe that was interpreted as a frontoparietal abscess. The MRI also revealed enhancement of the frontal sinuses consistent with frontal sinusitis. History taken from family members days later was positive for mild, self-treated, sinusitis-like symptoms immediately prior to his presentation.\nAn emergent frontoparietal craniotomy was performed, and a loculated abscess was found in the anterior tip of the left frontal lobe. It was evacuated and the space was irrigated with bacitracin. Prior to irrigation, samples were taken from the subdural space and the abscess.\nThe patient did well postoperatively and by postoperative day five he was extubated and verbal, but with some anomic aphasia. Hemiparesis was present immediately postoperative but improved rapidly along with his facial droop. Cultures from the abscess grew Streptococcus anginosus. He did have some seizure activity, myokymia of the face, and left upper extremity tremors on postoperative day one, which were controlled with levetiracetam 1000 mg, twice per day. The patient continued to improve neurologically and was well enough to be discharged on postoperative day eight to an inpatient rehab facility for rehabilitation and continued intravenous antibiotics. |
A 49-year-old Caucasian female, with a long history of heavy cigarette smoking and a previous diagnosis of supraventricular tachycardia, for which cardiac ablation was performed 2 years ago, was admitted to the hospital for an elective sacral neuromodulator lead change to treat urinary incontinence. In the operating room, the patient received monitored anesthesia care (MAC) in the prone position. During the procedure she required increasing sedation (IV propofol infusion) and after thirty minutes the patient showed progressive hypercapnia from the nasal cannula sample line, while receiving supplemental oxygen (4 L/min). An oral airway was placed, but end-tidal CO2 ranged from 65 to 75 mmHg and did not improve. It was then decided to place a laryngeal mask and to supply positive pressure ventilation; however no improvement of the hypercapnia ensued and we could not provide adequate ventilation. The patient was then emergently intubated to secure an airway and provide adequate ventilation. This required interruption of surgery, turning the patient supine, ventilating oxygen by mask and a succinylcholine bolus. During the intubation process, marked edema above the vocal cords was noticed by an experienced anesthesiologist, allowing only for a smaller sized endotracheal tube to be inserted (6 mm with difficulty, but not a 7 mm tube). The patient was given dexamethasone 8 mg and diphenhydramine 25 mg for the airway edema. Following intubation and after securing the tube in place, we started positive pressure ventilation and returned the patient to the prone position, and the end tidal CO2 returned within normal range. However, within a few minutes, the patient developed bibasilar rales, with abundant frothy, blood – tinged secretions appearing in the tube. We maintained the patient prone while the surgical procedure was concluded. The edema fluid was adequately suctioned and abated after 10 minutes of positive pressure ventilation. Postoperatively the patient remained intubated and sedated on a propofol infusion. While in recovery, an arterial blood gas obtained on Synchronized Intermittent Mechanical Ventilation (SIMV) and FiO2 of 30%, showed a PO2 of 92 mmHg, PCO2 of 46 mmHg, pH of 7.34 and HCO3 of 25 mEq/L with a SaO2 of 97%. Chest X-ray performed after surgery indicated clear lung fields with normal size heart and no evidence of pulmonary edema. The patient’s condition steadily improved and she was subsequently extubated two hours later but continued to be closely monitored overnight and was discharged the following day without requiring an ICU stay. |
A 64-year-old male patient presented to his primary care physician complaining of a dry cough and recurrent bouts of bronchitis as well as frequent orthostatic dizziness. At the age of 20 years, he had undergone repair of CO-A through a left thoracotomy using an interposition Dacron graft between the left SCA and the mid thoracic aorta. His chest radiograph raised suspicion of AN of the thoracic aorta. Computed tomographic angiogram then confirmed the presence of two large anastomotic PANs at both ends of the graft (\n) as well as an occluded right SCA, stenosis of the left vertebral artery, and bovine origin of the carotid arteries (\n).\nIn view of this complex anatomy, the decision was made to use a hybrid approach to address these findings. In a first stage, a right carotid artery to right SCA bypass was done using a short segment of Goretex graft (\n). Next, using single lung ventilation, a right posterolateral thoracotomy was performed and the chest entered through the fifth intercostal space. With the diaphragm retracted inferiorly, the pericardium, posterior to the phrenic nerve, was gently lifted with a long clamp, thus allowing exposure of the distal most portion of the thoracic aorta, medial to the inferior vena cava (IVC). After heparinization, the aorta was partially clamped and a 22 mm Hemashield graft anastomosed in an end-to-side fashion to an appropriate aortotomy using a continuous suture of 5–0 Prolene (\n). The graft was then brought posteriorly to the IVC and anteriorly to the right hilum, then anastomosed in an end-to-side similar fashion to the partially occluded ascending aorta through a vertical pericardial opening (\n,\n).\nThe immediate postoperative course was uneventful, and 5 days later, the patient was brought to the hybrid operating room for completion angiogram. This confirmed patency of the extra-anatomic graft as well as the presence of a 4 cm proximal left SCA anastomotic PAN with close extension to the left vertebral artery origin, and a 4.5 cm distal anastomotic PAN. EVS of the proximal PAN was done using a 16 × 80 mm covered Medtronic stent deployed across the origin of the left vertebral artery (\n).The distal PAN was excluded using a 26 × 100 mm covered Medtronic stent extending from the level of the coarctation to the insertion of the extra-anatomic bypass graft (\n). Completion angiogram confirmed patency of the latter and occlusion of the left SCA to thoracic aorta graft as well as of both anastomotic PANs (\n). The postoperative course was uneventful and the patient remains asymptomatic 2 years later and will be followed up on a yearly basis. |
A 58-year-old man with progressive difficulties in swallowing and articulation was referred to our department for radiotherapy of locally advanced squamous cell carcinoma of the base of tongue. The patient was a heavy smoker and had a history of drug abuse.\nOn physical examination, the patient had a lesion fixed to the right part of the base of tongue and ipsilateral palpable posterior cervical lymph nodes. The CT scan revealed a 59 × 43 × 56 mm lesion of the right tongue base, invading the uvula and extending to the ipsilateral site of the epiglottis (red-circled area in ) as well as a lymph node block of 35 mm diameter with central necrosis of the ipsilateral level IIA-IIB (red arrow in ). Moreover, enlarged nodes of the ipsilateral level VA and nodes up to 16 mm of the contralateral levels IIB and upper VA have been found.\nPositron emission tomography (PET) or PET-CT is a very important diagnostic procedure to be performed prior any kind of treatment in head and neck cancer. Indeed, PET-CT scan improves the accuracy of pre-treatment staging in terms of sensitivity and specificity and helps in better delineating the radiotherapy target. However, no PET-CT was performed to our patient due to the lack of PET-CT scanner in our center or nearby. Finally, our patient was human papillomavirus (HPV)-negative although in international literature we found that 40% to 45% of oropharyngeal carcinomas are HPV-positive.\nSince there was no evidence of metastatic disease according to chest computing tomography and liver ultrasound, the patient was staged as T4aN2c and the multidisciplinary meeting decided to offer him a course of concomitant radiochemotherapy. Given the refusal of the patient to attend the standard long course of radiotherapy and his relatively bad compliance due to drug abuse, we finally decided to offer him a course of hypofractionated radiotherapy with weekly cisplatin 40 mg/m2.\nThe patient was referred for dental assessment and placement of a percutaneous endoscopic gastrostomy tube in order to bypass feeding and eating difficulties during the radiotherapy course. Then he underwent a planning CT scan of the head and neck on the basis of which we prepared his treatment plan.\nThe patient was treated with hypofractionated 3D conformal radiotherapy. He received 30 Gy in 10 fractions in the primary tumour, neck and supraclavicular fossa and 15 Gy as a boost in the gross tumour volume, reaching a total dose of 45 Gy, which is biologically equivalent to 54 Gy and clearly lower than the usual total therapeutic dose of 70 Gy given by conventional fractionation of 2 Gy per fraction.\nThe patient was assessed every week for a toxicity report and followed-up regularly both clinically and radiographically after the completion of radiotherapy.\nAt the end of the radiotherapy course there was significant clinical improvement with a substantial decrease in gross tumour volume and also in the level of pain, an increase in tongue mobility and improvement in articulation. Early toxicity was limited to grade II mucositis and skin toxicity no higher than grade II was observed. Overall treatment tolerance was excellent.\nTwelve months after the completion of radiotherapy, the CT scan confirmed a near-total remission of primary lesion and a full regression of metastatic lymphnode, along with clinical improvement of the patient (). On further radiological and clinical follow-up there was no sign of local recurrence.\nEighteen months after the completion of radiotherapy the patient is free of recurrence with no swallowing or articulation difficulties and no signs of late skin toxicity. |
A 59-year-old male with a past medical history of tobacco and marijuana use, chronic back pain not on narcotics, and untreated hypertension and dyslipidemia was transferred from an outside hospital for further workup after presenting there with acute substernal chest pain radiating to his anterior neck. He was found to be hypotensive and bradycardic with a blood pressure of 70s/40s and a heart rate in the 40s. He was given atropine in route and on arrival received a normal saline bolus, morphine, ondansetron, and lorazepam for his symptoms. The electrocardiogram showed sinus bradycardia with no ST-segment elevations. Chest radiography showed a mildly enlarged cardiac silhouette with no acute abnormalities. Labs were significant for a negative troponin and an elevated D dimer to 35 mg/L FEU. He was transferred to our facility because their CT scan was not functioning.\nOn arrival, the patient had a blood pressure of 114/45 with a heart rate of 54 bpm. He continued to have constant chest pain. On physical exam, he had normal heart sounds and equal radial and femoral pulses. His blood pressure could not initially be obtained on the right upper extremity, but after being taken manually was similar to the left. His electrocardiogram showed low voltage sinus bradycardia, and so a bedside transthoracic echo (TTE) was done. This showed a dilated aortic root with an intimal flap suggestive of aortic dissection (Figures , ).\nThe abdominal aorta was then evaluated and also showed a flap extending to the renal arteries (Figures , ).\nNo effusions or right heart strain was noted. The patient was taken emergently to CT scan, and the results were discussed with the radiologist over the phone immediately. This information was then relayed to the cardiothoracic surgeon.\nThe CTA chest and abdomen/pelvis showed a Stanford type A aortic dissection (Figures , ).\nAn intimal flap began at the aortic root and extended throughout the ascending and descending thoracic aorta into the bilateral common iliac arteries (Figure ).\nThe dissection also extended into the right brachiocephalic artery and proximal right common carotid artery. The ascending thoracic aortic aneurysm measured up to 4.8 cm, and the proximal descending thoracic aortic aneurysm measured up to 3.9 cm.\nThe cardiothoracic surgeon immediately reviewed the images and took the patient for an emergent aortic dissection repair with a 32-mm hemashield graft placed and resuspension of the aortic valve. He was discharged 10 days later to a cardiac rehabilitation facility after an unremarkable postoperative course. |
The patient was a 60-year-old man. Abdominal ultrasonography indicated that he had an abdominal mass. He reported having perceived lower abdominal pain 7 months before the initial examination, but it spontaneously remitted. The patient had no fever nor any history of conditions such as hypertension. A blood test revealed that he did not have anemia, thrombocytopenia, or coagulation abnormalities. The patient demonstrated slightly elevated levels of carcinoembryonic antigen (4.7 ng/ml) and soluble interleukin-2 receptor (603 U/ml), tumor markers that are elevated in gastrointestinal cancers and malignant lymphoma. Contrast-enhanced CT showed a 33× 31 mm abdominal aortic aneurysm and a 106 × 81 mm mass peripheral to the abdominal aorta. The mass did not feature a contrast effect, and its margin was enhanced in the venous phase (Fig. ). Sigmoid wall thickening and inferior vena cava thrombosis were also observed (Fig. ). We suspected the patient had a neoplastic disease, such as a malignant lymphoma or sigmoid colon cancer. Detailed examinations were consequently performed, and direct oral anticoagulant therapy was initiated to treat the inferior vena cava thrombosis.\nThe FDG-PET revealed an abnormal accumulation of FDG in the margin of the mass and the sigmoid colon, but not in any other organs. Colonoscopy showed only mild inflammation in the sigmoid colon, and a sigmoid colon biopsy revealed no significant findings. Chronic inflamed fibrous connective tissue was harvested from the periaortic mass using CT-guided biopsy. There were no findings indicative of infection or neoplastic disease, thus precluding a pathological diagnosis.\nAt 1 month after the initial examination, intermittent claudication occurred, prompting the performance of follow-up contrast-enhanced CT scanning. On CT scanning, we found an arterial occlusion from the infrarenal abdominal aorta to the bilateral common iliac arteries, the right external iliac artery, and the right internal iliac artery (Fig. ). These findings indicated the potential progress of the tumor to the aorta. Via a midline incision, biopsies of the mass and the lymph nodes peripheral to the sigmoid colon were obtained. However, these biopsies showed only connective tissue with inflammation and fibrosis and did not yield any significant findings. Since the developments, we elected to resect the mass and perform bypass surgery for definitive diagnosis and treatment after 3 weeks from biopsy.\nSurgery was performed using a transperitoneal approach via a midline incision. Adhesion was observed peripheral to the abdominal aorta, the bilateral iliac arteries, and the retroperitoneal mass. The abdominal aortic lumen was completely occluded by an old thrombus and plaque. Removal of the thrombus revealed a 35-mm intimal defect consistent with the direction of the retroperitoneal mass (Fig. ). Communication was observed between the intimal defect site and the retroperitoneal mass, which was filled with a partially organized old thrombus. During the intraoperative rapid pathological examination, the tissue inside the mass was diagnosed as a fibrin clot. The tumor tissue was not assessed. The area from the infrarenal abdominal aorta to the right common femoral artery and the left common iliac artery was bypassed using a Y-shaped woven Dacron graft. Pathologic examination showed that the mass tissue was a partially organized fibrin clot, thus agreeing with our intraoperative rapid diagnosis. On the basis of the intraoperative and pathologic findings, the patient was diagnosed with CCR-AAA with abdominal aortic occlusion. His intermittent claudication improved, and he was discharged on day 19 after surgery. Six months after the operation, the retroperitoneal mass disappeared on CT. |
We present a case of 40-year-old male patient with a colloid cyst in the third ventricle. The patient was admitted at Clinic of Radiology, with symptoms of headache and episodes of vomit. In clinical examination patient had symptoms of brain tumor. He was treated for depression since 2017. A year ago he underwent a MRI head scan, where dilation of lateral ventricle was seen as sign of early hydrocephalus.\nThe MRI scan demonstrated a cyst in foramen of Monro region with typical signal intensities in the cystic formation that confirmed the diagnosis of colloid cyst (). In MRI images, they present as homogeneously to heterogeneously hypointense or hyperintense masses in T1 and T2 weighted acquisitions. The outstanding characteristic is a sharply outlined round or ovoid mass in the anterior and superior third ventricle, near or at the foramen of Monro region. Hydrocephalus is observed in most patients4. After diagnosis the patients was recommended to consult with a Neurosurgeon for intervention. The patient underwent surgical intervention and the cyst was removed. Similar findings were revealed also on histopathological examination (, Panels a, b, c, d, e and f). After the intervention the patient did post-operative MRI exam control as recommended where it was evidenced successful tumoral extraction while the histopathologic results described findings compatible with colloid cyst Fig2.\nA colloid cyst can be removed with a craniotomy. A craniotomy is a surgery where an incision is made in the scalp, and part of the skull is removed for the duration of the surgery then the skull is put back in place.\nOn the T1-weighted images, the lesion showed a central signal intensity slightly higher than that of the surrounding brain with a mildly hypointense rim (, Panel a). On proton density–weighted images, the colloid cyst was of homogeneous signal intensity, and on T2-weighted images, it showed a central region of hypointensity surrounded by a rim of relative hyperintensity (similar to that of white matter) (, Panels b and c). The T2-weighted appearance was a reversal of that seen on the T1-weighted study. The colloid cyst appeared slightly oval on this projection. |
