A 48-year-old Chinese female was admitted to Department of Oncological Surgery (Zhejiang Cancer Hospital, Hangzhou) with left upper abdominal pain, accompanying with belching and weight loss of 2.5 kg over the previous 3 months. Prior to this presentation, she had been healthy, without any previous medical illness. On physical examination, the left upper quadrant was slight tender but no abdominal mass was detected by abdominal palpation. Laboratory data were all normal. Computed tomography (CT) demonstrated a low-density mass with a CT value of 1 Hounsfield Unit (HU), measuring about 3.2 × 2.2 cm in the right lobe of liver (), and a solid low-density mass with a CT value of 8HU, measur-ing about 5.2 cm × 4.6 cm in the upper pole of the spleen ().
Based on the physical examination and CT, the preoperative diagnosis of the hepatic cyst was definite but the preoperative diagnosis of the splenic tumor was difficult. Then a laparo-scopic splenectomy was performed under a presumed diagnosis of a malignancy of the spleen. Intraoperatively, a solid mass ap-proximately 5 cm in diameter was found in the upper pole of the spleen. Intra-abdominal lymph nodes and the adjacent organs looked normal. There were not any signs of metasta-ses or any visible neoplastic tissue remnant at the operation site. Laparoscopic fenestration of the hepatic cyst was also performed. The re-sected spleen measured 11 × 10 × 8 cm, weighed 740 g. Histopathology showed that tumor cells were pleomorphic and consisted of fibroblasts, histiocytes, and myofibroblast-like cells arranged in a storiform pattern (). Immunohistochemistry showed these tu-mor cells were positive for vimentin (Vim), α-smooth muscle actin (α-ACT) and CD68 (). Based on these results of histopathology and immunohistochemistry, the final diagnosis of MFH of the spleen was conformed. The postoperative course of this patient was uneventful. The patient was discharged on the 7th day after the initial operation and was not given any additional treatment. She was followed up for 13 months without recurrence or metastases after the initial surgery at the time of reporting.


A 4-month-old female child presented with haemangioma since birth, involving the left lower leg near the ankle, with ulceration for the past 2 months. The infant was admitted. After complete evaluation in conjunction with the cardiologist that involved pretreatment cardiac workup and an in-hospital titration of propranolol, the infant was given oral propranolol following the standard treatment protocol.[] A course of an oral antibiotic was given for a period of 1 week before starting treatment with propranolol. The infant was carefully monitored for systemic side effects. Propranolol was given as an oral liquid (1 mg/mL), at an initial dose of 1 mg/kg daily divided into three equal doses and increased to 2 mg/kg daily after 1 week. The infant was monitored for a 4-h period in hospital after the first dose and 1 week later when the dose was increased. During the 4-h period, the heart rate and blood pressure were monitored every 30 min. For the first 2 weeks, the heart rate and blood pressure were checked twice weekly and thereafter once weekly on a shared-care basis with the paediatric team.
The child was admitted for every dose of propranolol. The response was slow till the 12th week. However, during the subsequent visit, there was a significant reduction in the size with a change in the colour and texture of the lesion. The lesion healed with an atrophic scar at the site of ulceration [Figures –]. Medication was continued for a further period of 2 months and then stopped. The child is being followed up and there has been no recurrence in the past 6 months.


A 45-year-old man presented to us with dry cough and mild chest discomfort since 1 year. His chest examination was unremarkable. Plain X-ray of the chest showed an abnormal spherical shadow in the upper portion of the right lung. A CT scan of the thorax revealed a 9 × 6 cm2 sized well-circumscribed lesion with fat attenuation in the upper lobe of the right lung suggestive of pleural lipoma []. All other haematological and biochemical investigations were within normal limits. The lesion was removed by VATS []. Histopathological examination of the retrieved specimen revealed that the lesion was composed of well-differentiated mature adipocytes, which is suggestive of a lipoma.


A 54-year-old man presented with a 3-month history of cough and shortness of breath during exercise. On physical examination, he had an audible expiratory stridor. The chest X-ray was normal. Computed tomography (CT) of the neck and chest revealed a 1.5 cm polypoid tumor arising from the posterior wall of the upper thoracic trachea (). A flexible bronchoscopy showed a double cone shaped, polypoid tumor arising from the posterior wall of the trachea, 5 cm distal to the vocal cords, and 7 cm proximal to the carina (). Based on the biopsy specimens, the mass was diagnosed as a glomus tumor. To check for an extension of the tumor into the surrounding tissue, an endoscopic ultrasonic exam was performed, and it showed that the tumor was confined to the trachea. Since the tumor was located in the upper trachea, we decided to approach the tumor through a cervical collar incision. After exposure of the trachea, the location of the tumor was identified between the 5th and 6th tracheal rings using flexible bronchoscopy. We divided the trachea one cartilage ring below the tumor and a sterile laryngectomy tube was inserted into the distal trachea. A proximal tracheal division was made one cartilage ring above the tumor. A frozen section analysis confirmed negative margins. Anastomotic sutures were placed using interrupted 4-0 Vicryl sutures (Ethicon, Somerville, NJ).
The surgical specimen was 3 cm in length with a luminal diameter of 2.3 cm. There was a 1.3×1.2 cm blue-red protruding nodule on the mucosal surface of the trachea (). The tumor did not show an infiltrative growth into the tracheal cartilage. At the microscopic level, the tumor showed glomus cells, vascular structures, and smooth muscle tissue (). The glomus cells had undergone a transition to elongated, mature smooth muscle cells in some foci (); this feature differentiated this tumor from ordinary glomus tumors. The tumor cells showed positive reactions for smooth muscle actin and CD34, but negative reactions for S-100 and cytokeratin on immunohistochemical staining. These results led to the diagnosis of glomangiomyoma.
Bronchoscopy and a chest CT were performed 3 months after the operation.
They showed well anastomosed tracheal mucosa and a good tracheal air column (). The patient was asymptomatic for 24 months after surgery.


A 61-year-old diabetic lady with history of Rheumatoid Arthritis and Sjögren's syndrome was referred to urology clinic for storage symptoms of urgency and dysurea, but no hematuria nor history of weight loss were noticed. There was no family history of malignancy. Her physical examination was unremarkable. Her blood work and urine test were within normal limits. Ultrasound of abdomen and pelvis revealed no hydronephrosis or masses of the upper tract. Although partially filled, the bladder revealed a mass []. Computed Tomography (CT) scan confirmed the presence of the bladder tumor without any evidence of distant metastasis [].
The patient underwent cystoscopy, which revealed a protruding mass from the anterior wall of the bladder, but with normal covering urothelium. It was better visualized with help of suprapubic compression. The resecting loop was used to incise the urothelium and complete enucleation of the mass was successful bluntly by using the tip of the loop, using low pressure irrigation and continuous emptying of the bladder to avoid any significant extravasations. The patient had an uneventful post-operative period and a Foley's catheter was left for 3 days. Grossly the mass measured 1.7 × 1.1 × 0.8 cm in greatest dimensions with smooth surface and hard consistency. Microscopic description revealed typical features of Leiomyoma, positive for Desmin and negative HMB45 and CD117 [Figures and ].
During follow-up, the patient was free of recurrence, documented on ultrasound and CT scan done at 3 months and 12 months, respectively [Figures and ]. She continued to have urgency, which was controlled by Oxybutanin 5 mg twice a day.