A previously healthy 43-year-old female presented to the surgical oncologist after evaluation by medical oncology, who felt her open wounds made her a poor candidate for chemotherapy. She presented with a large fungating right breast mass (Figure ). She endorsed a history of a chronic, non-healing right breast wound that began after mastopexy (five years prior) for extreme weight loss (~100 lbs) after consecutive pregnancies. Her contralateral wound healed after reconstruction, but the right breast wound continued to progress despite attempted reconstruction. She noted a small mass at the surgical incision, which grew rapidly in the subsequent months developing into a fungating lesion. Outside hospital biopsy was significant for squamous cell carcinoma (SCC), although the pathologist was unable to differentiate between cutaneous and metaplastic breast SCC. Physical exam confirmed a large fungating lesion of the right breast expanding onto her anterior abdominal wall and axilla with a fixed mass on the chest wall, with multiple palpable axillary nodes on the right. Erythema extended over the entirety of the breast footprint, crossing the midline with satellite lesions on the upper abdominal wall.\nShe underwent excision with planned immediate reconstruction for wound coverage rather than breast reconstruction. Despite radical excision of her breast, pectoralis major, and level 1/2 lymph nodes, the surgeon felt she had disease adherent to the ribs. Given the extent of the unanticipated disease, the decision was made to delay reconstruction until further staging and resultant pathology. While pathology was pending, the patient underwent positron emission tomography (PET) scan for staging on post-operative day 1 notable for increased activity concerning for malignancy in the right internal mammary lymph node chain and left axially lymph node enlargement (Figure ). On repeat visits to the operating room to exchange her wound vacuum, the surgeon noted signs of progressive disease, which was confirmed by intraoperative pathology.\nPathology from the initial excision confirmed the diagnosis of squamous cell carcinoma, with a 9.5-cm central mass with extensive involvement of the breast parenchyma and skeletal muscle. Additionally, positive deep surgical margins and four out of 11 positive lymph nodes reaffirmed the decision to delay reconstruction. Lastly, the pathologist echoed a cutaneous origin of SCC disease citing the presence of SCC in situ focally at the periphery of the ulcer, the lack of admixed conventional invasion and in situ breast carcinoma, and the clinical history of a long-standing ulcer. An attempt at chest wall resection was aborted due to extensive disease on exploration. Critical to note is that surgical manipulation of the tumor bed seemed to trigger a massive proliferation with local extension occurring rapidly. On the two following operating room trips, numerous additional nodules beyond the original margins sprouted in all directions. Therefore, radical excision with intent for curative resection was not possible.\nTo prevent further delay of non-surgically amenable disease, the patient was offered split-thickness skin grafting over her open wound to allow for prompt radiation. On HD21, she was taken for skin grafting. After debridement, the final wound bed measured 45 x 40 cm. Elevation of the supraclavicular tissue, overlying latissimus skin, medialization of the contralateral breast, and reverse abdominoplasty reduced the area to 20 x 15 cm, which was skin-grafted over. She was discharged on post operative day 4 (HD25) with home health. Despite systemic and local therapy with cemiplimab, the patient had disease progression (Figure ) and subsequently succumbed to her disease. |
A 59-year-old Caucasian woman with a history of intermittent abdominal pain and significant weight loss for the last two years was admitted to hospital for elective aorto-mesenteric bypass surgery. Over the course of the two years she had presented to multiple doctors who had performed various procedures including gastroscopy and cholecystectomy without finding and eliminating the course for the patient's symptoms. A CT scan was performed which showed focal narrowing of the coeliac trunk. The patient was then referred to our hospital for a mesenteric artery angiogram. This demonstrated a high grade stenosis of the proximal coeliac artery, complete occlusion of the superior mesenteric artery and hypertrophy of the inferior mesenteric artery with evidence of a wondering artery of Drummond supplying the branches of the superior mesenteric artery and thus confirmed the suspected diagnosis of occlusive mesenteric vascular disease.\nThe patient described the abdominal pain as intermittent, mostly appearing 20 to 30 minutes after eating big meals. As a consequence she had begun to associate food with pain and had developed sitophobia (fear of food) resulting in significant weight loss. Her medical history was also significant for smoking and hypercholesterolaemia.\nOn physical examination in pre-admission clinic the patient appeared cachectic. Her blood pressure was 110/80 on the right side and 105/75 on the left, pulse was 80 beats/min and regular, respiratory rate was 15 breaths/min and oral temperature was 36.7°C. Her examination also revealed normal jugular venous pressure and normal breath sounds over both lung bases. Heart sounds were distant, and peripheral pulses were normal. The remainder of the examination findings were unremarkable.\nThe ECG performed showed normal sinus rhythm without any ST-segment changes. Blood results including liver function tests and coagulation studies were normal. The chest radiograph obtained showed no abnormalities.\nAfter routine preparations for surgery the patient went to theatre and mesenteric revascularization was performed with an antegrade prosthetic graft bypass. The proximal trunk of the small-calibre bifurcated prosthetic graft was anastomosed to the supracoeliac aorta, and the distal limbs were sewn to the coeliac artery and superior mesenteric artery, just beyond the stenotic segments. Intraoperative duplex ultrasound examination confirmed the technical adequacy of the revascularization. Postoperatively the patient was transfered to the intensive care unit. However, the next day blood tests showed a steep rise in her liver enzymes (ALT 1792 U/L, AST 2731 U/L) suggestive of hepatic ischemia and duplex ultrasonography confirmed the suspected diagnosis of graft occlusion. The patient was taken back to theatre for emergency thrombendarterectomy. Both limbs of the graft were found to be occluded and the coeliac limb was shortened. Overnight the arterial blood lactate level began to increase so that a laparotomy was performed revealing a dusky small bowel segment which was resected. The patient was commenced on transparenteral nutrition and remained stable until 18 days post initial surgery when signs of sepsis and peritonitis became evident . Laparotomy was again performed resulting in splenectomy, resection of the small bowel anastomosis and abdominal wash-out. The recovery period was then prolonged but without further complications and the patient was transferred for rehabilitation. |
In March 2016, a 43-year-old male was referred to an odontology clinic with toothache and fever. His previous medical history was unremarkable. On physical examination, the upper airway was open but the right nasal cavity appeared to be closed by tumoral lesion. Edema of the right palate with the involvement of the hard and soft palates was seen. In the right side of the neck, multiple lymph nodes were touched along the sternocleidomastoid (SCM). Owing to high grade fever, complete laboratory blood tests were requested. In complete blood cell count (CBC), increase of white blood cells 11,500 per microliter (3.5–10) was detected. It appeared that the patient had a source of infection in the mouth. Despite receiving high doses of antibiotics, no signs of healing were observed. Also, it seemed that the main lesion was beyond simple infection. Ultrasound investigation of the right lateral neck chain revealed two heteroechoic lymph nodes with irregular lobulated borders containing internal necrosis, with one of them measuring 21 × 14 mm (with conglomerate feature composed of edema associated with matting), and the other one measuring 12 × 24 mm (with the suspicious feature of malignancy) and containing necrosis. Another lymph node was situated anteriorly to the carotid artery with a reactive feature measuring 9 × 3 mm in diameter. Left neck ultrasound revealed multiple lymph nodes with some of them having a reactive feature and one having a suspicious feature for malignancy in the left supraclavicular area. Tri-dimensional head and neck computed tomography (CT) scan revealed increasing thickness of soft tissue of the right maxillary sinus area with massive destruction of the right maxillary sinus wall (Figures , , and ). The patient underwent biopsy via endoscopic procedure of right maxillary sinus mass in an outpatient maxillofacial surgery clinic. The first pathology report was basaloid squamous cell carcinoma, without any immunohistochemical study. For confirmation of diagnosis and to decide treatment protocol, the sample was sent back for consultation to our center. Histopathological examination of Hematoxylin and Eosin (H&E) stained glass slides revealed a tumoral lesion composed of dark hyperchromatic neoplastic cells with round nuclei (basaloid-like cells) that were arranged in solid nests and acinar structures with peripheral palisading pattern in the fibromyxoid stroma without characteristic features of adenoid cystic carcinoma (). In differential diagnosis, solid type of ACC was considered. For definite diagnosis IHC studies were done and neoplastic cells showed positive reaction pattern to the S-100 (diffusely cytoplasmic and nuclear staining) (Figures and ), pan-cytokeratin (AE1/AE3) (cell membrane staining) (), Vimentin, p53, C-kit, alpha smooth muscle actin, and also a ki67 labeling index of 60% (nuclear staining) (Figures and ) (Dako, Denmark). All neoplastic cells showed negative staining pattern for p63 (). The negative reaction of the malignant cells for p63 were not justifiable, because each of the two tumors under discussion (adenoid cystic carcinoma and basaloid squamous cell carcinoma) should have a positive reaction to this marker, and only their pattern of staining was different. Therefore, according to histopathologic findings, this tumor was suggested to be basaloid squamous cell carcinoma. The patient underwent hemimaxillectomy, semi-hard palate resection, cheek bone, right neck, submental and submandibular lymph nodes dissection and resection of the fascia of maxillary sinus, nose, and soft palate, and also palatine tonsillectomy. Due to the excessive extension of tumor cells into the surrounding tissues and the involvement of orbital bone, safe margins could not be made during the surgery (). The histopathological examination of received tissues showed tumoral lesion composed of small, dark, round cells with angulated nuclei, arranged in solid nests, trabeculae, cribriform, and tubular structures within fibromyxoid stroma in the depth of the tumor, with characteristic features of ACC tumors (). Numerous perineural and intraneural invasions with lymph node metastasis were present (). IHC studies revealed compartment staining pattern of neoplastic cells for p63 (Figures , , and ). Based on histopathological and IHC findings, the final diagnosis of tumor was grade 2 ACC. After surgery, adjuvant radiotherapy was considered for the patient. The patient has taken three courses of chemotherapy and thirty courses of radiotherapy. At the end of radiation therapy, he lost his right eye vision.\nFollow-up: seventeen months from initial diagnosis, he was still alive without lung or distant metastasis. |
A 57-year-old male had received left below-knee amputation and autogenous skin graft in another hospital, due to an accident which occurred on March 24 2007, when he slid into a de-icing machine. The patient was transferred to the Department of Physical Medicine and Rehabilitation at our hospital 2 weeks after the accident. The patient was on anti-hypertensive medication, but did not suffer from other underlying diseases, such as diabetes or vascular diseases. Application of the prosthesis was performed after muscle strengthening exercise and knee joint stretching exercise, massage over the distal stump and desensitization training. As the partial thickness wound and pain caused by attaching the prosthesis posed difficulty to rehabilitation training, we tried to adjust the prosthesis many times, but the symptoms were not improved. Stump revision was performed once at the Department of Orthopedic Surgery of our hospital, to manage stump wound which persistently occurred for more than 5 months after the accident. Subsequently, the wound was healed. After the amputation stump matured and scar was formed, the prosthesis of lower extremities was replaced with a new custom prosthesis. However, the stump pain has persisted since then and did not respond to various analgesics, including acetaminophen, non-steroidal anti-inflammatory drugs (NSAIDs), tramadol hydrochloride and oxycodone hydrochloride. When the prosthesis was fitted, persistent and unbearably excruciating pain at the stump site precluded the patient from doing rehabilitation program, including gait training.\nDuring physical examination performed on May 16 2008, mild skin scraping and erythema, but no edema or adhesion were observed at the amputation site, within the prosthetic socket. The range of motion (ROM) during knee flexion and extension above the amputation site was normal. Manual muscle testing found that the muscle strength of flexors and extensors of the left hip and knee was decreased by 20%, compared to those of the right hip and knee. A small, palpable, movable mass was observed in the lateral side of the amputation stump, near the amputation site of fibula. Hyperalgesia and hyperpathia developed in response to light palpation. The level of produced pain evaluated on the visual analog scale (VAS) pain rating was 10.\nNeurological examination revealed no abnormal findings and the patient did not complain of phantom limb pain. The prosthesis was well adjusted to fit the patient's amputation stump, but the patient developed a gingerly gait and circumduction gait, in order to reduce the weight load which caused pain on the affected side.\nRadiological examination found that the left tibia and fibula were amputated, the length of remaining tibia from the knee joint was 14.4 cm, and there was spur formation. Ultrasonography LOGIQ 5 PRO (GE Healthcare Korea, Seoul, Korea) revealed discrete hypoechoic mass (1.55×0.67×0.35 cm3) in the distal stump, directly continuous to the common peroneal nerve (), which was consistent with ultrasonographic findings of neuroma. When the neuroma was pressed using a linear transducer (10 MHz), extreme pain was produced again.\nThe patient received an information packet providing him with the risks and benefits of the procedure. We gave him plenty of time to ask any questions. After describing the procedure to the patient, we obtained written informed consent.\nWe performed the ultrasound-guided injection of dehydrate alcohol into the stump neuroma. The patient was placed in supine position, the region of injection site was sterilized with betadine and alcohol and reconfirming the location of neuroma using ultrasound guidance. A 22-gauge, 3.2-cm, short beveled needle was advanced into the neuroma under real-time control and used to inject 1.2 ml of 100% alcohol solution 100% dehydrated alcohol (Daihan Pharmacy, Seoul, Korea). Following injection, we observed the spread of alcohol within the lesion on sonography (). The pain significantly reduced immediately after injection and the patient complained of numbness around the injection site, but there were no specific side effects. During the outpatient follow-up observation, the patient complained of moderate reoccurring pain. The visual analogue scale was 7 points. Over 50 days after the first neurolysis, a second alcohol neurolysis was conducted using a smaller amount (0.3 ml) of alcohol, and subsequently the patient's VAS pain rating was reduced to 3 point. Thereafter, the patient has not experienced much pain or has encountered much difficulty in leading a normal life and walking with prosthesis. There were no side effects. |
A 48-year-old man complained of slowly progressed swelling in the anterior neck for 20 years. The patient had mild tracheal compressive symptoms and no dyspnea and dysphagia. He suffered no pain or voice change caused by the swelling. He denied a history of fine-needle aspiration (FNA) and trauma, and any cervical procedures or thyroid diseases among the family members were also excluded. This case report was in accordance with the ethical standards of Xingtai People's Hospital. Written consent was given by the patient for his information to be stored in the hospital database and used for research.\nA solid and well-circumscribed mass with a smooth surface (4.5 × 4 cm) was identified on physical and clinical examinations in the thyroid region. Routine blood test and thyroid function results showed no abnormalities. Ultrasound (US) scan revealed a hypoechogenic mass located in the right lobe of thyroid gland. CT scan showed a mass of 4 × 3.5 cm in the right lobe of thyroid gland with heterogeneous enhancement, which compressed the trachea toward the left side slightly. Consequently, the mass was preoperatively diagnosed as nodular thyroid goiter or thyroid adenoma. FNA examination was rejected and the patient received a right lobectomy of the thyroid. The intraoperative frozen section examination was performed.\nGross morphology showed a nodular lesion with clear margin and surrounding fibrous tissues. The cut surface showed many reddish-brown, sponge-like, minute cystic spaces. The bloody discharge was drained out of the nodule through the surgical incision. On histopathology, the lesion consisted of multiple irregular, dilated vessel lumens filled with red blood cells and hemorrhagic zone, resulting in the atrophy of the adjacent thyroid tissue (Fig. A, B). A benign lesion was diagnosed according to the frozen section. On paraffin section, microscopic examination revealed abundant proliferated vessels with irregular shape in the lesion. A monolayer of endothelial cells, without dysplasia, were located in the inner wall of the lumen, and the fibrous tissues found in the outer wall. Some red blood cells were also observed in the lumen. This thyroid lesion was characterized by positive endothelial markers (CD31, CD34, and FVIII), and negative epithelial markers (cytokeratin, thyroglobulin, and thyroid transcription factor-1) (Fig. C–F). On the basis of the above results, the diagnosis for this patient was primary intrathyroid cavernous hemangioma. The patient had been followed up for 10 months after surgery without complications and remained asymptomatic. |