A 65-year-old obese female with remote past history of cholecystectomy (for cholelithiasis), presented with complaint of nausea, mild jaundice and persistent dull aching pain in whole upper abdomen of few weeks duration. There was no significant past or present medical history. Per abdomen examination revealed a well defined, smooth mass in left hypochondrium. On laboratory analysis, liver function test revealed increase in total bilirubin (3.24 mg/dl), conjugated bilirubin (2.17 mg/dl), and alkaline phosphatase (224 U/L). Renal function test was unremarkable. Routine blood examination revealed mildly decreased hemoglobin (8 g/dL). Endocrine evaluation of the adrenal mass revealed normal 24 hours urinary metanephrine (0.9 mg/24 hours) and plasma cortisol (<5 μg/dl). Furthermore, the levels of two tumor markers, carcinoembryonic antigen (1.25 ng/ml) and alpha-fetoprotein (1.00 ng/ml) were also within normal range. The patient was sent to the radiology department for evaluation of obstructive jaundice and left hypochondrial lump.
Ultrasound examination revealed a large well encapsulated, heterogeneous left suprarenal mass lesion []. In addition, non-visualization of gall bladder (status post-cholecystectomy for cholelithiasis), with dilatation of common bile duct (CBD 20.8 mm diameter), increased width and prominence of central intrahepatic biliary radicals (IBHR) in absence of any intraluminal filling defect were also noted. The plain and contrast computed tomography image [] of the abdomen revealed a well circumscribed, heterogeneous enhancing mass of 91.4 (AP) × 67.3 (TR) × 101.3 (SI) mm in the left supra-renal region causing inferior displacement of the left kidney. The lesion was predominantly of fat density (–30 to –90 HU) with few enhancing soft tissue components and irregular septations. Few tiny flecks of calcifications were also visible in the lateral wall of the mass. The coronal and sagittal CT images [] confirmed the adrenal origin of the mass. The function of both kidneys appeared normal. Abdominal magnetic resonance imaging revealed a heterogeneous, high intensity mass on T1 and T2-weighted images [], with hypointense septae. Linear hyperintense areas posteriorly on T2WI represented intratumoral bleed. Fat-saturated T2- weighted gradient echo sequences [] showed loss of signal intensity in the fatty part of the mass, while the hematopoietic tissue and hemorrhage demonstrated persistent areas of increased signal intensities. Magnetic resonance cholangiopancreatography (MRCP) confirmed the ultrasonographic finding of dilated CBD and central IHBR; T2-haste coronal sequence demonstrated partial benign stricture at ampullary end of CBD as a cause of biliary dilation [].
The imaging findings were highly suggestive of left adrenal myelolipoma, with CBD and central IHBR dilatation due to benign stricture at ampullary end of CBD. Right adrenal gland was normal. The patient underwent surgical resection of large adrenal mass and endoscopic stenting of the biliary stricture. Histopathology of resected left adrenal mass showed mature fat cells admixed with hematopoietic tissues without cellular atypia. The diagnosis of adrenal myelolipoma was confirmed. The postoperative recovery was uneventful.


A 33-year-old obese female presented with vague right abdominal pain for last three months. She had no other significant medical history. On laboratory analysis, hemogram, biochemical and endocrine function tests were within normal limits. Liver and renal function tests were also normal. Levels of tumor markers such as carcinoembryonic-antigen and alpha-fetoprotein were unremarkable. Physical examination revealed a right hypochondrial lump.
Ultrasound examination of abdomen showed a huge, well encapsulated, solid echogenic mass lesion between the liver and right kidney []. Normally distended gallbladder with multiple intraluminal calculi was also noted. CBD and IHBR were normal. Plain and contrast CT abdomen [] revealed a well circumscribed 83.9 (AP) × 72.2 (TR) × 104.4 (SI) mm sized right suprarenal mass predominantly of fat density (–50 to –90 HU) with peripheral small enhancing soft tissue component on the postero-medial aspect of the mass. A punctate focal calcified area was also seen adjacent to the soft tissue component. Furthermore, multiple small focal calcific lesions (calcified granulomas) randomly distributed within the splenic parenchyma were seen. Suprarenal nature of the mass was confirmed by coronal and sagittal CT images [], which also showed postero-inferior displacement of right kidney. Left adrenal gland and both kidneys appeared normal. MR abdomen revealed predominantly hyperintense mass on T1-and T2-weighted image []. T2W-fat-suppression image [] showed loss of signal from large fatty portion of the mass, but persistence of increased signal intensity in areas occupied by hematopoietic tissue and intratumoral hemorrhage. Double echo chemical shift gradient echo imaging [] demonstrated few focal areas of signal loss in out-of-phase image as compared to in-phase image, consistent with the presence of microscopic (intracellular) fat, while the major portion of the mass demonstrated high signal intensity consistent with the presence of macroscopic (extracellular) fat.
The imaging finding and hormonal evaluation highly suggested the diagnosis of a nonfunctioning right adrenal myelolipoma. Right adrenalectomy and cholecystectomy was performed in the same setting to prevent future risk of spontaneous hemorrhage and gall stone associated complications. Histopathology confirmed the diagnosis. Post-operative period was uneventful.