We report the single clinical case of a 61-year-old right-handed male patient suffered a brainstem ischemic stroke caused by a thrombosis of the basilar artery and a dissection of the right vertebral artery at 55 years of age. He was treated with thrombolytic drugs, which proved ultimately ineffective and did not prevent the induction of irreversible stroke damage in several brain regions. T1 sequence magnetic resonance imaging (MRI) images acquired at the time of admission in the emergency room revealed a large lesion in the bulbo-pontine junction impinging on part of the left cerebral peduncle, and also small areas of ischemic damage in right cerebellar and left capsulo-thalamic structures. The patient was clinically diagnosed with an incomplete locked-in syndrome and tetra-paresia. Months following the stroke, the patient started several motor rehabilitation regimes aiming to preserve further functional loss and eventually improve the function of his four limbs, his face and trunk, favoring walking, equilibrium and allowing manual control of an automated wheelchair, ensuring for the patient a certain level of autonomy. After ~4 years of intense rehabilitation, the clinical status of the patient was characterized by a severe dysarthria and enduring tetra-paresia, impacting dramatically the left side of his body, including his left upper and lower limbs, and allowing only slight voluntary movements with his left fifth hand finger. In contrast, motion of his right hemibody parts was better preserved, allowing him to mobilize himself by operating an automated wheelchair with wrist flexion/extension movements. Regardless, his right hand opened incompletely and fine finger motor skills remained very limited, making manipulation of small items impossible. Additional rehabilitation regimes were implemented to improve swallowing difficulties (often conveying saliva and food to respiratory airways) and phonation disorders, which both endured regardless of the rehabilitation regimes. Oral communication was affected, nonetheless, the patient remained at all times fully able to understand verbal and written language and follow instructions. A neuropsychological assessment suggested significant memory encoding impairments, and limited attention and mental flexibility.\nForty-nine months (~4 years) following the stroke event, the patient experienced AHs as “someone speaking to him on his left ear” and reported such as the voice of his female speech therapist. He also suffered a continuous flow of persecutory AHs, described as voices professing insults addressed to his person. Nonetheless, AHs were not spontaneously reported to medical personnel by the patient himself but rather communicated by his relatives. Importantly, the patient did not show any prior history of psychiatric disorders or drug abuse before stroke. AHs initially disappeared spontaneously 3 months following the onset of such events. Nonetheless, only 9 months thereafter, AHs reappeared under an even more disturbing form, preventing the patient from sleeping and triggering unprecedented aggressive behavior. AHs were initially treated with neuroleptic medication (hemibody, 3 mg/day orally), however, this treatment was subsequently discontinued due to poor tolerance. Nineteen months later, the patient experienced again AHs, this time associated with a paranoid delusion and a depressive syndrome. Delusion and depression disappeared with antidepressant medication (Paroxetine, 20 mg/day orally), nonetheless AHs persisted.\nA previously tested therapeutic regime using low-frequency (1 Hz) rTMS delivered to the left TPJ (, ) was prescribed to treat hallucinatory symptoms reported as being highly impairing and disabling by patient’s relatives. The first 1 Hz rTMS session over the left TPJ was delivered 20 months following the onset of AHs and 69 months (i.e., 5 years and 9 months) following the stroke event. Figure indicate the different events (stroke, episodes of AHs, and rTMS therapy) presented in this clinical case. At the time of arrival, the patient was taking aspirin (Kardegic, 300 mg/day orally) to thin the blood and prevent clots; bromide pyridostigmine (Mestinon, 60 mg/day orally) for the treatment of myasthenia and intestinal tonus loss and a proton pump inhibitor (Lansoprazole, 15 mg/day orally) to prevent gastric acidity and reflux. |
A 70-year-old woman with a history of hypertension and diabetes mellitus complained of epigastric pain. During an enhanced computed tomography (CT) scan, a visceral artery aneurysm in the upper abdomen has been revealed. She was immediately referred to our hospital for treatment of aneurysm. Physical examination revealed a pulsatile epigastric mass. CT imaging revealed an 11×9 cm aneurysm, with a substantial mural thrombosis. The orifice of inflow into the aneurysm started 12 mm distal to the origin of the celiac artery, and the splenic artery originated from the celiac artery just proximal to the aneurysm (). Although the proper hepatic artery and the gastroduodenal artery have been determined to run very close to the aneurysm, the location of the outflow orifice was found to be less apparent. The preoperative diagnosis was aneurysm of the common hepatic artery with impending rupture. Emergent open surgical repair was undertaken due to uncertainty regarding the anatomical suitability for endovascular treatment.\nBecause of the size of the aneurysm, clamping of the proximal artery to block antegrade blood flow seemed difficult (); therefore, intra-aortic balloon occlusion technique was instead utilized. A 14 French (Fr) GORE® DrySeal Flex introducer sheath with a hydrophilic coating (W. L. Gore & Associates, Inc., Flagstaff, AZ, USA) was then inserted into the left femoral artery, and a 12 Fr Reliant stent graft balloon catheter for occlusion (Medtronic Vascular, Inc., Santa Rosa, CA, USA) was placed in the abdominal aorta at the level of the celiac artery. After the upper midline laparotomy, the surface of the aneurysm was confirmed through gastrohepatic omentum (). The occlusion balloon was then inflated, and disappearance of pulsation of the aneurysm was confirmed (). We did not perform an occlusion test of the arterial supply to the liver because it has been shown that vascular occlusions of the liver can be extended safely for up to 60 min. Gross examination of the aneurysm showed no inflammatory or posttraumatic changes such as wall thickening or dense adhesion to the surrounding tissue. Then, the aneurysm was incised, and mural thrombus was removed. The inflow orifice was located on the dorsal side of the aneurysm, while the outflow orifice was located on the right side. Some bleeding was observed from the outflow orifice of the aneurysm, which was taken care of by finger compression of the orifice.\nDirect closure of the inflow and outflow orifices was easily performed from the inside. The balloon occlusion time was determined to be around 9 min. The continuity of the proper hepatic artery and gastroduodenal artery was confirmed visually. After visual inspection to ensure that the liver showed no ischemic change, the laparotomy was closed. Intraoperative blood loss was determined to be at 450 mL.\nThe postoperative course was determined to be uneventful except for a slight elevation in hepatic enzymes with maximum aspartate transaminase 121 IU/L (reference range 13–30 IU/L) and maximum alanine transaminase 110 IU/L (reference range 7–23 IU/L). The patient was discharged home on postoperative day 12 and continued to be well throughout the 3-year follow-up after surgery.\nHistopathological examination of cross sections of the aneurysm wall has revealed thickening of the intima with fibromyxoid hyperplasia, and dense fibrosis of the media and adventitia with neovascularization and focal residual thin layers of smooth muscle cells of the media in the subintima (). The internal elastic lamina had mostly disappeared with a few residual fragments. Myxoid change has been observed in the residual thin smooth muscle cell layers (), and Alcian blue staining was able to reveal the deposition of a mucosubstance in all layers of the aneurysm wall (). Typical atheromatous change was not obvious. Focal lymphoplasmacytic infiltration was then observed in the adventitia, and CD138 immunohistochemistry confirmed the presence of plasma cells; however, these cells were found to be not immunoreactive for IgG4. No active inflammation with fibrinoid necrosis or granulomatous change with any multinucleated giant cells was observed. Consequently, atherosclerosis was not identified to be the main cause of aneurysm; furthermore, associating aneurysm with IgG4-related disease or systemic vasculitis syndrome was deemed not plausible. The pathologic findings were not consistent with fibromuscular dysplasia. Association with infection, trauma, drugs, and any collagen diseases or Kawasaki disease was also not supported clinically. Thus, considering the myxoid degeneration of the residual medial smooth muscle layers and the deposition of a mucosubstance in all layers of the aneurysm wall, any degenerative abnormality of the media that impaired the elasticity of the arterial wall would be the likely etiology for the development of the aneurysm. |
A 66-year-old male presented with pain and edema of the right leg for 2 d.
Pain and edema of the right leg for 2 d prior to hospital admission, no fever, chest pain, or dyspnea.
Multiple soft tissue injuries caused by falls 12 d before admission.
The patient had no remarkable personal or family history.
Swelling of the right lower extremity with palpable pitting edema, normal skin temperature, and a good arterial pulse within this extremity.
At the time of admission, the patient had a D-dimer level of 2789 ng/mL (NG, 0-500 ng/mL).
At the time of admission, color Doppler ultrasonography revealed right iliac-femoral vein thrombosis with decreased distal blood flow velocity. In order to accurately assess the anatomy of the retroperitoneal venous system in this patient, contrast-enhanced computed tomography was conducted after the thrombolysis procedure, which revealed that the right common iliac vein was located behind the left common iliac artery and was clearly compressed and stenotic. Many collateral vessels had formed in the pelvic cavity, and the left iliac vein was significantly wider than that on the right side. Abnormal left-sided IVC drainage was the result of the union of the two common iliac veins at the level of the 4th lumbar vertebra. It ascended vertically to the left side of the abdominal aorta where it connected with the left and right renal vein at the levels of the 2nd and 3rd lumbar vertebrae, respectively. The right renal vein additionally crossed posteriorly to the abdominal aorta. The left-sided IVC continued to ascend and intersected with the hemiazygos vein at the level of the 2nd lumbar vertebra, after which it crossed posteriorly to the left diaphragm crura and entered the thorax. It then ascended through the posterior mediastinum to the left of the four inferior thoracic vertebrae, crossing posteriorly to the thoracic aorta and the azygos vein at the level of 8th thoracic vertebra, arching over the root of the right |
A 35-year-old woman with no past medical and surgical history presented to the emergency department with severe colicky right flank pain radiating to right lower quadrant. The patient reported that the pain started two weeks previously and had been worsening since the last few days.\nA complete history was taken and physical exam performed, along with comprehensive laboratory and radiological evaluation. Elevated white blood cell (WBC) count with borderline renal function was noted, and an abdominal computed tomography (CT) was done without intravenous contrast (Figure ). This demonstrated a calcified 1.5 cm mass next to anterior aspect of right psoas muscle, which was misdiagnosed as possible ureteral calculus, and a presumed diagnosis of obstructive uropathy was established.\nAfter the diagnosis was made, the patient was admitted to the urology service, actively resuscitated with fluids, given pain medication, and observed. She deteriorated during the course of admission with increasing fever, chills, and worsening pain. We obtained repeat blood work along with the cultures from blood and urine, and the patient was immediately taken to the operating room for cystoscopy, retrograde ureterography, and ureteral stenting as a temporary measure of managing obstructive uropathy. The patient was taken to the operating room where cystoscopy revealed orthotopic ureteral orifices with unremarkable bladder architecture. An intraoperative stent was placed, and fluoroscopic examination revealed the presence of extra-ureteral lesion with calcification and the absence of any filling defect. The ureter ran medial to the calcification, and the J stent was left in situ. She was informed of her findings and was advised to have further investigation and management by excisional biopsy. However, the patient left the hospital against medical advice.\nShe returned to the emergency room after three months, complaining of persisting right flank pain that was on and off, and was subsequently re-admitted to the urology service. Repeat CT scan confirmed the persistence of the mass. She consented for diagnostic laparoscopy and possible excisional biopsy of the mass. She was subsequently taken to the operating room where a laparoscopic evaluation was done, and the right retroperitoneal mass was noted (Figure ), which was subsequently resected without any complications or difficulties. All intra-abdominal structures were evaluated and were unremarkable.\nThe patient was transferred to the post-anesthesia care unit (PACU) and was given optimal postoperative care.\nThe specimen was dissected, and on removal, we noted an ellipsoid mass with a thick capsule which was beige in color, with a septae compartmentalizing a darker center. The proximal end had a indurated pellet of uncertain composition. We sent it for further histopathological exam which reported right retroperitoneal mass with changes consistent with pseudoaneurysm wall (Figure ). The specimen was extensively collagenized and partially calcified without any evidence of ectopic organs or neoplasia. |
A 9-year-old girl visited the department of pedodontics and preventive dentistry with the chief complaint of throbbing pain in the upper front tooth. The patient's mother gave a history of trauma before 1 year. The tooth had fractured but was asymptomatic. The patient had visited a dentist for a fractured tooth and had a tooth-colored restoration done, which had fractured within a week thereafter. The patient had a throbbing pain in the upper front tooth since 2 days, especially on lying down; hence, she reported to our department. General examination revealed right-hand thumb agenesis with no significant known medical history. Intraoral examination revealed a single central incisor at the maxillary midline having a fractured incisal edge involving enamel and dentin []. No dental avulsions or tooth extractions in the region of the incisors were reported. In addition, she had maxillary atresia, the philtrum and incisive papilla were indistinct [], and the maxillary frenum was absent. Both parents were of normal stature and were healthy. There was no history of consanguinity or hereditary disease in her family.\nOrthopantogram showed a single central incisor at the midline and all other permanent teeth were present except third molars []. A radiographic investigation by a posteroanterior view [] also confirmed the presence of a single central incisor in the midline and suggested deviated nasal septum toward left and a narrow pyriform aperture. Chromosomal analysis by karyotyping was advised and if required, genetic counseling was planned after the analysis. In addition, other systemic disorders were ruled out and intellectual ability seemed to be average after pediatric consultation. Karyotyping with G-banding of peripheral blood cells suggested no major structural and numerical abnormalities of the chromosomes [ and ].\nAs karyotyping results were normal, the 9-year-old girl turned out to be a case of SMMCI without associated systemic involvement due to major structural and numerical defects of chromosomes. This case may be sporadic because there was no evidence of consanguinity or any other defects in siblings or family members. Root canal treatment followed by full coverage restoration was given in this case [ and ]. She was satisfied with the appearance of her teeth and was not aware of her orthodontic problems.\nOrthodontic and prosthetic treatment options are possible. As there was generalized spacing in maxillary anteriors and the patient was in mixed dentition, fixed orthodontic treatment would be considered at a later stage. After orthodontic alignment, prosthodontic replacement of missing central incisor with a removable partial denture could be done. After relative cessation of growth, i.e., around 18–20 years, fixed partial denture or a single tooth implant could also be considered. Preventive care, orthodontic treatment, and esthetic restorative dentistry are important in long-term dental management.\nHence, she was advised regular follow-up every 6 months and she continues to be followed by pediatrician and genetic consultant. After 3 years of follow-up [], the patient has no complaints regarding the esthetics of her teeth. Furthermore, she has showed progressive growth and development, further ruling out any systemic involvement. |
A 21-year-old woman presented with left upper quadrant pain. She underwent routine blood tests and non-contrast computed tomography (CT). The blood tests did not show any abnormalities, and the CT showed the presence of three, similarly sized spleens, but no other abnormalities. As the patient did not have any other symptoms, she was sent home with a prescription for an analgesic. However, the abdominal pain did not improve and she returned to the hospital 2 days later. Her inflammatory markers were somewhat elevated, and an enhanced CT showed that one of the multiple spleens did not pick up the contrast (Fig. a). We diagnosed her with splenic infarction; however, the cause of the infarction was unclear, and torsion or embolism was considered possibilities. The patient was admitted and began conservative therapy, including fasting and antibiotic administration. However, neither her abdominal pain nor inflammatory marker levels improved (Fig. b). Hence, we performed a follow-up enhanced-CT scan, 2 days after admission, which showed that the splenic infarction had not improved and that ascitic fluid was present around the spleen and in the pelvic space (Fig. ). At this point, we decided to surgically remove the infarcted spleen. Considering that the patient was a young woman, we elected to perform a laparoscopic splenectomy after receiving informed consent.\nThe surgery was performed under general anesthesia, with the patient in a supine position and her legs spread apart. We created an umbilical incision and inserted three operating ports along the left subcostal margin (5 mm, 12 mm, and 5 mm in size), and a 5-mm operating port on the left side of the abdomen (Fig. ). The port sites were selected along the lines of a left subcostal incision, in case conversion to open surgery became necessary. These port sites were also in a co-axial position to the surgeon. There were no adhesions observed in the abdominal cavity. First, we incised the omentum and opened the bursa, detecting two non-infarcted spleens in front of the pancreas. Behind these spleens, there was an infarcted spleen surrounded by fluid. We incised the inflamed adipose tissue around the spleen to expose the pedicle, which was twisted; consequently, we diagnosed splenic torsion (Fig. a). Using an automatic suturing device, we dissected the pedicle of the infarcted spleen. The umbilical incision was extended to remove the resected spleen (78 × 57 × 35 mm) (Fig. b). After confirming the absence of active bleeding, we sutured the incisions. The surgical time was 119 min, and there was little blood loss. The patient did not experience any complications and was discharged 4 days after surgery. |