A 22-year-old man was admitted to our hospital on March 28, 2011 complaining of seven days of retrosternal discomfort and a single episode of vomiting of approximately 50 mL of fresh red blood. The patient had swallowed a chicken bone prior to the onset of the discomfort. His vital signs were stable during physical examination, and there was no fever on arrival at the hospital. Laboratory findings revealed a white blood cell (WBC) count of 19.9×109/L and hemoglobin (Hb) levels of 84 g/L. He vomited approximately 300 ml of bright red blood and produced a large amount of tarry stool on March 29, 2011. His Hb levels subsequently decreased to 73 g/L. CT angiography (CTA) demonstrated a mediastinal esophageal fistula with inflammatory exudate surrounding the descending aorta. An aortic pseudoaneurysm at the anterior wall of the descending aorta was also observed at the lower edge of thoracic vertebrate 5 (). A diagnosis of AEF was highly suspected. Angiography was performed on March 31, 2011 and showed a 0.7 cm×0.4 cm pseudoaneurysm of the descending aorta approximately 4.5 cm below the origin of the left subclavian artery, which was consistent with the results of the CTA. This finding confirmed the diagnosis of AEF. After an immediate multidisciplinary discussion, a plan for definitive treatment was established. An endovascular stent-graft (catalog number: TF2424C200X, Medtronic, Inc., USA) was placed inside the descending aorta to isolate the pseudoaneurysm. Postoperative angiography showed satisfactory exclusion results, and the patient was transferred to the operating room for thoracoscopic exploration. Swelling in the esophageal bed and inflammatory exudates were observed under the thoracoscope. Immediate mediastinal debridement and irrigation was performed using the thoracoscope. Two F16 silicone tubes were placed in the pleural cavity and the mediastinum to facilitate postoperative drainage. The patient underwent an esophageal endoscopy, and an esophageal fistula approximately 1.0 cm in diameter was found 25 cm away from the incisors (). A single-use retrievable esophageal stent-graft (Micro-Tech Co., Ltd. Nanjing China, length 8.0 cm, diameter 2.0 cm) was implanted to isolate the esophageal perforation followed by the placement of a three-lumen gastrojejunal tube for additional enteral nutrition. After spending three days in the intensive care unit (ICU), the patient was transferred back to our department. He had no melena and no fever or vomiting, and the mediastinal and pleural drainage gradually became clear. On the 7th day after the operation, esophageal imaging with meglumine diatrizoate showed no leakage, and the three-lumen gastrojejunal tube was removed. The patient was provided with a liquid diet. Both of the drainage tubes in the pleural cavity and the mediastinum were removed 25 days after the operation. Repeat CTA confirmed successful tamponade of the fistula 40 days after the operation (). Antibiotic treatment was administered during hospitalization. Laboratory findings showed that the patient's WBC count was 23.0×109/mL with 79.1% neutrophils (NEs) on the second day after the operation. The WBC count and NE returned to normal after 14 consecutive days of administration of the antibiotic meropenem based on cultures of the mediastinal exudates, which were positive for Pseudomonas aeruginosa seven days after the operation. The patient was switched to oral antibiotics and discharged on May 18, 2011. He was re-admitted to remove the esophageal stent 80 days after the operation. Esophageal endoscopy revealed a complete healing of the original fistula after removal of the esophageal stent (). No fever was observed during the treatment follow-up. After continuous administration of oral antibiotics for three months, the patient stopped the medication when blood laboratory findings and chest X-ray results were normal. The patient lived a normal life during six months of follow-up after the operation.


A 31-year-old female presented with asymptomatic multiple skin colored or erythematous greasy papules on the face, which developed in her late 20s. She was otherwise well, without any relevant previous history of skin or medical disease. She said that her relatives on the maternal side, including her grandfather, mother, uncle, two aunts, and two brothers, had the same skin lesions. She had never been treated for her skin problem before.
Physical examination revealed multiple skin colored or erythematous papules on the forehead, eyelids, and areas along paranasal and nasolabial folds. Clinically, these papules looked like trichoepithelioma or syringoma, or comedones of acne vulgaris. Her facial skin was greasy (). Otherwise, the scalp, trunk, palms, soles, nails, oral mucosa and teeth were normal.
Skin histopathology of facial papules showed dilated follicular infundibulum, containing keratotic materials with parakeratotic cells. At the lateral aspect and base of the follicular infundibulum, suprabasal acantholysis led to formation of clefts, lacunae, and villi. Dyskeratotic cells, including corp ronds and grains, were observed, as well as brisk perifollicular inflammatory cellular infiltration ().
According to her clinical and histopathological features, we finally diagnosed comedonal Darier's disease. We treated her with oral minocycline and topical tacrolimus ointment; however, her condition showed little improvement and she refused further treatment.


A 77-year-old male, a farmer and a chronic tobacco chewer, presented with a sacral mass since 3 months. To begin with, he developed a small nodule in the sacral region 8 months back and was operated for the same. After the operation, there was an ulcerative lesion that increased in size to develop into the present sacral mass. There was no history of diabetes mellitus, hypertension or tuberculosis. Clinical examination revealed a mass in the sacral region measuring 7 cm × 5 cm × 5 cm with ulceration of the skin measuring 3 cm × 2 cm. A systemic examination of the cardiovascular, respiratory and central nervous systems was unremarkable. His hemoglobin, complete blood count, liver and kidney function tests were normal and he was nonreactive for human immunodeficiency virus and hepatitis B surface antigen. The wedge biopsy of the mass was reported as spindle cell carcinoma.
Grossly, a specimen of an excised mass covered with skin measuring 10 cm × 8 cm × 6.5 cm was received. The skin showed brownish discoloration and ulceration of 3 cm × 2 cm []. The cut surface revealed a nodular grayish-white, soft fleshy tumor measuring 7 cm × 5 cm × 5 cm []. Microscopically, the tumor comprised of fascicles of spindle cells with focal areas of hemorrhage and necrosis. The spindle cells had eosinophilic cytoplasm with hyperchromatic “cigar-shaped” nuclei with inconspicuous to prominent nucleoli []. The tumor showed marked nuclear pleomorphism with tumor giant cells. The mitotic activity was 20/10 high-power fields, with the presence of atypical mitosis []. The inflammatory component was present throughout the tumor. The surgical cut margins except the base were free of tumor.
The tumor cells were immunoreactive for smooth muscle actin (SMA) while being negative for S-100, desmin, CK-7 and CD34 .


A 10-year-old girl presented with high-grade intermittent fever with cough for 20 days and a generalized rash for 15 days following ingestion of phenobarbital for 6 weeks. There was h/o seizures with fever from the age of 18 months, but the severity and frequency of seizures started increasing, occurring even without fever for which she was put on oral phenobarbital.
On admission, we found that the girl was toxic, febrile, icteric, with tachypnea, tachycardia, conjunctival congestion, stomatitis, and cervical, axillary and inguinal lymph node enlargement. There was a diffuse erythematous, maculopapular rash without vesiculation or blistering []. There were bilateral diffuse crepitations over the chest and tender hepatomegaly.
Initial investigations showed leukocytosis, eosinophilia (absolute eosinophil count: 1586/dl), high bilirubin, elevated liver enzymes, microscopic hematuria and minimal pyuria. Imaging studies revealed hepatomegaly with features of hepatitis, mild pericardial effusion on echocardiography, and features of diffuse pneumonitis. Initial blood culture and urine culture were negative.
The patient was put on empirical antibiotics and other supportive measures. Antibiotics were changed and steroid (i.v. dexamethasone) was added on persistence of fever and dyspnea, following which there was a short period of improvement. But after a few days, the girl again complained of sudden onset exacerbation of respiratory distress and we found that she had developed subcutaneous emphysema and pneumomediastinum []. The patient had intermittent high spiking fever. A repeat blood culture showed growth of Acinetobacter baumanii and we switched over to sensitive antibiotics. But the general condition deteriorated and the patient developed ARDS for which she was ventilated. She improved dramatically and was extubated after 6 days. The girl again developed fever and dyspnea 2 days postextubation. There were findings of ARDS and pneumothorax on chest X-ray. She was ventilated again but succumbed after 5 days.