A 56-year-old male with no history of allergic symptoms reported to the department of periodontology for the removal of a well-placed implant with prosthesis in relation to upper left central incisor. The patient's history revealed implant placement in the left upper central incisor region 6 months back by private practitioner after which the patient noticed generalized eruptions involving almost all the surfaces of the body including axilla, groin, chest, shoulder, forearms, hands, within a week of dental implant placement [] with no obvious peri-oral signs and symptoms. Following this he first consulted a dermatologist after 10 days of implant placement, who considered this a skin problem and diagnosed it as generalized pruritus. Raised eosinophilic count was present in hematological reports after 2 weeks of implant placement. He was prescribed antihistaminic (H1 antagonists) as an anti-allergic, but the eczema did not resolve. Skin biopsy was advised to rule out dermatitis herpetiformis and gliadin antibody (IgG, IgA) along with tissue transglutaminase (Ttg) test after 2 months to rule out celiac disease after observing continuous eruptions without much relief with anti-allergic as well. All tests reported negative. There were no significant pathologic findings observed around the implants [], and radiographically, the surrounding bone appeared normal. The patient was systemically healthy, with no history of medications or other suggestive medical treatment. The patient went to numerous skin clinics and hospitals and had taken anti-allergic medication, local and systemic steroids, and obtained only symptomatic relief but complained of the eruptions again after stopping the medications. The patient had also reported back to the private clinic after 4 months and requested for implant removal for which dentist convince him not to get it removed considering no perioral signs and symptoms and placed the crown as well. With regular medications and consulting dentist and dermatologists, he finally visited to the department of periodontology for the removal of implant. Considering the clinical symptoms, laboratory investigations and opinions from the departments of dermatology and medicine, a diagnosis of titanium implant allergy was made, and the patient was advised to go for the patch test before the removal of implant. The informed consent was obtained for the retrieval of the implant and for displaying the clinical images of the patient. After denial of the consent for the patch test by the patient, the implant removal was done atraumatically. The anti-allergic medications were stopped after 1 week as there was considerable relief in symptoms. The patient was rehabilitated with removable partial denture for the esthetic reasons. At 6th-month follow-up, the lesions on all the body surfaces disappeared completely, and there were no signs of itching or pain []. The patient exhibited remarkable progress with complete remission of signs and symptoms. |
A 71-year-old male patient was transferred to our department due to soft tissue defect in the left lower leg and infected Achilles tendinitis. The patient underwent incision and drainage of both lower legs with necrotizing fasciitis, at another hospital two months ago. Continuous wound care was performed; however, the left leg open Achilles tendinitis and soft tissue defects were not resolved.\nPhysical examination revealed a 12 × 5 cm wound with exposed Achilles tendon over the posteromedial aspect of lower one-third of the leg (Fig. ). His wound culture grew methicillin-resistant Staphylococcus aureus (MRSA).\nWe performed an operation with the patient placed in the supine position. All infection associated with necrotic Achilles tendon in the proximal muscle tissue was excised (Fig. ). After debridement, the patient had a 16 cm tendon defect from the muscle with the ankle joint in neutral position. He had 2 cm of the distal tendon attached to the calcaneus. We extended the skin incision to the outside of the zone of injury in the anterior aspect of the ankle, dissected anterior tibial artery and vena comitantes to perform vascular anastomosis out of injury zone. We made a template with surgical glove, which included the vascularized fascia lata for the reconstruction of Achilles tendon (Fig. ). We used the already manufactured template on the ipsilateral thigh, centering the flap over the perforator and drew the flap larger than the recipient site (Fig. ). A 14 × 7 cm ALF flap with a large piece of fascia lata (bilaterally, approximately 2 cm extra fascia is taken) was harvested (Fig. ). The donor defect was closed primarily over a silicon drain. For the reconstruction of tendon, the fascia lata was repaired first using multiple figure-eight sutures and modified Becker method[ with 4–0 prolene sutures at the separated end-to-end of the Achilles tendon. An end-to-end microvascular anastomosis was performed between the anterior tibial vessels and the flap pedicle vessels using 9–0 sutures microscopically after inserting the flap into the defect (Fig. ).\nPostoperatively, the ankle and leg were wrapped in a bulky dressing and immobilized with an above-knee splint and the flap was monitored intensively for 7 days. Anticoagulation therapy with prostaglandin E1 (10 μg/day) and heparin (5000 units/day) were administered for 1 week and aspirin 100 mg once a day for 4 weeks after the surgery. The flap survived completely without complications. Passive and active exercise of the ankle joint was started at 6 weeks after surgery. Subsequently, the patient underwent a graduated rehabilitation program, from a non-weight bearing exercise to partial-weight bearing exercise. Twelve weeks after the surgery, the patient was permitted full-weight bearing with gait training.\nAt 12 months of follow-up, the patient was able to resume full daily activities, felt a little discomfort at the donor site after more than 2 h of hiking, but was able to walk without pain and without the need for support, also was able to squat, showed an ankle range of motion of 15° dorsiflexion and 45° plantar flexion, and the American Orthopaedic Foot and Ankle Society (AOFAS) score was 94 (Fig. ). |
The patient was a 60-year-old man. Abdominal ultrasonography indicated that he had an abdominal mass. He reported having perceived lower abdominal pain 7 months before the initial examination, but it spontaneously remitted. The patient had no fever nor any history of conditions such as hypertension. A blood test revealed that he did not have anemia, thrombocytopenia, or coagulation abnormalities. The patient demonstrated slightly elevated levels of carcinoembryonic antigen (4.7 ng/ml) and soluble interleukin-2 receptor (603 U/ml), tumor markers that are elevated in gastrointestinal cancers and malignant lymphoma. Contrast-enhanced CT showed a 33× 31 mm abdominal aortic aneurysm and a 106 × 81 mm mass peripheral to the abdominal aorta. The mass did not feature a contrast effect, and its margin was enhanced in the venous phase (Fig. ). Sigmoid wall thickening and inferior vena cava thrombosis were also observed (Fig. ). We suspected the patient had a neoplastic disease, such as a malignant lymphoma or sigmoid colon cancer. Detailed examinations were consequently performed, and direct oral anticoagulant therapy was initiated to treat the inferior vena cava thrombosis.\nThe FDG-PET revealed an abnormal accumulation of FDG in the margin of the mass and the sigmoid colon, but not in any other organs. Colonoscopy showed only mild inflammation in the sigmoid colon, and a sigmoid colon biopsy revealed no significant findings. Chronic inflamed fibrous connective tissue was harvested from the periaortic mass using CT-guided biopsy. There were no findings indicative of infection or neoplastic disease, thus precluding a pathological diagnosis.\nAt 1 month after the initial examination, intermittent claudication occurred, prompting the performance of follow-up contrast-enhanced CT scanning. On CT scanning, we found an arterial occlusion from the infrarenal abdominal aorta to the bilateral common iliac arteries, the right external iliac artery, and the right internal iliac artery (Fig. ). These findings indicated the potential progress of the tumor to the aorta. Via a midline incision, biopsies of the mass and the lymph nodes peripheral to the sigmoid colon were obtained. However, these biopsies showed only connective tissue with inflammation and fibrosis and did not yield any significant findings. Since the developments, we elected to resect the mass and perform bypass surgery for definitive diagnosis and treatment after 3 weeks from biopsy.\nSurgery was performed using a transperitoneal approach via a midline incision. Adhesion was observed peripheral to the abdominal aorta, the bilateral iliac arteries, and the retroperitoneal mass. The abdominal aortic lumen was completely occluded by an old thrombus and plaque. Removal of the thrombus revealed a 35-mm intimal defect consistent with the direction of the retroperitoneal mass (Fig. ). Communication was observed between the intimal defect site and the retroperitoneal mass, which was filled with a partially organized old thrombus. During the intraoperative rapid pathological examination, the tissue inside the mass was diagnosed as a fibrin clot. The tumor tissue was not assessed. The area from the infrarenal abdominal aorta to the right common femoral artery and the left common iliac artery was bypassed using a Y-shaped woven Dacron graft. Pathologic examination showed that the mass tissue was a partially organized fibrin clot, thus agreeing with our intraoperative rapid diagnosis. On the basis of the intraoperative and pathologic findings, the patient was diagnosed with CCR-AAA with abdominal aortic occlusion. His intermittent claudication improved, and he was discharged on day 19 after surgery. Six months after the operation, the retroperitoneal mass disappeared on CT. |
A 73-year-old Chinese woman with a history of hypertension presented with a one-month duration of fever and chills. There were no localizing symptoms to suggest a source for her fever. Physical examination was unremarkable.\nIn the past month before admission, she had received 3 courses of oral antibiotics with no resolution of fever. She was worked up extensively for pyrexia of unknown origin. Blood, urine, fungal, sputum AFB cultures, autoimmune and hepatitis viral screens were negative. Syphillic markers were negative. There was no evidence of a malignancy. Computed tomography of the abdomen and pelvis were normal.\nA transthoracic echocardiogram showed a severely dilated ascending aorta with an end diastolic diameter of 5.8 cm. There were no valvular vegetations. A contrast enhanced computed tomography aortogram revealed a large eccentric, cresenteric thrombus at the anterior aspect of the ascending aorta with two large ulcerations in its anterolateral aspects as a result of a chronic type A aortic dissection (Figures and ).\nIntra-operative findings showed a chronic Type A dissection with a large 3 cm tear involving the anterior and anterolateral aspects of the ascending aorta. There were large amounts of intramural thrombus extending from the aortic root to the origin of the innominate artery. The aortic valve and coronary ostia were not involved. A hemiarch replacement with a 28 mm Gleweave Vascutek Graft was performed with resuspension of aortic valve commisures. Bacterial cultures from the aortic thrombus revealed coagulase negative Staphylococcus aureus and repeat blood cultures were negative. Histology of the ascending aorta showed chronic dissection of the aorta with a split occurring in the outer third of the aortic media. There were atheromatous changes throughout the resected segment, with marked destruction of the elastic tissue framework which was particularly extensive at the dissection site. There were previous episodes of hemorrhage with varying degrees of organization and fibrosis. No granulomas or signs of vasculitis were visible.\nThe patient was treated with intravenous cefazolin for a 6-week duration and made good progress. |
A Japanese 26-year-old man without any relevant medical history had progressive refractory massive ascites of unknown origin. He shortly developed hematemesis. An endoscopy revealed spurting bleeding from esophageal varices, and he was referred to and then admitted into our hospital. He underwent emergency endoscopic injection sclerotherapy (EIS). The laboratory data on admission showed severe liver dysfunction without any suggestion of chronic liver diseases, such as cirrhosis. The hepatitis viral markers, including hepatitis B and C, were all negative. Color Doppler ultrasonography (US) revealed a heterogeneous enlarged liver, shunt vessels between his middle and right hepatic veins (MHV and RHV, respectively), and retrograde flow in MHV. Computed tomography (CT) is a more sensitive modality for showing parenchymal abnormalities. In our case, enhanced CT revealed much clearer heterogeneity of the liver with decreased attenuation in the periphery. It also showed shunt vessels between MHV and RHV. The latter was markedly dilated, and it seemed to have some flow into the IVC, whereas his left hepatic vein was obstructed. Furthermore, sagittal CT images suggested severe stricture of his IVC at the level of the liver. Angiograms confirmed marked narrowing of his IVC at the level of the liver (Figure ). Magnetic resonance imaging (MRI) revealed findings similar to those of the CT. From these findings, and the rapid onset of the clinical manifestations, we diagnosed this patient as having acute BCS. Within one week, our patient developed massive hematemesis again. Subsequently, he showed manifestations of acute liver failure, possibly due to "shock liver". Since we could not find a donor for deceased liver donor transplantation (DLDT), we decided to perform PTA first. A balloon catheter was inserted into his right femoral vein, and the stricture of the IVC at the level of his liver was dilated using a 12 × 40 mm balloon catheter. After PTA, the pressure difference between his IVC and his right atrium decreased from 250 mmH2O to 50 mmH2O, and color Doppler US showed that the blood flow in his MHV had recovered to the normal antegrade flow. His clinical manifestations, such as ascites, dramatically improved, and his liver function also gradually recovered with time. About one month after the PTA, an enhanced 3 D CT examination revealed that the heterogeneity of the liver, the IVC stricture at the level of the liver, and the liver dysfunction had markedly improved. The endoscopic findings of the varices were also alleviated. Our patient was discharged, and regular follow-up was performed.\nAfter one year, color Doppler US showed that the hepatic venous outflow had deteriorated again, despite our patient being given an anticoagulant after the initial PTA. Sagittal CT images again showed severe stricture of his IVC at the level of the hepatic veins. Our patient then developed massive hematemesis again due to a rupture of the varices. We performed emergency EIS, to control the bleeding. Although we performed PTA again, balloon dilatation was not effective this time. Doppler US showed that his RHV and MHV were completely occluded with regurgitation in his MHV, and thick shunt vessels from his MHV to RHV were noticed (Figure ). His clinical condition deteriorated over several weeks, including the development of severe hepatic failure. This time, our patient's brother volunteered to become a donor for LDLT. After the physician and surgeon obtained written informed consent from the donor, LDLT was performed using the left lobe of the brother's liver. Macroscopically, the recipient liver was swollen and reddish, indicating severe secondary congestion (Figure ). Microscopic examination showed evidence of impaired blood outflow, including congestion, coagulative necrosis, and loss of hepatocytes without severe inflammatory infiltrates associated with fibrosis development (Figure ). After the operation, our patient's general condition improved and his manifestations were dramatically alleviated. One year after the LDLT, our patient was discharged with his liver functioning well. The laboratory data indicated a healthy status compared to his status before PTA and surgery (Table ). Moreover, multi-slice CT imaging showed that the congestion of the liver had completely disappeared with normal flow inside the vessels (Figure ). |
A 12-year-old female child came to our emergency department with complaints of gradually progressive weakness of bilateral lower limb and difficulty in walking for 4 days. On examination, the child was conscious and oriented with a lower limb power of 2/5 and generalized areflexia. Upper limb power was 4/5 and there was no cranial nerve involvement. A provisional diagnosis of GBS was made while stool samples for polio were also sent. Since the day of admission, patient was noticed to have early respiratory muscle weakness in the form of paradoxical chest wall movement. IVIG was started on clinical ground at a dose of 0.4g/kg/day for 5 days, but on the fourth day of admission the patient left against medical advice to another health facility. Till that time, she was stable and was maintaining vitals on oxygen despite some respiratory muscle weakness. Investigations at admission included complete blood count, liver function test, kidney function test and the results were found to be normal. Chest x-ray at admission was also essentially normal. Stool sample was negative for polio virus. After 6 days after leaving our hospital, she was again brought to our facility. This time she was not having paradoxical respiration but had complaints of respiratory difficulty of 2 days’ duration with marked tachypnea and hypoxia. On taking detailed history, it was revealed that after they went to another tertiary care hospital, she was again given three daily doses of IVIG despite transfer summary from our hospital had details of IVIG treatment and was subsequently referred to other hospital as ventilator support was not available for the patient. Unfortunately, again in the private nursing home she received full dose of IVIG. So as per our calculation, she received 5.2g/kg of IVIG, which exceeds far from the normal therapeutic dose of IVIG in GBS. On examination, chest was full of crepitations bilaterally. Chest x-ray had ill-defined opacities in bilateral lung field with no cardiomegaly whereas arterial blood gas showed compensated respiratory acidosis. So on the basis of the clinical scenario and investigations, we made a presumptive diagnosis of TRALI. Broad-spectrum antibiotic was started; she was kept on high-flow oxygen through face mask and intravenous fluid. After 48h, patient showed improvement clinically and radiologically. Echocardiography did not reveal any abnormality and blood cultures were also sterile. She could be gradually weaned from oxygen by day 5. She was treated with 14 days of antibiotic therapy. She is under regular follow-up after discharge and completely asymptomatic at present with no neurological deficit or respiratory morbidity. |
A 50-year-old Sri Lankan woman complained of pain and increase in the size of a swelling on her anterior mandible, which had been present for over 1 year (Fig. ). This significant change in size was noted 1 month prior to presentation with accompanying numbness of the left half of her lip. She was otherwise healthy (not on any medication) and her past medical and social history (she did not have risk habits such as tobacco smoking, betel chewing, smokeless tobacco, and alcohol) did not reveal any significant contributions to her current presentation. She was a homemaker and had two children. Exploration of her family history did not reveal that a similar pathology affected her parents, siblings, and children.\nA general examination of our patient revealed no significant findings and all vital signs (blood pressure, pulse rate, temperature, and respiratory rate) were within normal range. A well-defined, 4 × 4 cm swelling with redness on the overlying skin was noted on her submental region towards the left side. On intraoral examination, extensions of the same swelling measured 2 × 1.2 cm in size and involved the 31, 32, 41, and 42 teeth. No skin involvement was noted and the swelling appeared to be ill defined. The swelling was bony hard in consistency. Neurosensory evaluation revealed loss of sensation for fine touch, pressure, and temperature in lower left-sided labial skin.\nThese changes of recent onset were suspected to be an infection of an odontogenic cyst of her mandible and a cone beam computed tomography (CT) was performed. The results showed an irregular radiolucent lesion involving most of her mandible from the 35 to the 47 region. In the anterior part, both buccal and lingual cortical bone erosion was seen from the 33 to the 44 teeth of the mandible (Figs. and ). Except in the anterior part, very little buccolingual expansion was seen that suggested a KCOT. The roots of the 34 and the 44 teeth were involved in the lesion but no root reabsorption was evident.\nRoutine hematological investigations were carried out (full blood count, serum electrolytes, and liver function test) with renal function test and the results were well within limits.\nAn incisional biopsy revealed a SCC arising from a preexisting KCOT. An ultrasound scan and a CT scan of the bilateral neck revealed no metastasis migration to the neck nodes (T4N0M0).\nFollowing incision biopsy, left-side neck dissection (levels I to III) and segmental mandibulectomy to include a bony and a soft tissue margin of 1 cm were performed. The bony and soft tissue defects were reconstructed with a titanium reconstruction plate and a pectoralis major myocutaneous flap. Her recovery was uneventful. She had mild discomfort during the first few months following surgery and adapted to the changes subsequently. Close to the 16 months’ review, the titanium plate was seen externalizing intraorally. She complained of a mild pain during wide opening of her mouth due to the reconstruction plate. She had been routinely reviewed in the clinic with 1-month review intervals and currently she has been disease free for the last 18 months; a free fibula flap is planned for the mandibular reconstruction.\nOn histopathologic examination the lesion was reported as a moderately differentiated SCC arising from a KCOT (Figs. , , and ). Complete excision of the lesion was reported in the biopsy and the left-side neck nodes of all three levels were negative.\nPostoperative radiotherapy or chemotherapy was not prescribed considering the histopathology findings (a timeline is shown in Fig. ). |