An eight-year-old boy was referred to our hospital due to generalized edema for two weeks and sudden onset of scrotal swelling that occurred two days prior to admission. Since one year before the admission, he had repetitive, acute, self-limited episodes of fever at varying intervals with an elevated level of acute phase reactants. Despite thorough evaluation, the reason for the fever was unable to be determined. After an initial examination, he had been followed up under the impression of juvenile rheumatoid arthritis even though he did not demonstrate joint symptoms. Family history revealed that his father died due to myocardial infarction at the age of 40 and there was no history of renal disease, periodic fever or auto-inflammatory disease.
Upon physical examination, his fever was 38.4℃, blood pressure was 108/75 mm Hg, and body weight increased from 23 kg to 26 kg in last two weeks prior to admission. Pretibial pitting edema and swollen abdomen were also observed without rash. Laboratory findings on admission showed inflammatory conditions of very high C-reactive protein (140 mg/L) and high erythrocyte sedimentation rate (120 mm/hr). Anemia (8.4 g/dL), hypoalbuminemia (1.0 g/dL), and markedly elevated fibrinogen (1029 mg/dL) were observed as well. Massive nephrotic-range proteinuria (urine protein/creatinine ratio was 14.49, total microprotein 12297 mg/day, microalbumin 10249 mg/day) was observed with 24-hour collected urine. Different markers for autoimmune diseases such as anti-nuclear antibody, and anti-cardiolipin antibody were negative and complement levels as well as immunoglobulin concentrations were all within normal range.
After admission, he complained of abdominal pain and abdominal ultrasonography revealed slightly increased renal parenchymal echogenicity with splenomegaly (11 cm) and no hydrocele in both scrotal sacs. Abdominal computed tomography showed enlargement of bilateral kidneys; a well-enhancing mass lesion in the right common iliac chain; small bowel mesentery, suspected as lymphadenopathy; a small amount of ascites; and pleural effusion. Echocardiography showed minimal pericardial effusion with decreased deceleration time of the mitral valve (80-100 ms), which suggested a poor prognosis.
Renal biopsy was performed for evaluation of the massive proteinuria, and the pathologic diagnosis was confirmed as amyloidosis type AA (). Tests for monoclonal gammopathy to rule out AL amyloidosis, including serum/urine protein electrophoresis, immunofixation electrophoresis, and serum κ-, λ-free light chain assays, were all negative. Cochicine therapy was started with a dose of 0.025 mg/kg/day under the impression of FMF and his clinical symptoms gradually improved.
In order to make the clinical diagnosis, the clinical entities of periodic fever and systemic inflammatory reactions combined with amyloidosis were considered, which were indicative of FMF (OMIM no. 249100), familial periodic fever (FPF, TRAPS; OMIM no. 142680), hyperimmunoglobulinemia D with periodic fever syndrome (HIDS; OMIM no. 260920), and Muckle-Wells syndrome (MWS; OMIM no. 191900). The patient was able to be diagnosed as FMF since he showed typical attacks fulfilling three major criteria (pleuritis, fever and incomplete abdominal attack) of the Tel-Hashomer criteria. To verify the underlying cause of AA amyloidosis, DNA analysis of the MEFV gene was initially performed and two point mutations (p.Pro369Ser, p.Arg408Gln) were identified (). For differential diagnosis, other genes, including MVK for HIDS, NLRP3 for MWS and TNFRSF1A for FPF, that cause similar phenotypes were also analysed. Only several SNPs were identified in the analysis of MVK, NLRP3 and TNFRSF1A genes ().


A 35-year-old female presented to our hospital with squeezing chest pain for a duration of 15 minutes. She was a current smoker (20 pack years) without any other risk factors of cardiovascular disease. On physical examination, her blood pressure was 145/80 mm Hg, and pulse rate was 96 beats per minute. Chest radiography showed mild cardiomegaly with mild pulmonary congestion. The initial electrocardiogram (ECG) revealed 2-mm ST segment depression in leads V3-6 and up to a 1-mm ST segment depression in leads I, aVL, II, aVF with a 2-mm ST segment elevation in the aVR lead (). An initial cardiac enzyme profile revealed elevation of creatine phosphokinase (CK), 195 U/L (normal range, 26-174); CK-MB, 49 µg/L (normal range, 0-3); and elevated troponin-I of 3.87 ng/mL (normal range, 0-0.05). Emergency coronary angiography showed a 90% narrowing of the LMCA () and a normal right coronary artery without significant narrowing (). During coronary angiography, the patient complained of chest pain with a sudden decrease in systolic blood pressure to 90 mm Hg. However, after administering 100 µg of intracoronary nitrate, the chest pain was relieved and a re-angiography showed a completely resolved LMCA spasm without residual luminal stenosis (). IVUS showed only a minimal concentric plaque in the LMCA without significant narrowing (). Therefore, we stopped the procedure without intervention. After the procedure, an ST depression in leads I, aVL, II, aVF, and V3-6 and an ST elevation in aVR were completely resolved on the ECG (), and the cardiac enzymes had decreased to the normal range (CK, 41; CK-MB, 22 µg/L; and troponin-I, 1.31 ng/mL). The patient was discharged on the eighth hospital day receiving 40 mg of oral nitrate, 180 mg of diltiazem, 100 mg of aspirin, and 10 mg of rosuvastatin per day. A follow-up coronary angiography taken 9 months later showed no spasms or stenosis. The patient has exhibited no signs of angina for 2 years and has remained on medication in outpatient clinics.


A 52-year-old man who had been diagnosed with aplastic anemia and stable angina visited the emergency room of the authors' hospital with a complaint of chest pain. The chest pain had developed 1 hour before the patient's arrival at the hospital. The initial electrocardiogram showed a hyperacute tall T wave in the precordial leads (). In the initial laboratory findings, the cardiac markers were in the normal range, but thrombocytopenia was noted (initial white blood cell count: 3350, hemoglobin: 12.5 g/dL, platelet: 45000/µL). The patient's vital signs were stable. His past history revealed that he had been admitted to a hospital a year ago for acute coronary syndrome and percutaneous coronary intervention (PCI) of the left anterior descending artery (LAD), for which a Cypher Select stent (3.5×28 mm, Johnson & Johnson-Cordis, Miami, FL, USA) procedure was performed (). He had been taking 100 mg of aspirin, 75 mg of clopidogrel and 100 mg of oxymetholone (anabolic steroids) for aplastic anemia daily since the last one year. However, he had stopped taking aspirin and clopidogrel for 3 days before presenting himself at the hospital since he was scheduled to be treated for a decayed tooth. He also visited the hematology clinic routinely. His laboratory findings showed that he had severe thrombocytopenia (platelet count: 12000/µL). His hematologist recommended platelet transfusion for anticipated bleeding. Therefore, transfusion of 6 pints of platelet concentrates was performed on the same day of the patient's visit to the hospital. Four hours later, he developed chest pain.
The emergent coronary angiography demonstrated total occlusion
of the mid-LAD. Balloon angioplasty was done with a 3.0×15 mm non-complaint coronary balloon. Thrombolysis in myocardial infarction 3 distal flow was obtained. Thus, no additional stent was needed ().
A dose of 7,000 units of unfractionated heparin was injected during the CAG with another loading dose of 300 mg of clopidogrel. After the PCI, the chest pain subsided. On the second day, blood started oozing from the puncture site, hence a manual compression dressing was applied for 1 hour, after which there was no more complications. The follow-up platelet count was 12000/µL. On the 12th day, the patient's dental caries was treated with tooth extraction without bleeding complications, and the patient was discharged. He was prescribed triple antiplatelet therapy with aspirin, clopidogrel, and cilostazol. He was also asked to continue oxymetholone (anabolic steroids) therapy for aplastic anemia.


A 44-year-old man developed anaphylaxis after an oral intake of fresh ginseng. He complained of rhinorrhea and nasal stiffness, followed by respiratory difficulty and abdominal pain 10 minutes after ingestion. He had suffered from seasonal allergic rhinitis during the spring season for several years. Upon presentation, a physical examination, chest radiography, and routine laboratory tests were unremarkable, with normal serum IgE levels. To confirm the causal relationship, ginseng extracts were prepared and used for SPT, as well as for enzyme-linked immunosorbent assay (ELISA) and BAT. The SPT results (A/H ratio) showed positive responses to ginseng extract (2+ with 1:500 w/v) and fresh ginseng (3+) as well as to alder pollens (4+) and birch pollens (4+). The methacholine bronchial challenge test produced a positive result at 5.83 mg/mL. The open oral challenge was performed using 50 g of fresh ginseng, and the patient showed immediate onset of facial flushing, cough, respiratory difficulty with wheeze, and abdominal pain. The patient's blood pressure was 140/90 mmHg, respiration rate was 22 breaths/minute, pulse rate was 66/minute, and body temperature was 36.6℃. His oxygen saturation decreased to 90% from a baseline value of 96%, with a significant decrease of FEV1 (54% from baseline). The patient was treated with nasal oxygen, IV steroids and salbutamol nebulization leading to a rapid recovery. Serum-specific antibodies were detected by ELISA, and BAT was performed using flow cytometry, according to previously described methods., Serum-specific IgE and IgG4 were not detected, but a higher level of serum-specific IgG1 was noted in the patient samples, as compared to the control samples. BAT showed a dose-dependent increase in the expression of CD203c and CD63 on the basophils of the patient in response to ginseng extracts, while no changes were observed in the controls ().


A 67-year-old male presented to the outpatient clinic with a chief complaint of ambulatory difficulty and a history of an ankle fracture and deformity. The patient's daughter reported that her father sustained an ankle fracture 6 weeks ago and was hospitalized. The history revealed that the patient was placed in a short-leg cast and surgery was considered but not performed since the ankle mortise was reduced at that time and the patient's ankle displayed multiple fracture blisters. The patient was discharged to a rehabilitation facility during which time the cast was removed secondary to non-compliance and skin compromise. The patient's daughter stated that her father was fully ambulatory at 2 weeks after the initial injury despite medical advice. Unfortunately, he was unaware of his ankle fracture since he suffered from dementia and Alzheimer's disease. His medical history was also significant for poorly controlled diabetes mellitus, peripheral neuropathy, peripheral vascular disease, prior alcohol abuse, seizures, aortic stenosis, meningioma, and hypertension. A physical examination revealed a well-nourished individual who was in no acute distress and was alert but not oriented to time, person, or place. His vital signs were stable. The left lower extremity was edematous with a non-infected necrotic wound over the medial malleolus. The ankle and rearfoot displayed severe valgus deformity with the medial malleolus causing pressure necrosis to the overlying soft tissue. He had weakly palpable pulses and loss of protective sensation when examined with a 5.07 Semmes-Weinstein monofilament. Radiographic evaluation revealed a displaced fibula fracture with significant ankle joint subluxation and widening of the medial clear space (). The patient was admitted to the hospital and stratified as moderate risk for surgical intervention after optimized by both internal medicine and cardiology.
Non-invasive vascular studies that consisted of ankle-brachial index, toe-brachial index, and pulse volume recordings demonstrated moderate arterial occlusive disease with a falsely elevated ankle-brachial index secondary to calcified vessels. The patient was then evaluated by vascular surgery and after angiography was performed, adequate collateral circulation was present and their recommendation was to proceed with appropriate surgical intervention. The patient was brought to the operating room after obtaining informed consent from his daughter, as he was deemed not competent to make his own medical decisions. The patient underwent operative intervention that consisted of ORIF of his fibula fracture with placement of multiple pro-syndesmotic screws and application of a multi-plane circular external fixator. Postoperatively, the patient was permitted to fully weight-bear with the external fixator and discharged to a rehabilitation facility. The patient developed a postoperative infection that was evident 3 weeks after surgery, as wound dehiscence and drainage were apparent. The patient was readmitted to the hospital for inpatient care. The patient was brought back to the operating theater during this admission for an incision and drainage of the ankle wound with collection of deep intra-operative soft tissue cultures and reclosure of the wound over a drain. The drain was removed at 72 h and then wet to dry dressings were applied daily until the wound was healed. Deep intra-operative cultures grew methicillin-resistant Staphylococcus aureus. The patient was initially placed on parenteral administration of Vancomycin but was switched to oral administration of Zyvox after the first week as the patient continued to remove his peripherally inserted central catheter line. His antibiotic regimen was continued 6 weeks for treatment of deep infection with retained hardware. The patient healed his wound at 6 weeks postoperatively and was full weight-bearing with the external fixator for 12 weeks total time.
The external fixator was removed and the patient was placed into a walking total contact cast for a total of 6 weeks with cast changes performed at 2 week intervals. The patient was able to resume full ambulatory status with prefabricated extra-depth diabetic shoes with soft inlays at 18 weeks postoperatively. The patient continued to maintain an ambulatory status with no further complications at his 8-month follow-up ().