A 60-year-old female patient presented with complaints of pain in right upper abdomen and right lower chest during inspiration for 4 months. No history of fever and jaundice or any associated co-morbidities. Ultrasonography showed a 10 cm × 7 cm × 7 cm thick walled cyst in segment VIII of liver with multiple loculations, septations suggestive of daughter cysts and partially calcified cyst wall [].\nContrast enhanced computed tomography (CT) shows a multiloculated cyst in the segment VIII of liver with calcification at multiple sites in the wall and humping of the diaphragm []. The diagnosis being hydatid cyst WHO classification type 3 B.\nAs the hydatid cyst was WHO type 3B with solid matrix and few areas of calcification in the wall, PAIR was not possible. The location of the cyst was such that laparoscopic treatment was not feasible. The patient was already treated with albendazole tablet for 3 weeks.\nWe thought of an innovative technique, using a nephroscope for percutaneous evacuation of the cyst.\nThe procedure was done under general anaesthesia. Patient positioned with 45° with right side up, to gain an easy access to the cyst percutaneously. Ultrasonography [] was to identify the cyst and a 18 Fr spinal needle was used to puncture the hydatid wall through the midaxillary line. The cyst was punctured where it was closest to the parietal wall. After puncture intracystic position of the needle was confirmed by aspiration and a guide wire was introduced using Seldinger's technique.\nMetal Dilators of increasing size from 9 to 27 were introduced over the guide wire. After sufficient dilation of tract, an Amplatz sheath was slided over the dilators and the metal dilators were then removed. As the dilators were removed there was a gush of clear fluid with white membranous material.\nThrough the Amplatz sheath suction was then introduced in the cavity and fluid with pieces of hydatid membrane were sucked out. 3% normal saline (NS) was introduced into the cyst cavity and kept in for 20 min, then sucked out. A nephroscope was introduced into the cyst and the hydatid material was evacuated under vision [Figures and ].\nThe cavity was repeatedly irrigated with 3% NS. Suction irrigation of cavity was done under nephroscopic control until all daughter cysts and membranes were evacuated. Nephroscopic examination confirmed complete evacuation of cyst contents and inner wall and no bile noted into the cavity.\nCompletion ultrasonography confirmed an empty cavity. 28 no abdominal drain placed through the Amplatz sheath indwelling the cavity. Post-operative course of the patient was uneventfull. The drain on post-operative day 1 was 50 ml and 25 ml on day 2. Cavity was washed with 3% hypertonic NS twice in the post-operative period.\nOn post-operative day 2, a plain CT scan was done to confirm the absence of hydatid contents in the cavity. The patient was discharged on day 5 with drain in situ as it was draining 15–20 ml/day. On follow-up after 4 days, the drain collection was almost nil and hence the drain was removed.\nOn follow-up after 2 months, the patient was asymptomatic and a ultrasonography showed a completely collapsed cavity with no residual fluid or daughter cysts.\nSoni et al. have described a technique of single incision laparoscopic hydatid cystectomy as a curative approach for sement VII liver hydatid.[] |
A healthy Caucasian boy who was 11 years old presented with a spontaneous sudden pain that was localized in his right shoulder and unilateral upper arm (right hand was spared) in the absence of trauma. His family doctor prescribed paracetamol, but there was no relief of the pain. Therefore, he visited the emergency department where shoulder and upper arm radiographs show no fractures. However, an orthopedist recommended dressing and immobilization of the arm for 2 weeks. Ten days later, because of constant and persistent pain, the family doctor requested some laboratory tests. A blood cell count, levels of C-reactive protein, creatine-phosphokinase, and lactate dehydrogenase, urinalysis, and culture of a pharyngeal sample were normal. When the orthopedist removed the dressing, the boy presented with considerable weakness in the upper arm. The family doctor then recommended a pediatric rheumatological examination. There was pain to acupressure in the low cervical area with insertion and along the ridge of the right biceps with strength deficit of this muscle. This specialist doctor prescribed ex-juvantibus therapy with gabapentin 300 mg three times a day for 3 weeks as a neuroprotective action. The severe pain persisted and the muscle weakness worsened until appearance of paralysis. At this time, pain and muscle weakness also appeared on the right hand and his family doctor recommended hospitalization.\nIn our Pediatric Unit, the boy presented with an antalgic posture of the right arm that appeared adduct and outstretched with severe pain at acupressure of the right trapezium muscle, shoulder, and elbow. We gradually withdrew gabapentin because of problems of concentration as reported by the mother and we prescribed ibuprofen 10 mg/kg three times a day.\nA neurological examination showed normal sensory function, no cranial nerve deficit, no neural sensitivity defects, no motor deficit in the lower limbs, normal tendon reflexes, and absence of Babinski sign. Muscle weakness was apparent only in the right arm with a functional limitation. In particular, strength deficit of the biceps and flexor digitorum muscles was observed, suggesting a median nerve deficiency. No signs of deficiency in other muscle groups were evident, although the intense acupressure pain made accurate assessment difficult. Cerebellum-vestibular tests were negative, except for the index finger-nose test on the right side because of pain. The neurologist (an adult neurologist) mentioned an adult syndrome with the same symptoms as those seen in our child called NA, but she had never seen NA in children.\nWe then repeated blood examinations (complete blood count, C-reactive protein, creatine-phosphokinase, lactate dehydrogenase, serum protein electrophoresis, rheumatoid factor, and anti-nuclear, anti-nucleolar, anti-neutrophil cytoplasm and anti-Borrelia antibodies), which were normal. The orthopedist repeated shoulder and cervical radiographs, which were negative.\nThe shoulder and cervical MRI, performed to rule out expansive lesions of the cervical spine and cervical hernias, showed no abnormalities. In particular, MRI showed no signs of neuritis with regular signal intensity of bone parts of the shoulder, regular rotator cuff tendons, and normal tropism of the shoulder girdle muscles (see , , and ). There was normal signal intensity of examined vertebral metameres (from C1 to T8) and of the spinal cord.\nThen, NCSs and electromyographic syudies have performed. No brachial plexus lesions were observed. Therapy with steroids and vitamin B6 was recommended by the neurologist. In particular, NCSs showed normally evoked compound muscle action potentials and sensory nerve action potentials with preserved conduction velocity, distal latency, and amplitudes. There was symmetrical evocability of the F wave as shown by analyzing the sensory ulnar and median bilateral nerves and circumflex nerve, the deltoid muscle, the right motor ulnar and median nerves, stimuli at the wrist, elbow, arm, and axilla, the left ulnar and median nerves from elbow to wrist, in the ulnar and musculocutaneous nerves, bilateral biceps muscle, and Erb’s point. Needle electromyography showed no pathological resting potentials by analyzing the supraspinatus, deltoid, triceps, biceps, extensor index, and thenar and hypothenar eminence. An increased insertion activity of the needle was observed and voluntary activity of the muscles was poorly assessable because of antalgic contraction. Furthermore, motor unit action potentials were of normal amplitude, morphology, and duration. At this time, therapy with steroids and vitamin B6 was recommended by the neurologist.\nA physiatrist assessed all exams and symptoms and suggested the diagnosis of brachial plexopathy or NA. None of the patient’s family members ever had similar symptoms. Approximately 4 months after the onset of symptoms and after intensive bilateral physical therapy, the boy no longer had any symptoms. A slight strength deficit was still present in the right biceps and in the right flexor digitorum muscles. Five months after onset, he had complete regression of clinical signs.\nWritten informed consent for publication was obtained from a parent of the patient. |
A 61-year old woman presented to her primary care doctor complaining of chest pain. She had a past medical history of hypothyroidism and 6 years earlier was diagnosed with stage IIIC papillary carcinoma of the right ovary, for which she underwent surgery and chemotherapy. Imaging and laboratory testing confirmed recurrence of ovarian carcinoma, and she again underwent surgery and was placed on chemotherapy with paclitaxel and carboplatin. There was a good therapeutic response to this therapy; her CA-125 levels normalized, and she became clinically asymptomatic.\nPrior to her cancer, the patient had a long history of depressive symptoms treated with a variety of antidepressants. She reported mild depressive symptoms associated with recurrent headaches following a hysterectomy at age 35. She had been treated with several antidepressants with partial benefit, including nortriptyline, citalopram, paroxetine, trazodone, and venlafaxine. It is noted that the patient had a family history of depression and anxiety in her mother, who did not respond well to treatment with antidepressants. The patient was first diagnosed with a major depressive episode after her initial cancer diagnosis, which was followed by the deaths of both of her parents. She was treated with sertraline for several years with benefit, but had further headaches and discontinued the sertraline for 4 years. At the time of her cancer recurrence, she experienced an acute worsening of her depression and the new onset of severe anxiety. Additional stressors included her concerns about illnesses that her grandson and best friend were experiencing. At this time (after cancer recurrence), she was diagnosed by her psychiatrist with recurrent MDD and generalized anxiety disorder. She was treated with escitalopram 20 mg/day, augmented with buspirone 30 mg/day (4 months after starting escitalopram), as well as lorazepam 0.25 mg as needed for anxiety. Despite these therapies, she continued to experience significant depression and anxiety.\nNine months after her cancer recurrence and following normalization of her CA-125 levels, and 6 months after starting escitalopram, the patient was started on maintenance oral chemotherapy with niraparib 300 mg at bedtime. The patient reported a dramatic response in her mood after initiating treatment with niraparib, and she awoke the next morning feeling no depression or anxiety. She was very surprised to feel so good, as she had not experienced a day free of depression and anxiety since her cancer recurrence. This mood improvement persisted for approximately 2 weeks.\nUnfortunately, the patient developed side effects to niraparib, including insomnia, thrombocytopenia, and neutropenia. One month after initiation, niraparib was held for 2 weeks due to the thrombocytopenia. During this 2 week period, the patient described a worsening of her mood, particularly her anxiety. Niraparib was then restarted at a decreased dose of 200 mg/day. The patient again experienced a dramatic improvement in mood and anxiety—an effect that lasted for 10 days.\nNiraparib was temporarily held again 2 months later due to neutropenia, and was later restarted at the dose of 100 mg/day. Again the patient described experiencing a large improvement in her depression and especially in her anxiety when the niraparib was restarted. Again on this occasion, the improvement lasted about 10 days. Eventually, due to thrombocytopenia and neutropenia, niraparib dosing was lowered to 100 mg every other day.\nThe patient’s psychiatric medication regimen was adjusted during the same time period she was being treated with niraparib. While on niraparib, the patient was able to decrease her lorazepam usage down from 0.25–0.50 mg per day to 0.25–0.50 mg once a week. Several weeks after the niraparib was begun, the patient was weaned off escitalopram and buspirone due to lack of efficacy, and she was begun on desvenlafaxine 50 mg/day and mirtazapine 45 mg/day. Five months later, she was weaned off mirtazapine but stayed on desvenlafaxine 50 mg. The patient reported only partial benefit from these antidepressant medications, in contrast to the complete, albeit temporary, symptomatic resolution she initially experienced with niraparib.\nFor this report, the patient was interviewed by board-certified psychiatrist coauthor MNM approximately 10 months after her initial treatment with niraparib. MNM conducted a psychiatric interview and administered three self-report evaluations (Patient Health Questionnaire, PHQ-9; Generalized Anxiety Disorder-7 item, GAD7; Mood Disorder Questionnaire, MDQ). At the time of the interview, the patient’s cancer continued to be in remission. As a result of the interview and self-assessments, it was determined that her mood continued to be mildly dysphoric and anxious but was overall stable on 50 mg desvenlafaxine daily and 100 mg of niraparib every other day. There was no evidence of hypomania or mania in the MDQ, and also no evidence of bipolar disorder in the patient’s history. |
A 24-year-old male patient presented with a history of pain in the right knee for 2 years. He had a history of blunt injury by a gas cylinder over the inner aspect of the lower thigh about 2 years back. His pain started after this blunt injury, which was dull aching and occasionally increased in severity on exertion and walking long distance. However, he complained of increased pain for 1 month prior to his consultation in our clinic. He had a second episode of blunt trauma following which pain was present throughout the day which increased on walking even a short distance. He was unable to squat and sit on the floor due to pain. On clinical examination, he was found to have diffuse swelling on the medial aspect of the knee. It was tender to palpation [], and the margins of the swelling could not be demarcated clinically. Swelling turned prominent on flexion of the knee [Figure and ]. The skin over the swelling was normal. The patient was initially evaluated with X-rays and since the clinical diagnosis could not be made, the patient was further evaluated with magnetic resonance imaging (MRI), computed tomography (CT) scan, and ultrasonography (USG) screening. X-ray examination was unremarkable, and MRI was reported to have venolymphatic malformation over the medial femoral condyle with minimal extension into the medial femoral condyle and vastus medialis obliqus. All the other blood parameters such as erythrocyte sedimentation rate, C-reactive protein, and rheumatoid factors were within the normal limits. The patient was taken for diagnostic arthroscopy and excision biopsy. Through standard anteromedial and anterolateral portals, the knee joint was examined; there were no intraarticular bleeding tendencies until the lesion was probed. Multiple, small, dark maroon, grape-like multilobulated cystic tissue [Figure - and ] was identified in the medial gutter over the medial femoral condylar area and over the medial wall extending into the medial capsule and articular surface of the vastus medialis muscle. Rest of the knee corners were normal, and the lesional area was identified to be highly vascular [] with prominent vessels seen arthroscopically. The tissue was bleeding on probing, biopsy of the lesion was taken, and extension into the bone and vastus medialis area was debrided with shaver and radiofrequency (RF) probe []. RF uses rates of oscillations (rapid movements/vibrations in the back-and-forth motion) in the range of around 3 kHz to 300 GHz (which corresponds to the frequency of radiowaves, and the alternating currents which carry radio signals). RF usually refers to electrical rather than mechanical oscillations, but mechanical ones do exist. The process of removing all the tissues takes place with the RF probe. The main goal is to perform coblation, which is controlled ablation. Ablation is the removal or cutting of tissue, and this offers the form of cutting or removing tissue by delivering large amounts of energy or heat. First, a plasma layer of reactive particles that disrupts the tissue's organic bonds is applied which rapidly disintegrates the tissue. This allows for large amounts of tissue removal with minimal damage to the surrounding tissue. This plasma layer is created by a current flowing between closely spaced electrodes. It vaporizes a layer of conductive fluid, and then the plasma layer is formed which is very reactive with energized electrons and ions. Next, an ablative layer is established quickly, using high current density, and then coblation occurs. Hemostasis was achieved, and wound was closed over a drain. The postoperative period was uneventful, and the patient had very good pain relief after the surgery. He was asymptomatic at the time of final followup of 1 year. |