A 57 year old male was admitted to the Department of Oncology complaining of dyspepsia for 2 months and sudden onset of hematemesis. He underwent endoscopy and computed tomography (CT) and was diagnosed with esophageal cancer and stomach cancer. He had a history of complete atrioventricular block and had a pacemaker implemented in 1992. He had diabetes mellitus and his social history was significant for drinking a bottle of alcohol daily and smoking one pack of cigarettes for 30 years. He had no significant family history.
He was chronic ill-looking, and alert. His blood pressure was 120/80 mm Hg, pulse 60/min, respiration 20/min, body temperature 36.5℃. His skin was dry and warm, with pale icterus and dry tongue. His lungs were clear to auscultation and his heart was regular without murmur or rubs. His bowel movement was normal without abdominal pain or tenderness. His liver and spleen were not palpable. The complete blood count showed white blood cell count 9,610/mm3, Hb 10.4 g/dL, Plt 284×103/mm3. His blood chemistry was normal, with tumor markers carcinoembryonic antigen (CEA) 2.22 ng/mL and squamous cell carcinoma antigen (SCC Ag) 2.2 µg/L. The chest X-ray and simple abdomen showed no abnormality. On CT, there was an ulcerofungating mass from the pylorus of the stomach to lower esophagus. The stomach lesion was abutting the tail of the pancreas and the diaphragm on CT (). The positron emission tomography-computed tomography (PET-CT) showed no distant metastasis other than suspicious cancerous lesion in stomach and esophagus. He then underwent upper gastrointestinal endoscopy and endoscopic ultrasound with biopsy. The endoscopic finding revealed an irregular shaped lesion with decreased vascular marking from 31 to 39 cm from upper incisor. The main cancer lesion in the esophagus was 35 cm from UI with serosa invasion and local lymph node metastasis. It was poorly-differentiated squamous cell carcinoma (). The stomach lesion was Bormann III shaped, moderately-differentiated squamous cell carcinoma, with serosa exposure and local lymph node invasion by 9 mm ().
According to above findings, the stomach lesion was resectable and he underwent total gastrectomy and esophagoduodenostomy. The operative findings showed a 6×6 cm sizedulcerofungating mass in the lesser curvature of the stomach that invaded soft tissue of the pancreas (). There was no distant metastasis to liver or rectum. Postoperative findings demonstrated a squamous cell carcinoma of the stomach, pT4N2M0 with serosa invasion, 4 out of 41 local lymph nodes metastasis with perineurovascular invasion. The pathologic finding showed that the proximal resection margin of the gastric lesion was negative. This suggests the squamous cell carcinoma of the stomach was not a direct extension from squamous cell carcinoma of esophagus. They were indeed two separate cancers with different pathology.
The patient was in good condition after the surgery, and underwent combination chemoradiotherapy (5-Fluorouracil/Cisplatin: 5FU 1 g/m2 per day on the first 4 days of weeks 1, 5, 8, and 11th weeks, Cisplatin 75 mg/m2 on first days of 1, 5, 8, 11th weeks, Radiotherapy 2.0 Gy for 5 days a week). He is now in complete remission and regularly followed up in outpatient clinic.


A 46-yr-old woman was referred to our hospital for generalized edema. Her edema had started one week previously after a one-week history of mild febrile sense, headache and cough. The laboratory test on admission showed anemia (hemoglobin 9.1 g/dL, hematocrit 26.8%), thrombocytopenia 98 × 103/µL) with acute renal failure (blood urea nitrogen 31 mg/dL, creatinine 1.5 mg/dL) and her urinalysis showed moderate hematuria and proteinuria (4.6 g/day). Her serologic tests were abnormal with high ANA titer of 1:160, low C3 of 35 mg/dL (normal 70-150 mg/dL) and C4 of 6 mg/dL (normal 10-35 mg/dL), and elevated anti-dsDNA autoantibody titer of 146 IU/mL (normal < 10 IU/mL). She had a kidney biopsy and was diagnosed with DPLN (ISN/RPS Class IV-G(A)).
In the absence of any evidence of infection, she received two courses of steroid pulse therapy (1 g of methylprednisolone) followed by oral high dose steroid (1 mg/kg of prednisolone). Before receiving the third course of steroid pulse therapy, her chest radiograph showed a 4 cm-sized round opacity in her right upper lung field, although she had no respiratory symptoms. Computed tomography (CT) scan of the chest disclosed a mass with central necrosis surrounded by a halo of ground-glass attenuation. These images were highly suggestive of invasive pulmonary aspergillosis. Therefore, she underwent a percutaneous needle biopsy for the lesion, but no microorganisms were revealed by histological examinations including Grocott's methenamine silver staining, acid-fast staining, periodic acid-Schiff (Ed- the acronyms GMS and PAS are unnecessary as they are not used anywhere else in the paper) staining and Gram staining of the lung tissue specimen.
By clinical impression, the patient commenced a course of amphotericin B due to the possibility of other fungal infections, such as mucormycosis. After one month, the lesion was cleared up in the chest CT, amphotericin B was stopped, and chest radiograph did not show any lung lesion suggestive of active infection such as fungal infection. Subsequently, she received nine courses of cyclophosphamide pulse therapy (monthly 6 times and quarterly 3 times with total dose of 5.75 g) over 15 months. Oral steroid was tapered down from daily 5 mg/kg to daily 5 mg. At this time, her urinalysis showed no hematuria and proteinuria was around 100 mg/day with normal kidney function (0.9 mg/dL), indicating remission of DPLN.
While waiting for the next (10th) cyclophosphamide pulse therapy, the patient suffered two episodes of massive epistaxis and magnetic resonance imaging (MRI) of the paranasal sinuses (PNS) showed both maxillary sinusitis and a 1 cm-sized, strongly enhanced lesion in the left sphenoid sinus with focal bony defect in the posterolateral aspect of left sphenoid sinus, suggesting aneurysm of the left cavernous ICA.
The patient was transferred to the interventional neuroradiology unit, where transfemoral cerebral angiography revealed one aneurysm in the cavernous segment of the left carotid artery (). A graft stent was deployed in the cavernous ICA for parent vessel reconstruction, and there was no filling of contrast media into the aneurysm at delayed angiography (). Endosinus sphenoidectomy was performed to remove the suspected infection source and to identify the pathogen. Histological examination of the sinus mucosa biopsy specimen revealed fungal hyphae that were morphologically consistent with Aspergillus, although the culture study for sinus secretion and repeated serum Aspergillus galactomannan antigen assay were negative. The patient underwent PNS MRI every two months while receiving voriconazole therapy. Since she was still on remission of DPLN with a minimal maintenance dose of steroid (daily 5 mg) and in the absence of any evidence of infection such as paranasal sinusitis and cerebral mycotic aneurysms on follow up MRI images, voriconazole was stopped seven months later.
Three months after stopping anti-fungal therapy, one small aneurysm was suspected at the right middle cerebral artery bifurcation on routine follow up MRI images. Transfemoral cerebral angiography revealed two aneurysms: one at the right middle cerebral artery bifurcation, and one at the anterior choroidal artery that was not seen on MRI (). Aneurismal neck clipping of the affected artery was done by a neurosurgeon ().
Chest CT and brain MRI images were performed regularly afterwards and up to one-year follow-up there was no evidence of further infections such as cerebral aneurysms, sinusitis and pulmonary nodules.