A 51-year-old male patient with recent-onset bilateral mild-to-moderate knee pain and progressive deformity just below the knee joints was referred to us during his in-patient treatment at the department of gastroenterology of our institution. The patient had a history of chronic pancreatitis related to the excessive alcohol consumption over the last 15 years. His pancreatitis was complicated with type II diabetes that required insulin treatment for the last 4 years. He had chronic diarrhea for the last 3 years with up to 7–8 defecations per day. The patient had no history of therapeutic steroid use.\nHis knee pain after activities such as walking, stair climbing, or kneeling first appeared on the right side and 1 month later on the left side. A progressive deformity in weight bearing (Fig. ) associated with swelling were the major complaints despite relatively painless progress. Upon physical examination, both knees had large effusions and slightly limited range of motion (a range of 5–120° of flexion was possible bilaterally). Mediolateral instability at the level of proximal tibia, not of the knee joint itself, was remarkable on both knees and more striking in weight bearing. Despite the progressive deformity and instability, clinical tests and his gait were relatively painless. Due to the progressive instability of both sides, the patient had a difficulty in walking and had to use crutches. All findings were more prominent on the right side.\nThe patient had stocking-glove-type paresthesia (decreased sensitivity to pain, temperature, and pressure) typical for all cases with polyneuropathy on both lower limbs particularly below the knee joint and decreased patellar and achilles tendon reflexes. On the anterolateral surface of the middle one-third of the left lower leg, there was old scar tissue secondary to diabetic ulceration. The common femoral arteries, the popliteal arteries, the posterior tibial arteries, and the dorsalis pedis arteries on both lower limbs were palpable on examination and the intensity of the pulse was graded as 3+.\nSerial standard X-rays of both knees revealed a progressive insufficiency fracture at the level of medial tibial plateau that was always more severe on the right side (Fig. ). CT and MRI examinations of both knees delineated a more distinct fracture line, dense bone marrow edema, edema of soft tissues around the knee, and joint effusion that also showed some constitutional signal changes (hypointense marrow signal on all T1 and T2 sequences) on distal femurs (Figs. , ).\nThe white blood cell counts and CRP of the patient obtained before surgery were normal. MRI findings before surgery were also confirming for ruling out osteomyelitis as a differential diagnosis.\nA DEXA analysis of lumbar spine and the right hip region revealed a generalized decrease in bone density. A high-degree sensorio-motor polyneuropathy (diffuse slowing of sensory and motor nerve conduction velocities) has been detected in an EMG examination of both limbs consistent with diabetic neuropathy. Laboratory and ultrasonographic findings disclosed no abnormality of thyroid and parathyroid glands.\nThe Knee Society knee scores were 49 and 38 points for the left and right knee, respectively, and the function score was 0 (house-bound, impossible stairs, crutches on both sides).\nOur initial method of choice for treatment was conservative, which consisted of protective weight bearing and brace usage. The insufficiency fracture that began from the medial tibial plateau progressed into an intra-articular level just lateral to the tibial eminences. Fragmentation and destruction coalescence also appeared. This destructive progression occurred in a relatively short period of time, only in 3 months from the onset of symptoms, and accentuated the instability (Fig. ).\nOur management with conservative measures failed related to the rapid progression in bone loss, deformity, and instability. In order to stop relentless destruction and to get pain-free and functional knee joints, we changed our treatment modality for replacement surgeries. Since relatively demanding and open-to-complication surgeries for each side, we planned to operate on in two separate sessions. We had to remove all necrotic bone from the tibial plateau and replace them with metallic augments and to use a long-stemmed tibial component. We implanted a current tricompartmental PCL-replacing-type primary prosthesis (NexGen Complete Knee Solution System, Zimmer, Warsaw, IN, USA) with an extension stem and metallic augments on the tibial component. As the amount of resected necrotic bone was more than expected, we had to modify and combine augments to replace the defect, although the prosthesis has not been designed for this type of modification. We did not use any bone graft or bone substituting material. Nor we did use a tumor-resection-type or a custom-made prosthesis to get a more functional and durable outcome. In order to compensate the removal of a large segment and support medial soft tissues, we preferred to use a hinged brace postoperatively.\nImmediately after the first surgery on the right side, we encountered a weakness of the ankle and toe extensors. We related this problem to the stretching of the peroneal nerve during extensive surgery. Fortunately, it resolved spontaneously within a week and did not postpone the scheduled surgery for the left side. After a 15-day interval, we performed the similar surgery on the left side. Despite the heel protection, most likely related to the pain insensitivity, a pressure ulcer occurred under the left heel and healed with wound care within 6 weeks after the operation.\nThe histopathological tissue examination was of significant importance in differentiating the neuropathic arthropathy from an isolated insufficiency fracture that was related to chronic mineral imbalance and osteonecrosis. It revealed joint surface cartilage has been destroyed in many areas, and the remaining areas of articular cartilage contained vertical clefts without obvious chondrocyte proliferation. Subchondral bone was highly osteoporotic. There was synovial hyperplasia accompanied by abundant fragments of cartilage and bony debris within the soft tissues around the joints, denoting a rapid breakdown of the joint. Presence of minimal inflammation with mononuclear cells was helpful for differential diagnosis from rapidly destructive inflammatory arthritis, such as rheumatoid arthritis. Osteonecrosis was not found. Considering the clinical findings, the histopathologic changes were accepted to be in accordance with neuropathic arthropathy (Charcot joint) (Fig. ). Intra-operative cultures did not revealed any microorganism.\nThree months after the surgeries, we discontinued the brace usage on the right side because no dynamic instability during gait persisted. We followed up the patient regularly up to 5 years. During the latest follow-up at 60th months, the patient could walk without any knee problems. He was brace-free and without any dynamic instability. He was able to flex both knees to 120° and had a full extension (Fig. a, b). But his walking capacity was limited related to the Charcot foot on the left side developed in last 2 years. Radiographically, the components of both knees were stable without any sign of loosening (Fig. a, b, c). At the latest follow-up, the Knee Society knee and function scores improved significantly. The knee scores increased to 95 and 85 points for the left and right knees, respectively. Function score was only 80 points as the Charcot’s foot-related walking problems limited walking capacity.\nInformed consent of the patient was obtained before we prepare this case report. |
A 64-year-old Caucasian female patient sought dental care due to injury in a swelling on the left lateral border of the tongue. The patient noticed a swelling about 3 years back which was progressively increasing in size and presented as an enlarged tongue. At present, the patient reports difficulty in speech and swallowing. Her medical history revealed type 2 diabetes mellitus (T2DM) and hypertension for almost a decade. She was taking medication such as metformin, gliclazide, and beta blockers for her systemic pathologies. She has no history of smoking or alcohol usage. Clinical intraoral examination revealed the patient to be a removable denture wearer with good oral hygiene status. A characteristic nodular lesion which was elastic in consistency was noticed on the left lateral border on the anterior two-thirds of the tongue [].\nAn ultrasonography of the lesion was requested which showed a solid area about 20 × 20 × 13 mm in its largest diameter, located in the left lateral border of the tongue. It showed regular, well-defined margins with homogeneous texture. A Doppler study showed some peripheral vessels associated with the lesion [].\nThe patient was referred to her physician for pre-operative routine examination following which, the patient underwent excisional biopsy under local anesthesia. The lesion was removed after dilatation and dissection of the surrounding tissues [].\nAfter irrigation, the surgical wound was sutured with 4.0 vicryl suture material. The patient received appropriate postoperative care and was discharged. The specimen was sent for pathological examination. Macroscopic examination revealed a nodular lesion measuring approximately 2.2 × 1.8 × 1.3 cm in its greatest dimension covered by a smooth, shiny, yellowish translucent capsule [].\nMicroscopic examination revealed expansive mesenchymal neoplasm composed of sheets of mature fat cells with typical fine vascular network, thick fibrous beams, and spindle cells permeating the sheets of fat cells. The subcapsular margins were free of tumor invasion. The features were consistent with a SCL [].\nThe post-operative follow-up demonstrated regression of the lesion with good tissue repair. No complaint in mastication and speech was noted. |
A 55-year-old male with a past medical history of bilateral popliteal deep vein thrombosis six months prior, hypertension and end-stage renal disease secondary to focal segmental glomerulonephritis developed acute onset of left testicular pain during hemodialysis. The patient was undergoing a hypercoagulation workup at the time for a renal transplant, and his warfarin had been discontinued six days earlier. The patient also reported a new left shoulder pain radiating down his left upper extremity beginning the evening preceding the acute testicular pain. Scrotal ultrasound with color Doppler showed poor blood flow within the left testicle, and bowel or fat was noted superior to the left testicle; see . The patient was taken to surgery for a scrotal exploration which revealed an ischemic left testicle and no evidence of testicular torsion. The left testicle was wrapped in a warm saline soaked laparotomy pad, while the contralateral side was explored. The right testicle and cord structures were normal, and a 3-point orchidopexy was performed. Attention was returned to the left testicle, and a small incision of the tunica albuginea revealed protrusion of hemorrhagic seminiferous tubules. A left scrotal orchiectomy was completed, and the patient did well during the immediate postoperative period. The patient was not restarted on warfarin in order to complete the hypercoagulable workup. The microscopic evaluation revealed interstitial hemorrhage and focal atrophy, with no evidence of malignancy.\nThree weeks later, the patient developed acute right sided scrotal pain with associated left lower extremity pain above the knee. A scrotal ultrasound revealed no blood flow, , and physical examination revealed an edematous right testicle that was very tender to palpation. The patient was taken to the operating room for scrotal exploration, and intraoperative findings included no evidence of torsion of the right spermatic cord but the testis was indurated, necrotic, and unsalvageable. A right orchiectomy was performed and no bleeding was noted at the excised edges of the cord suggesting thrombosis of the arteries. Pathologic evaluation again showed benign testis with acute interstitial hemorrhage and focal atrophy; see . The patient was started on enoxaparin sodium, warfarin sodium, and aspirin to prevent further ischemic events and an extensive hypercoagulation workup by hematology revealed no objective evidence for a hypercoagulable state. Laboratory results including C-reactive protein, erythrocyte sedimentation rate, perinuclear and cytoplasmic ANCA, Hepatitis B surface and core antibodies, Hepatitis C virus antibodies, cryoglobulins, Protein C, and Protein S were all within normal limits.\nThe patient was readmitted two weeks later with sudden abdominal pain and new erythematous skin changes of the abdomen. Initially, a rectus sheath hematoma was suspected, but a CT scan of the abdomen and pelvis with intravenous contrast showed nonspecific skin thickening, subcutaneous edema, and fat stranding throughout the anterior abdominal wall but no distinct hematoma. The abdominal skin progressively worsened over the next few hours to a 19 × 14 cm well-demarcated livedoid patch with a central irregularly shaped area of impending necrosis measuring 9 × 12 cm that was distinctly cold to touch. The patient also developed sudden onset of penile pain with swelling and darkening of the penile skin. On the genitourinary examination, the glans penis was dusky, swollen, and cold. The corpora cavernosa were flaccid bilaterally. The patient had no tactile sensation in the glans penis, but the rest of the penis was tender to palpation. A penile artery Doppler study revealed pulsatile penile artery flow and a penile brachial index within the normal range. Initially a diagnosis of warfarin-induced skin necrosis was considered, but the skin biopsy of the anterior abdominal wall contained thrombotic vasculopathy with leukocytoclastic vasculitis; see . The microscopic differential diagnosis included septic vasculitis, polyarteritis nodosa, Churg-Strauss syndrome, Wegener's granulomatosis, and rheumatoid vasculitis. The patient also experienced temporary loss of vision in the left eye that resolved after one hour. The slit lamp exam was normal except for a small area of hemorrhage. Based on criteria set by the American College of Rheumatology () and the histopathology of the cutaneous anterior abdominal wall specimen, the patient was diagnosed with polyarteritis nodosa, and high-dose steroids and cyclophosphamide were initiated.\nOne month later, the patient developed a wound infection at the site of the abdominal skin necrosis requiring discontinuation of the cyclophosphamide, and the methylprednisone was slowly tapered. The patient improved clinically with no further ischemic events at 6-month followup. |
A 42-year-old woman with no significant medical history presented to an outside facility with progressive right eye proptosis and diplopia on the left gaze over 2 years. An MRI demonstrated a right sphenoid wing mass with extension into the temporalis muscle and orbit. She was found to have normal visual acuity, no evidence of optic neuropathy, restricted right ocular motility, and proptosis on the right of 5 mm compared to her left eye. She was fully oriented with intact cranial nerves, normal ocular alignment, motor, and sensory function throughout. Gait was normal. She underwent a right orbitocranial approach for resection of the mass, requiring complete removal of the greater wing of the sphenoid and the lesser wing lateral to the anterior clinoid process. Her postoperative course was unremarkable and she was discharged home on postoperative day 4. Postoperative MRI demonstrated gross total resection of the mass and a superior orbital wall defect []; pathology was consistent with a WHO Grade I meningioma. She followed up in clinic on postoperative day 8 complaining of diplopia when both eyes were open, which was treated with patching. On ophthalmologic evaluation, she was noted to have oscillopsia related to orbital pulsations synchronous with her cardiac cycle. She underwent magnetic resonance angiography, which was negative for carotid-cavernous fistula. At the last follow-up 21 months postoperatively, the patient continues to experience pulse synchronous oscillopsia.\nA 42-year-old woman with hypothyroidism presented to our clinic with several months of lip and tongue tingling. MRI revealed an anterior skull base meningioma with extension into the left cavernous sinus. She did not undergo ophthalmologic evaluation preoperatively, as she did not have any visual complaints. She underwent a left one-piece orbitopterional craniotomy with complete removal of the sphenoid wing and lateral orbital wall and partial resection of the superior orbital wall and was brought thereafter to the Neuro ICU, where she was found to have a left trochlear nerve palsy, resulting in double vision, and hypoesthesia in CN V2-V3 distribution. Her postoperative course was complicated by seizures, requiring treatment with levetiracetam for 6 months. She was discharged home on postoperative day 4. At her initial postoperative visit, her diplopia had improved, and on ophthalmologic evaluation 4 months later, she was found to have oscillopsia related to orbital pulsations synchronous with her cardiac cycle []. A CT scan of the orbits demonstrated postsurgical defects in the superior and lateral orbital walls []. The patient was enrolled in school, and this prevented her from being able to read. At the last follow-up, 26 months after surgery, the patient’s oscillopsia had resolved and her residual diplopia was amenable to corrective lenses with prism. |
In June 2011, a 56-year-old male was referred to our department by head and neck surgeon in order to improve his upper denture retention and stability. The patient was diagnosed with a squamous cell carcinoma of the maxillary gingiva (T4N0M0) in May 2005 and underwent an extended left maxillectomy, an anterior and middle cranial base resection, a left ophthalmectomy, and a flap reconstruction using the rectus abdominis muscle were performed. On physical examination, a recessed deformation on the left side of his face could be seen because of the left ophthalmectomy. The function of the left levator palpebrae muscle was eliminated to the level of a slight elevation by using the frontal muscle. A metal plate was anchored to the inferior wall of orbit. The left ethmoid bone, inferior nasal turbinate, the maxilla, alisphenoid, medial and lateral pterygoid muscle were already excised during the mesh titanium plate reconstruction of the anterior wall from the maxillary orbital region. Intraorally, the left maxilla had been excised from the midline, with the rectus abdominis muscle flap sutured directly to the soft palate. The peripheral mucous membrane around the left upper lip was already scarred, without the oral vestibule, and the flap margin had moved along with the surrounding soft tissue. The 70 × 50 mm flap was sagging from its weight and was in contact with the mandibular molars, reducing the volume of the oral cavity unless dentures were worn. The maxilla was removed from the midline to the maxillary tuberosity, while the mandible was removed from the anterior border of the ramus to the coronoid process. Dead space was eliminated because the abdominal rectus muscle was placed from the anterior cranial base to the oral cavity during reconstruction (Fig. ). No expiratory leakage or food reflux was observed, and the rhinopharyngeal closure was maintained. Prior to performing surgery, there was no tumor recurrence or metastasis. The patient had a mouth opening of 43 mm, which we judged operable and then conducted the flap reduction and elevation under general anesthesia in Dec 2008. Informed consent was obtained from the patient’s parents prior to study initiation, and all procedures were performed in accordance with the Declaration of Helsinki.\nSurgical reconstruction was performed as follows:An incision was made from the buccal side of the sutured edge (scar) in the abdominal rectus muscle flap (Fig. ). We can conduct a vestibular extension at the same time by incising this position. The adipose tissue was peeled from the buccal side to slightly beyond the skin flap center while maintaining approximately 5 mm thickness. The adipose tissue was reduced using a radio knife (8 g) (Fig. ). When we reduce fat tissue, we must avoid perforating of the skin. The skin was incised directly above the zygomatic bone, with tissue separation (avoiding exposure of the plate) to enable easy visibility of the zygomatic bone. Subsequently, the subcutaneous tissue was peeled from the zygomatic bone to the oral cavity for tunneling. Three mini QUICKANCHOR® (Depuy Mitek Surgical Products, Inc. Raynham, MA, USA) anchors were placed in the zygomatic bone, and anchor sutures were drawn through the subcutaneous tissue to lift the skin flap. A modeling compound was used to shape the margin of the celluloid splint (Fig. ). The advantage of flap suspension using Mitek anchors is the simple operability, less anchor positioning limitation, and easier length adjustment of the thread for suspension, which lead to easier fixation of soft tissue without slackness as well as clinically sufficient strength for fixation of ligament and tissue. On the other hand, less than 4 mm thickness of the cortical bone for suture anchor fixation causes insufficient fixation, therefore, determining placing position on the bone for fixation is necessary. Consequently, due to the versatility, the position that is considered optimal for stronger fixation and more efficient suspension can be selected as the anchor placing position, while the periosteum, corium, and scar tissue that are thought the most suitable for maintaining the strength can be chosen for the suture thread. Regarding the anchor placing position in this case, we determined 3 positions on the zygomatic bone and sutured flap corium taking into consideration a complete maxillectomy had been completed, which resulted in being able to lift the flap outward and upward.\nPostoperatively, the color of the skin flap was normal without congestion or necrosis. The celluloid splint was removed 10 days after the surgery with no infection or necrosis observed in the skin flap. We can find only fat, scar tissue, not carcinoma in the reduced fat tissure. At 3 months postoperatively, epithelialization and scarring were observed on the border of the skin flap and buccal mucosa, with no wound opening. Next, a denture that was stabilized to the right residual teeth with a clasp made. This prosthesis had two double Akers cast clasps unilaterally to retain the prosthesis by the four remaining molars. The major connector used anteria paratal plate. The patient was quite satisfied to be able to masticate, form an alimentary bolus, and swallow without any teeth falling out. No re-sagging of the skin flap or wound infection was observed at 3 years postoperatively. Patient follow-up will be continued at our department (Fig. ). |