A 27-year-old Asian male with dyspnea upon exertion was referred to our outpatient cardiology clinic. The patient's dyspnea had worsened gradually over the prior 5-6 years and had been exacerbated over the past 3 months. The physical examination result was normal habitus, without Marfan's syndrome phenotypes. His vital signs were as follows: blood pressure, 126/78 mm Hg; pulse rate, 95 beats/minute; respiratory rate, 22 breaths/minute; body temperature: 36.7℃. Both jugular veins were slightly engorged. A high-pitched, blowing, holodiastolic murmur (grade V/VI) was clearly audible at the right sternal border of the third intercostal space. No crepitation or wheezing was observed in his chest, and his abdominal examination was normal. An electrocardiogram showed left ventricular hypertrophy and bi-atrial enlargement in normal sinus rhythm. Chest radiography revealed mild cardiomegaly without pulmonary congestion (). All blood chemistry profiles were within normal limits; they were as follows: fasting glucose 92 mg/dL, total cholesterol 214 mg/dL, creatinine 1.0 g/dL, blood urea nitrogen 21 mg/dL, uric acid 5.7 mg/dL. Echocardiogram showed left ventricular enlargement (end-diastolic dimension, 73 mm), left ventricular hypertrophy (septal wall thickness on diastole, 15 mm), and moderate left ventricular systolic dysfunction with a left ventricular ejection fraction of 36% (). Dimensions of the sinus of Valsalva and aortic valve opening were 56 mm and 34 mm (). Morphology of his aortic valve leaflets in a 2 dimensional echocardiogram was normal, but severe aortic valve regurgitation due to aortic root dilation was observed in a Doppler echocardiogram (). The chest CT confirmed marked aortic root dilation and ATAA (). Multiple variable-sized cystic lesions in the liver and both kidneys were observed in an abdominal-pelvic CT scan that was compatible with ADPCKD (). An operation was undertaken involving composite graft replacement of the aortic valve, aortic root and ascending aorta, otherwise known as the Bentall operation. The aortic root was excised and replaced with a 26-mm INTERGARD woven vascular graft (MAQUET GmbH & Co. KG, Rastatt, Germany). An aortic valve replacement was done with a Saint-Jude 25 mm mechanical aortic valve (St. Jude Medical, St. Paul, MN, USA). Both coronary arteries were re-implanted on the side of the graft. During weaning from cardiopulmonary bypass post-operatively, an intra-aortic balloon counter pulsation was inserted for prophylactic reasons against systolic dysfunction. In the pathological examination of the excised aorta, cystic medial necrosis, which had been defined as mucoid material accumulation, was noticed in the media on Hematoxylin and eosin (H & E) staining (). Necrotized, disorganized smooth muscle cells, and elastic fragmentations were observed in elastic fiber staining (). These findings were compatible with typical pathologic findings of large vessels in ADPCKD. The patient's recovery was quick, and it was without incident as far out as 4 years post-operation, with the exception of one small renal cyst rupture. After the operation, the patient has continued to take furosemide, spironolactone, angiotensin-converting enzyme inhibitor, and warfarin. The last measured serum creatinine level was 1.30 mg/dL (upper normal limit, 1.20 mg/dL).


A 65-year-old man presented to the emergency department (ED) after a computed tomography (CT) guided lung biopsy by interventional radiology with an acute cerebral air embolism. Prior to presentation, the patient had experienced progressive dyspnea for the prior few months and had an outpatient CT that revealed a lung mass. The patient subsequently presented to the interventional radiology suite for a CT guided lung biopsy, which later revealed adenocarcinoma. He received Versed and fentanyl for the procedure but remained alert and conversive during the procedure.
Shortly after the procedure, the patient became unresponsive. The rapid response team was activated immediately, but prior to their arrival, the patient became more alert. Upon assessment, the patient was noted to be aphasic, with left hemipareiss. A head CT was done and revealed right middle cerebral artery watershed area air embolism and hypodensity consistent with acute infarct ( and ). The chest CT showed no evidence of air embolism in the thoracic vasculature, but demonstrated a right pneumothroax, and a chest tube was inserted. The patient was not oriented to self, time, and place, but could follow simple commands intermittently. At that point, which was roughly about 30 minutes after the initial onset of symptoms, the patient was transported to the ED.
Upon initial assessment in the ED, the patient had the following vital signs: temperature of 99.4°F, pulse of 86, respiratory rate of 18, and blood pressure of 120/91. The blood glucose obtained by finger stick was 149. The patient was hemodynamically stable, alert, speaking a few words in a confused manner, and was not following simple commands. Physical exam revealed bilateral breath sounds, with slightly decreased breath sounds on the right and a right-sided pigtail catheter visibly in place. On neurological exam, the patient was noted to be flaccid in the left arm, with 0/5 in motor strength and not responding to noxious stimuli. He had 3/5 in muscle strength on the left lower extremity and was responding sluggishly to noxious stimuli.
Throughout his assessment, the patient was placed in Trendelenburg position while receiving 100% oxygen via nonrebreather mask. Hyperbaric oxygen treatment was entertained during evaluation of the patient. However, the patient presented with a concomitant pneumothorax, which is a contraindication that could be exacerbated by hyperbaric treatment. In addition, although the patient was stable upon presentation, the patient presented acutely following air embolism introduced into the vasculature and needed to be in a closely monitored setting, such as in the ED, that was prepared for resuscitation.
We, therefore, proceeded with the decision to intubate the patient and began inducing therapeutic hypothermia for neuroprotection. We used a commercial cooling device, and the target temperature was set at 31° to 33°C with the intention of maintaining the patient's temperature at 33°C for 24 hours. The patient was also given boluses of 4 mg lorazepam and 10 mg vecuronium and started on a titratable midazolam drip to prevent shivering and maintain sedation. The patient was maintained on therapeutic hypothermia for 24 hours in the surgical intensive care unit, and the patient was extubated on hospital day 2.
Upon initial assessment following extubation, the patient was alert, oriented, and exhibited 2/5 muscle strength in the left upper extremity and 3/5 muscle strength in the left lower extremity, which was an improvement from the left upper extremity muscle strength of 0/5 on initial presentation to the ED. The patient began physical therapy daily and continued to show improvement neurologically. A repeat head CT showed resolution of air emboli on hospital day 4, with evolving right parietal-occipital hypodensity. On hospital day 5, the patient was assessed to have 4/5 in muscle strength in left upper and lower extremities, and left lower extremity muscle strength improved to 5/5 on hospital day 6. With continued effort in encouragement of out-of-bed activities and physical therapy, patient's left upper extremity muscle strength improved to 5/5 on hospital day 8. Overall, the patient made a full recovery neurologically and was later discharged on hospital day 21. The delay in discharge was mainly due to the patient developing Clostridium difficile infection.