A 20-year-old woman presented to the Department of Advanced General Dentistry, Yonsei University, with chronic gingival swelling and bleeding on the left central incisor. A periapical radiograph of the left central incisor showed the level of tooth structure was approximately equal to the osseous crest and so was insufficient for restoration without invasion of the biologic width (). Orthodontic forced eruption followed by single ceramic restoration treatment was determined on the basis of esthetic and economic considerations.\nAfter careful bridge cutting and removal of the crown on the left central incisor (), periodontal treatment was performed. A temporary crown with a post was set after the condition of the previous root canal treatment was evaluated by an endodontist. However, considering the adjacent ceramic crowns, conventional methods of forced eruption had an increased risk of ceramic damage. A modified appliance needed to be fabricated.\nIn the laboratory, an artificial tooth was set on the left central incisor area on the model and a clear appliance was fabricated using a plastic sheet (Bioplast, Scheu-Dental GmbH, Iserlohn, Germany) in the anterior teeth area. The artificial tooth was removed from the clear appliance and trimmed to leave only the labial shell. The labial shell of the artificial tooth was attached inside the clear appliance for esthetic maintenance. A window was formed on the palatal side of the left central incisor area of the clear appliance. A braided fiber-reinforced composite strip of 2 mm in diameter (Fiber Force, Synca, Le Gardeur, QC, Canada) was bonded to the inner surface of the window as a scaffold to increase the strength of the anchor (). An elastic thread was engaged to the loop wire in the root and the horizontal bar of the clear appliance, and activated with a 50 g force after a piece of wire (0.032 inch in diameter) ending in a loop was affixed to the prepared root using glass ionomer cement (). The occlusal interference due to the appliance was controlled by cutting the plastic sheet on the contact area of the centric stops. To avoid food impaction or foreign body entrapping, the window was covered with temporary filling material leaving the tooth space empty ( and ).\nThe amount of tooth movement was 1 mm after 1 week, and 2.5 mm after 2 weeks, and the necessary movement of the tooth (approximately 3.5 mm) to achieve the biologic width had been attained after 3 weeks (). A two-month retaining period and gingivectomy was planned along with careful periodontal hygiene control, and a new ceramic crown was fabricated on the tooth in concern (). The functionality and periodontal support of the restoration have been maintained in good condition for 2 years (). |
A 15-year-old boy of average built presented to the orthopaedics department with a draining non-healing sinus at the lateral aspect of his right lower thigh for the last 9 months (Figure\n). Pain was minimal. The margins of the sinus were hyperpigmented and thickened with excoriation of the surrounding skin; pus was expressed out on pressure. Palpation was painful but did not reveal any mass around the sinus. Regional lymph nodes were enlarged compared to the contralateral side. Distal neurovascular status was normal. The knee joint was not swollen, and the range of motion was normal except for a mild terminal restriction of flexion due to pain. Detailed history revealed that about 9 months ago, he fell down from a tree which is approximately 15 feet in height and sustained injury from wooden branches at the right thigh. He also had transient loss of consciousness from the impact. After consciousness was regained, he noticed a small wound over the outer aspect of his right thigh. There was not much bleeding, and he considered it a minor injury. The wound, however, nearly healed. After about 3 weeks, he noticed swelling and frank pus started coming out of the wound. He sought traditional treatment and got some temporary relief. The amount of pus decreased in-between and the size also diminished, but complete healing never occurred. During the whole course, he did not develop any fever, loss of weight, cough, or chest pain. After 9 months from the day of injury, he visited our hospital.\nAfter admitting him for detailed evaluation, the differential diagnosis considered chronic osteomyelitis, fungal infection, tuberculosis, and implanted foreign body. Blood parameters and X-ray examination of the area came out to be normal and osteomyelitis was ruled out (Figure\n). Pus culture revealed growth of Staphylococcus aureus. FNAC obtained from the enlarged regional lymph node showed nonspecific findings. Implanted radiolucent foreign body was thought as a possibility for persistent discharge. Musculoskeletal ultrasonography was ordered and performed by a radiologist using a 5- to 7.5-MHz linear probe (HP image point Hx system) which readily showed a single, about 7- to 8-cm-long foreign body within the vastus lateralis muscle with surrounding edema (Figure\n).\nAfter an informed consent, the patient was taken for exploration of the wound under spinal anesthesia under a pneumatic tourniquet. The incision was made over the old scar incorporating the sinus tract and explored in a systematic manner. The vastus lateralis was approached after incising the deep fascia. Finger dissection of the muscle revealed the foreign body which was identified as a long piece of wood measuring about 1.5 × 7.5 cm (Figure\n).\nDeep culture and biopsy were taken. The sinus tract was curetted and washed with hydrogen peroxide and povidone iodine solution. The incised wound was closed, leaving the sinus open. We could not use intraoperative sonography, but ordered repeat ultrasound examination to confirm complete removal of the wooden foreign body. Biopsy taken from the soft tissues surrounding the wooden foreign body demonstrated nonspecific chronic inflammation. Deep culture showed S. aureus again. Fungal culture could not be done. IV antibiotics were continued for 2 weeks according to culture sensitivity report. Surgical removal caused the sinus to heal uneventfully within 3 weeks.\nIn various cadaveric studies\n[], a sensitivity of 94% and a specificity of 99% have been demonstrated for sonography as a diagnostic modality in fresh cases. Theoretically, a combination of ultrasound and X-ray films should allow for diagnosis and localization of virtually all foreign bodies. Sonography can be used effectively to locate wooden foreign bodies as small as 2.5 mm in length\n[]. Given that many foreign bodies are radiographically undetectable, the accuracy and availability of sonography make it an excellent modality for evaluation of radiolucent foreign bodies\n[,]. The imaging appearance of wooden foreign bodies is variable; however, imaging can be quite specific\n[], and when taken in the appropriate clinical setting, the imaging should reliably suggest the diagnosis. Wood is highly echogenic and reveals pronounced acoustic shadowing on sonography\n[]. Sonography is frequently underused but proved most useful for the evaluation of retained wooden foreign bodies\n[]. Radiographs failed to reveal the retained radiolucent foreign bodies in all (12 out of 12) patients in a published report\n[]. While most of the metal and glass foreign bodies can be detected by plain radiography, organic substances such as wood and vegetative materials are radiolucent, and unfortunately, these radiolucent foreign bodies are usually more prone to cause an inflammatory reaction and infection\n[]. The detection can be even more difficult in cases of penetrating injuries\n[] with small innocuous skin wounds. The detection of wood is especially important because it may serve as an unrecognized nidus for infection. Wood, with its porous consistency and organic nature, is an excellent medium for microorganisms, and the retained wooden foreign matter may result in cellulitis, abscess, or fistula formation\n[]. The wooden foreign matter may also result in synovitis if a joint is violated or in osteomyelitis if adjacent osseous structures become involved. Radiographs have been reported to reveal a wooden foreign body in only 15% of patients and 38% of retained foreign bodies in the soft tissues were overlooked at initial examination\n[]. Xeroradiography has been reported as slightly more sensitive than conventional radiography for the detection of retained wood; however, xeroradiographs show negative results in 80% of patients and are not available in most radiology departments\n[]. Sonography is indeed a reliable investigation for the detection of foreign bodies in soft tissue\n[]. Sonography has been well studied in the evaluation of retained foreign bodies and has been proved to be both sensitive and specific\n[,,].\nGiven the markedly different acoustic impedance of wood and soft tissues, retained wooden foreign bodies are easily identified by radiologists, with the leading edge of the echogenic wood resulting in marked acoustic shadowing\n[]. Retained foreign bodies in the soft tissue of the extremities that were initially overlooked and discovered later are unfortunately common\n[]. Localization of embedded foreign body may prove to be very difficult. Exploration under regional or general anesthesia with application of a tourniquet (whenever possible) has been recommended to provide a better operative view\n[].\nIncomplete removal is a possibility, and exposure should be adequate to avoid it. Sonography can be a useful therapeutic adjuvant in the management of wooden splinter or other foreign bodies in the extremities\n[]; ultrasound can confirm completeness of removal of the foreign body after surgery as well\n[]. Notably, the identification of wooden foreign bodies may be exceedingly difficult on magnetic resonance imaging (MRI), especially when foreign bodies are small and there is no associated abscess or fluid collection\n[]. When compared with MRI, computed tomography (CT) scan has the advantage of being less expensive, more readily available, and faster to perform. However, sonography has been proved to be effective only for superficial foreign bodies. Foreign bodies and their accompanying shadowing or reverberation may not be well visualized if they are located adjacent to the bone or deep to subcutaneous gas\n[]. X-ray examinations will miss radiolucent foreign bodies. Sonography allows detection of a variety of soft-tissue foreign bodies, including wood splinters, glass, metal, and plastic, along with evaluation of their associated soft-tissue complications\n[].\nUltrasound examination can readily demonstrate such ‘radiolucent’ foreign bodies. Such implanted foreign body can lead to infection. Ultrasound examination can readily demonstrate such radiolucent foreign bodies, but sonography is not commonly ordered by orthopedists and clinicians alike in traumatic conditions of the limbs unlike X-ray examination. Therefore, this participatory, real-time, cheap, and easily available diagnostic modality is often ignored as a screening tool by clinicians, and instead, there is tendency to order a high-end imaging modality like MRI or high-radiation-based modality like CT scan. This case implies that a non-healing sinus with normal bone finding in radiograph following old trauma should raise the suspicion of implanted foreign body/bodies which can be detected at bedside by office sonography instead\n[]. |
A 46-year-old white woman presented to the Hypertension and Nephrology Clinic with significant weight gain and edema. She reported concerns regarding her weight gain of 70 pounds over the last six months. She stated that she is an active person and believed her weight gain was primarily due to fluid retention. She received laparoscopic adjustable gastric banding in 2003, followed by an abdominoplasty in 2016. She reported she was doing well and lost a significant amount of weight after her surgeries until six months prior to presentation. She consulted her surgeon and endocrinologist, both of whom reassured her that her gastric band was intact without any complications. Since August of 2018, she has been taking furosemide 20 mg by mouth, once daily along with spironolactone 25 mg by mouth once daily. The patient has not found any significant improvement with these medications and reported oliguria. She denied any prior history of diabetes, hypertension, or kidney stones.\nHer past medical history was significant for chronic idiopathic abdominal pain, varicose veins, and edema. Her surgical history begins in 1978 for a tonsillectomy, laparoscopic adjustable gastric banding and cholecystectomy in 2003, a bariatric bypass in 2009, a colonoscopy in 2013 followed by an abdominoplasty in 2016, and an esophagogastroduodenoscopy in 2018. Her family history is only significant for unspecified cancer in her father. The patient reports an allergy to latex and denies alcohol or tobacco use. She admits to smoking cannabis on rare social occasions. Her laboratory results and vital signs were unremarkable. However, her BMI was 33.39 kg/m2 as she was 68 inches tall and weighed 219 pounds.\nShe was referred to the Hypertension and Nephrology Clinic to assess whether her edema was due to any renal abnormalities or conditions. Patient’s lab work demonstrated that her kidney function was normal as all her electrolytes and creatinine levels were within the reference range. Given our patient had no heart failure, liver dysfunction, kidney dysfunction, malignancies, or protein malnutrition, she was diagnosed with idiopathic edema. This patient was referred to a lymphedema clinic for further evaluation. |
An 80-year-old man was admitted to our hospital for the treatment of a rectal tumor found incidentally by rectal examination. The tumor, about 3 cm in diameter, was located on the right side of the lower rectum 3 cm above the anal verge. The pathological analysis of the biopsy sample revealed that the tumor was a moderately differentiated adenocarcinoma. Abdominal computed tomography (CT) and magnetic resonance imaging (MRI) indicated that the rectal cancer invaded into the muscularis propria without distant metastases and that lateral pelvic lymph node (LPLN) was not enlarged with a maximum long-axis diameter <3 mm. The most important problem was that the patient had a huge benign prostatic hypertrophy, the size of which was 85 × 80 × 70 mm (Fig. a–c). To achieve complete TME with negative CRM, a hybrid transabdominal-transanal approach for ISR was conducted.\nFirst, vascular division and mobilization of the left colon were performed laparoscopically. The transabdominal approach was continued until the anterior dissection of the rectum became difficult due to a huge prostatic hypertrophy. Next, the circumferential rectal incision and subsequent intersphincteric dissection were performed under direct vision to enable attachment of a single port device (GelPoint Mini; Applied Medical). After closure of the anal orifice, the GelPoint Mini was placed to start the transanal approach. Posterior side of the rectum was first dissected until the transanal approach was connected to the dissection layer made by the transabdominal approach. The dissection procedure was extended to the lateral side. Bilateral pelvic splanchnic nerves were identified at the 5 and 7 o’clock positions. At the anterior side, the proper dissection layer cannot be easily identified because of the perineal body and the enlarged prostate. Once the dissection plane between the rectum and the prostate could be identified, it was relatively easy to continue along the same plane. The assistance provided by the laparoscopic approach was useful to determine the appropriate dissection line in the transanal approach. |
Our case is of a 25-year-old male patient who came into the clinic for a dental check-up of tooth number 21 (maxillary left central incisor), which presented a gradual colour change of the clinical crown with regard to the neighbor teeth over the past three years. Although this tooth was completely asymptomatic, the patient reported that he had suffered dental trauma 15 years ago when he fell while practicing sports, resulting in a complicated crown fracture. The day after the accident, his parents took him to see a dentist who simply “placed a screw and sealed the tooth with tooth-colored material.” The medical history revealed no history of systemic problems or allergies.\nUpon clinical inspection, a resin was observed at the palatal area and incisal edge, and the buccal and palatal mucosa appeared normal. There was no response to vertical or horizontal percussion or to palpation of the mucosa. Probing depth and tooth mobility were both within normal limits, and the adjacent teeth responded normally to both thermal (Endo-Ice, Hygenic, USA) and electrical (Digitest, Parkell, USA) tests. The radiographic inspection revealed the presence of an intraradicular post at the crown level, which was surrounded until the middle third of the root by sealing material (). A wide canal and an immature apex were detected. In addition, a radiopaque foreign object (approximately 10 mm in length and 1.5 mm in width) was also detected in the apical region of the immature canal. This foreign object was also displaced approximately 5 mm into the periapical tissues and was surrounded by a diffuse radiolucent periapical lesion. The morphology of the foreign object was very similar to that of the intraradicular post identified at the crown level. In view of this finding and based on the history reported by the patient, we assumed that his previous dentist had not been fully aware of the association between the patient's age and the immature apex while performing dental treatment 15 years earlier and accidentally forced the foreign object that is intended to be used as intraradicular post through the apex and into the periapical tissues while attempting to remove it. Consequently, the dentist likely inserted another intraradicular post and sealed the access cavity with resin.\nOnce all of the aforementioned information was collected, a pulpal diagnosis of previously initiated endodontic therapy with signs of infection was established, with periapical diagnosis of chronic apical periodontitis combined with iatrogenic displacement of a foreign body into the periapical tissues. Thereby, we decided to perform a surgical endodontic treatment of the tooth, including the retrograde removal of the foreign object, retrograde instrumentation, and retrograde obturation with a thermoplasticized gutta-percha injection. We concluded that trying to remove the foreign object using an orthograde approach could weaken the fragile immature root structure and could risk sending the object into a deeper anatomic area, such as the floor of the nasal cavity. The intentional replantation was considered only a last option due to the high probability of root fracture at the time of extraction. The initial prognosis for this patient was favourable.\nAfter performing a regional anaesthetic block with 1 : 100,000 articaine and waiting 10 min for deep action, a total thickness triangular flap was raised and the root apex was located with the use of long-shank round carbide bur number 4 mounted on a high speed hand piece with no direct air to the working area, under abundant saline irrigation delivered with a hypodermic syringe. After locating and exposing the periapical lesion, all tissue within the bony crypt was removed with a curette to locate the foreign body (metal fragment) (), which was easily removed with a hemostat (). Because the tooth had an incomplete root formation, once the bleeding was under control with the use of gauze impregnated with ferric sulphate, the root apex was remodeled only slightly with a Zekrya bur at high speed (). Due to the fact that there was still no apex and the apical deltas were not yet fully formed, the apical reduction of the last 3 mm was avoided.\nResin was detected inside the root canal using a retrograde approach with an endodontic explorer; thus, there was no need for a retrograde preparation of the entire length of the main canal. The ultrasonic retrograde preparation of the 3 mm apical portion was performed using ultrasonic tips (Endo retrograde Kit, NSK, Brasseler USA). Low intensity and very light pressure were used to prevent the creation of microfractures in the thin walls of the immature apex, and abundant saline irrigation was provided drop by drop at a distance. After drying the retrograde cavity with absorbent paper points, the cavity was filled by retrograde obturation with thermoplasticized injected gutta-percha (Obtura System II, SybronEndo, Coppell, Texas, USA), which was compacted and its placement was confirmed radiographically once it had cooled (). The flap was approximated and sutured gently with 5-0 nylon separate stitches. Following the procedure, the patient was provided with oral and written postoperative instructions and drug treatment. The patient returned after three days for suture removal and did not report any unusual discomfort. The histopathological diagnosis of the lesion was periapical granuloma.\nOne month later, the patient returned completely free of any symptoms and signs of infection, and probing depth and tooth mobility were both within normal limits. It was decided to change the crown restoration to prevent the coronal leakage of saliva; however, the patient returned for a follow-up after 12 months, at which time proper radiographic healing of the periapical lesion was observed, and no symptoms or signs of infection were found () and clinically there was no clinical coronal leakage. The patient was advised of the need to change the crown restoration and was asked to return for a follow-up after completing the reconstruction. Unfortunately, the patient ignored the previous recommendation and returned for a follow-up 52 months later. In spite of this, no clinical symptoms or signs of infection were observed. Probing depth and tooth mobility were again within normal limits, and radiographs revealed adequate periapical healing (). Therefore, based on the extensive follow-up period and the positive clinical and radiographic features, we considered the patient healed. |
A 4-year-old boy, born in Zhejiang Province of China, was admitted to Yuying Children's Hospital affiliated to Wenzhou Medical University in June 2014 with complaints of productive cough accompanied with high fever for 5 days. He was the first-born child to unrelated healthy parents, born at 38 weeks of gestation after an unremarkable pregnancy. His birth weight was 3.5 kg, and meconium was passed on the first day of life. The patient had no history of meconium ileus or diabetes mellitus and lacked family history of CF. Tracing back his medical history, the patient was formula feeding but failure to thrive with a weight of 6.8 kg at the age of 8 months and had intermittent diarrhea. For further evaluation of the condition of growth and development, the patient was taken to a local hospital at the age of 8 months, and received complete blood count and liver function tests. And the results indicated liver involvement with slightly elevated alanine aminotransferase (ALT) and aspartate aminotransferase (AST) with values of 78 and 82 U/L, respectively. The patient suffered from recurrently and slightly elevated ALT levels ranging from 70 to 92 U/L and AST levels ranging from 80 to 90 U/L. In addition, the common etiologies that easily lead to increased levels of ALT and AST were also excluded, such as cytomegalovirus and hepatitis B virus infection. Initially, these symptoms were not paid enough attention by the physicians or parents because the elevated levels of ALT and AST can recover to normal levels automatically without treatment or through the injection of magnesium isoglycyrrhizinate before four years of age.\nPhysical examination for the patient at the age of 4 years showed a weigh of 16.5 kg with a height of 104 cm. The patient had a BMI of 15.3, which was in the 50th percentile for his age. The physical examination also revealed tachypnea and a barrel-shaped chest. The liver was palpable ~2 cm below the right costal margin, and the spleen was palpable about 1 cm below the left costal margin. Clubbed fingers were absent. Laboratory examination indicated increased ALT and AST values of 93 and 92 U/L, respectively, whereas other markers such as γ-glutamyl transferase (GGT), bilirubin, bile acid, fasting blood glucose, albumin, and globulin were within normal limits. Other laboratory investigations including of complete blood count, serum electrolytes, urine, arterial blood gas, amylase, and lipase were normal. The sputum and bronchoalveolar lavage fluid cultures tested positive for Pseudomonas aeruginosa. The Sudan III dye test of fecal matter indicated fat droplet positivity. Pulmonary function tests failed to be performed because of the difficulty at this young age for the child. Additionally, the lack of laboratory facilities caused impracticability of the sweat chloride test. Utilizing computed tomography (CT), we identified severe bilateral paranasal sinusitis () and diffuse fatty infiltration of the liver () in the patient. In addition, the chest CT scan verified the presence of bilateral bronchiectasis and marked peribronchial thickening, especially in the middle and lower lobes (). Extensive sticky and purulent secretion were observed in the lungs by bronchoscopy (). Based on the aforementioned pathological findings, the patient was primarily diagnosed with CF.\nGenetic testing of the patient revealed a homozygous nonsense mutation from a C-to-T substitution (c.1657C > T) in the CFTR gene, which was inherited from both his father and mother (). This single-nucleotide variant changed an arginine at position 553 into a premature termination codon (p.R553X). Notably, CF screening using amniotic fluid of the mother during her second pregnancy also indicated the fetus (sibling) to be a p.R553X carrier.\nOther than hypertonic saline nebulization, high-frequency chest wall oscillation, expectorant administration, pancreatic enzyme replacement therapy, and supplementation with vitamins A, D, E, and K, the child was prescribed intravenous ceftriaxone to address the P. aeruginosa. Respiratory symptoms gradually improved after 7 days of treatment, and he was discharged on the 15th day after admission. Ursodeoxycholic acid was prescribed after confirmation of genetic diagnosis, but taken irregularly by the patient. Therefore, the medicine failed to bring about the desired effect. Remarkably, the patient was later re-hospitalized two times because of pulmonary infections and liver involvement. Liver function test showed that the levels of both ALT and AST ranged from 90 to 120 U/L. Further examination of abdominal CT and ultrasound have suggested the progression of hepatic cirrhosis. The final hospital admission in August 2016 was due to complaint of a stomachache for 3 days.\nAbdominal CT showed a wave-like margin of the liver and many areas of multifocal hypoattenuation in the liver, which indicated the occurrence of hepatic cirrhosis on the basis of diffuse hepatic steatosis (). Simultaneously, the patient presented with pancreatic atrophy and splenomegaly (). In addition, both chest CT scan and bronchoscopy showed the characteristics of bilateral bronchiectasis, marked peribronchial thickening, and extensive sticky and purulent secretion, similar to that observed in 2014 (). The abnormal prothrombin time (PT) and activated partial thromboplastin time were 20.2 s (normal control: 13 s) and 52.6 s (normal control: 36 s), respectively. The international normalized ratio (INR) was 1.85, which confirmed the diagnosis of liver failure. |
A nearly three year old male Caucasian (weight 14 kg) was admitted to the pediatric department of our hospital. The parents reported symptoms of an upper respiratory tract infection that had lasted for about ten days. During the last five days the boy developed a productive cough with fever. On the day of admission the parents recognized a diffuse swelling and redness of the forehead and the right orbital region.\nAn ongoing acute sinusitis was suspected and the child was admitted to the hospital. The preliminary physical examination revealed no further symptoms. A detailed inspection of nose and ears was not possible due to massive agitation and defensive behavior of the child. Laboratory results showed signs of a bacterial infection. Consequently intravenous antibiotics were administered.\nIn order to plan a possible surgical intervention an urgent MRI scan of the head was indicated by our otolaryngology department. Due to the uncooperativeness of the child and because of the intended immediate operative procedure it was decided to perform the MRI scan under general anesthesia.\nPrior to induction standard anesthesia monitoring including peripheral oxygen saturation, EKG and non-invasive blood pressure was established. Anesthesia was induced with propofol (5 mg/kg) and remifentanil (0.4 μg/kg/min). To facilitate orotracheal intubation mivacurium (0.2 mg/kg) was administered. After successful intubation anesthesia was maintained with sevoflurane (1.8 vol% end expiratory concentration) and continuous infusion of remifentanil (0.1 μg/kg/min).\nThe child was positioned in the 3 Tesla MRI scanner and a preliminary scout was run. The first image revealed an extinction of the face indicating a ferromagnetic object in the scanned region (Figure ). A quick inspection revealed no obvious foreign object in the facial region.\nTherefore a ferromagnetic foreign body in either mouth or nose was suspected and the child was removed from the MRI scanner. The detailed inspection of the nasal cavity revealed a small foreign body that could easily be removed by colleagues from our ear nose throat (ENT) department. After complete removal the object was identified as a small button battery (Figure ). A detailed examination revealed generalized swelling and a small mucosa lesion consistent with a burn or corrosion from acid. After a thorough lavage of the entire region the actual MRI scan was performed. While the initial scout was unremarkable detailed images still indicated ferromagnetic presence in the right nasal cavity (Figure ).\nTo exclude any further foreign body a computed tomogram of the head was performed. The imaged proved that no further object was located in the nasal cavity. The structures seen on the MRI scans were considered as corresponding to ionic material in the mucosa.\nAfter a second short inspection by the ENT colleagues anesthesia was discontinued and the child was extubated uneventfully.\nThe further post interventional course was unremarkable and the child was discharged from the hospital three days after the procedure. Oral antibiotics were continued for seven days.\nOn a follow-up visit four weeks later the child was doing remarkably well without any permanent issues. |
A seventy-six-year-old lady with a history of hypertension and previous sclera buckle for left eye retinal detachment presented to Singapore National Eye Centre with acute loss of vision of the right eye. There was no prior history of trauma or pain. On examination, visual acuity of the right eye was hand motions in the temporal region and perception of light in all four quadrants with a right grade 2 relative afferent pupillary defect. Intraocular pressure measured by Goldmann Applanation Tonometry was sixteen bilaterally. Anterior segment examination was unremarkable. Fundal examination of the right eye showed a pale optic disc and pale edematous macula with an evolving cherry red spot (). There was a very weak retinal venous flow which could be occluded on gentle pressure, indicating that the occlusion was likely incomplete. There were no plaques noted. Fundal examination of her left eye showed an epiretinal membrane and a flat retina with buckle indentation (). She underwent immediate ocular massage, anterior chamber paracentesis and intravenous acetazolamide 500 mg was administered. Optical coherence imaging using Heidelberg Spectralis was performed immediately after the above treatments and images showed opacification of inner retina with mild thickening and diffuse edema in the outer retina (Figures and ). Visual acuity subsequently improved slightly, and she was able to count fingers closely. She was admitted for complete work up to determine the cause of the event, and grave prognosis was conveyed to the patient. Carotid ultrasound showed an immobile 3 mm calcified plaque in the proximal segment of the right internal carotid artery, while 2D echocardiogram did not reveal any vegetations and fasting lipids and glucose were normal. Neurology consult was sought, and she was advised to start on aspirin 100 mg once a day, while no surgical intervention is required for the calcified plaque. Her vision remained stable and did not improve beyond counting fingers during her course of stay. The patient is currently considering left epiretinal membrane surgery for maximization of visual function. |
A 29-year-old multigravida patient who was 32 weeks pregnant was referred to our center because of a fetal ectopic intrathoracic kidney. Her past medical history and family history were unremarkable. She had not taken any medications during her pregnancy. Initial ultrasonography demonstrated a singleton female fetus with appropriate growth for its gestational age, but the right kidney was absent in the ipsilateral renal fossa, whilst the right lower thorax contained a mass with mixed echogenicity, suggesting an ectopic kidney ().\nThe left normal kidney and right ectopic one appeared to be of a similar size. Fetal echocardiography showed a mesocardia, a rotation of the fetal heart with the apex of the heart located in the mid-thorax with a structurally normal heart, and no signs of cardiac compression (). Left lung area to head circumference ratio was 2.54. There were no other abnormalities observed. Follow-up ultrasonography before delivery demonstrated a similar appearance of the intrathoracic kidney without any herniation of the abdominal contents. A female infant was delivered vaginally at 39.0 weeks of gestation with a birth weight of 3,230 g and Apgar scores at 1 and 5 minutes of 6 and 9, respectively.\nChest computed tomography at the 2nd day of birth showed a posterolateral herniated right kidney in the neonatal chest with no evidence of herniation of the abdominal viscera. The right renal artery was stretched to feed the intrathoracic kidney ().\nAt 7 days after birth, repair of the CDH was performed. There was 5 cm defect in the right diaphragm and only the ectopic kidney was herniated through this defect. The ectopic kidney was carefully reduced to the retroperitoneal cavity and the defect was repaired using a commercially available patch. The infant was discharged on day 26 after birth and a postoperative Tc-99m dimercaptosuccinic acid renal scan demonstrated symmetric renal function of 57% (left kidney) and 43% (right kidney). At the 15 month follow-up, the infant was doing well. |
A 78-year-old male patient visited our clinic for visual disturbance of the left eye which had developed one day previously. The patient had no ophthalmologic history, and he had been taking hypoglycemic agents for the control of diabetes mellitus for 3 years and was being medicated for chronic renal failure by the Department of Internal Medicine at our hospital. One day prior to presentation to our department, he developed systemic edema, and oral administration of 160 mg furosemide (Lasix; Handok Pharmaceuticals Co., Seoul, Korea) was administered by the Department of Renal Internal Medicine for the treatment of systemic edema. His blood pressure was 161 / 90. The basic data describing his systemic condition including body weight and height was not taken during his visit to the Internal Medicine Department. He did not have any history of furosemide treatment due to systemic edema. Blood tests at that time revealed an increase of blood urea nitrogen (BUN, 47 mg/dL) and creatinine (1.8 mg/dL), and urinary albumin was detected (500 mg/dL). On presentation to our department, the best corrected visual acuity of the right eye was 0.5, and that of the left eye was 0.125. The intraocular pressure of both eyes was within normal range. Upon fundus examination of the left eye, retinal edema was observed in the posterior pole, including the macula.\nOn fluorescein angiography, macular edema caused by vessel leakage was detected in the left eye (). Macular edema was confirmed by optical coherence tomography (). Two days after the oral administration of furosemide, the visual acuity of both eyes had improved to 0.6 and 0.3, respectively. After 7 days, the visual acuity of both eyes had improved to 0.9 and 0.6, respectively. Fundus examination and optical coherence tomography revealed complete disappearance of the sub-retinal fluid in the left eye (). Before the oral administration of furosemide, the right eye showed a mild abnormality on fluorescein angiography (). Because visual acuity of the right eye improved after administration of furosemide, we speculated that sub-retinal fluid was also present in the right eye. |
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