A 34-year-old male presented to an urgent care center, where he collapsed on arrival in the triage area, per providers in that department. He stated that he had emptied a full bottle of atropine eye drop solution into a glass of water and ingested it in an attempt to harm himself. The atropine concentration was 10 mg/mL, making for a total ingestion of 150 mg. On initial presentation, he had altered mental status with waxing and waning coherence, and when awake, he was very combative. He was also tachycardic with a heart rate (HR) of 125 beats per minute. A fingerstick glucose was normal. He was given 2 mg IV lorazepam, 4 mg IV ondansetron, 50 gm oral activated charcoal, and quickly transferred to a larger local hospital for further care.
In the emergency department at the accepting facility, the patient continued to have altered mental status, varying between severe sedation and uncontrolled agitation. His HR was 150 beats per minute, blood pressure (BP) 150/90 mmHg, respiratory rate 24 breaths per minute, and oxygen saturation 95% on room air. He had flushed skin, dry oral mucosa, nonreactive mydriasis, and a rectal temperature of 100.2°F. He showed no signs of trauma and had a nonfocal neurologic examination other than the gross altered mental status. The remainder of his physical exam was unremarkable. Electrocardiogram revealed sinus tachycardia and no interval or segment abnormalities.
Due to the intermittent somnolence and uncontrolled agitation, the emergency physicians at the accepting facility were concerned for the patient's ability to protect his airway enough to maintain oxygenation and ventilation. This, in combination with the recently administered charcoal and the possibility of emesis with subsequent aspiration, was enough cause for them to move towards rapid sequence induction (RSI) and endotracheal intubation. While preparing for the intubation, a medical toxicologist was consulted by the treating physicians. In an attempt to avoid the morbidity of the RSI, intubation, sedation, and mechanical ventilation, the decision was made to administer physostigmine to reverse the anticholinergic effects of the atropine.
Over the subsequent 75 minutes, physostigmine was titrated in 1 mg increments, each given over 3 to 5 minutes to a total dose of 11 mg. There was minimal change with the initial few doses, but after the fifth dose had been administered each 1 mg dose improved the patient's mental status along with normalizing the heart rate from the tachycardia listed above. Each of these signs would slowly worsen again over the ensuing 5 to 15 minutes, necessitating another dose. At the completion of treatment in the emergency department, the patient's HR had declined into the 90s beats per minute, and BP to 125/80 mmHg. His mental status was normalizing to the point where he was relatively calm and could provide a little bit of subjective history.
Laboratory evaluation at the second hospital included a negative serum ethanol, negative acetaminophen, normal basic metabolic panel, normal complete blood count, and negative urine drugs of abuse immunoassay for amphetamines, barbiturates, benzodiazepines, cannabinoids, cocaine, opiates, and phencyclidine. A serum atropine level later returned at 240 ng/mL (this was performed using the initial blood draw at the accepting facility).
He was admitted to the intensive care unit (ICU) for further observation and treatment. During the night in the ICU, he required 3 additional 1 mg doses of physostigmine for agitation that recurred. Endotracheal intubation was never performed. The remainder of the medical portion of his hospital stay was uneventful.


A 30-year-old man after sustaining a high-speed motorvehicle accident was referred to our tertiary level trauma center on second day postinjury. He had blunt trauma chest and fracture shaft of right femur. He was intubated in the emergency department for respiratory distress and a previously placed right intercostal drain (ICD) was repositionedprior to shifting the patient to intensive care unit (ICU).
He had decreased air entry in the right side of the chest with coarse crepitations. There was large continuous air leak from the right ICD and subcutaneous emphysema suggesting the possibility of tracheobronchial disruption. Despite a well-placed chest tube, chest radiograph showed persistent right pneumothorax and collapsed right lung []. Fiberoptic bronchoscopy done by anesthesiologist revealed the presence of a large rent of 1 cm at the level of carina []. The endotracheal tube was withdrawn under fiberoptic guidance, which showed no upward extension of the rent and finally the tube was left in situ just below the vocal cords. Computed tomography (CT) chest revealed fracture right 1st–3rd ribs with gross pneumomediastinum, right lung contusion, massive subcutaneous emphysema, and possibility of tracheal bronchus []. A second ICD was placed in 2nd intercostal space on the right side and connected to negative pressure suction. He was ventilated by assist-control mode ventilation with a tidal volume of 5 mL/kg, positive end-expiratory pressure (PEEP) of 3 cm (maximum PEEP at which there was no continuous air leak and prevented atelectasis) and the respiratory rate was adjusted according to the PaCO2 and pH. Peak inspiratory pressure, compliance, airway pressure, and resistance were monitored []. Adequate pain relief was ensured with 0.125% bupivacaine and 2 μg/mL fentanyl combination being administered through thoracic epidural positioned at D8–D9 level at rate of 4-7 mL/h. On third day postinjury, bronchoscopic sealing of the tracheopleural fistula was done using N-butyl-2-methyl cyanoacrylate (tissue glue). The flexible adult fiberoptic (size 4.9) scope was passed through endotracheal tube and the carinal rent was localized. A 5-French angiography catheter was passed through the working channel of the bronchoscope and 1 mL of the tissue glue along with lipoidol was instilled into the carinal rent. The continuous air leak in the ICD stopped at the end of procedure, lung expanded and pneumothorax relieved following the procedure and did not recur. Subsequently, the patient was extubated under fiberoptic guidance after 12 days of mechanical ventilation. Before and after the repair when the patient was on mechanical ventilation blind suctioning of the tracheal tube was avoided as it could further damage the injured carina.


A 48-year-old female presented with two year history of back pain and progressive difficultly walking. She gradually developed increasing numbness in both lower limbs over one year. She also developed increasing urinary retention. There was no history of trauma/fever or any other systemic disease. She was evaluated by an orthopedic spine surgeon and was referred to a physician following a negative lumbar magnetic resonance imaging (MRI). Neurological examination revealed presence of weakness in bilateral lower extremities (4-/5, Medical Research Council [MRC] grade) and asymmetrical sensory loss from T8 below consistent with a diagnosis of thoracic myelopathy (Nurick grade 4). Persistent symptoms with worsening gait imbalance led us to perform of thoracic spine MRI which surprisingly revealed presence of a focal anterior kink of the thoracic cord at the T7-T8 level (). The spinal cord at T7/8 level was narrow and deformed without any definite T2 signal abnormality with prominence of the dorsal subarachnoid space. The MRI imaging was suspicious of a diagnosis of anterior transdural spinal cord herniation (). Surgical management was planned and a unilateral right paramedian incision was used to perform a transpedicular exposure due to slight eccentric location of the spinal cord herniation to the right (). Intraoperative neuromonitoring was performed recording somatosensory evoked potentials (SSEPs) and motor evoked potentials (MEPs). T7/T8 pediculotomy was performed with partial costotransversectomy to provide the optimal anterolateral trajectory. A paramedian right dura incision was made, providing exquisite visualization of the anterior dural defect with the herniated spinal cord (). Using a Woodson dissector, the spinal cord was reduced back into the thoracic dura. The SSEP's were normal throughout the procedure. There was transient reduction in MEP amplitude during mobilization of the spinal cord to reduce the herniation. The MEP amplitude returned to baseline status an hour later. A Duragen (Integra Life Sciences Corp., Plainsboro, NJ, USA) patch was used as graft to seal the defect which was reinforced with Tisseel fibrin glue. (Baxter Healthcare Corp., Deerfield, IL, USA) No fusion or instrumentation was performed. Patient experienced improvement in sensory symptoms right after the surgery and had improvement in her pain, motor strength (4+/5, MRC grade) and gait at three months follow-up with improvement in her Nurick myelopathy grade to 2. Postoperative MRI revealed reduction of the spinal cord with no evidence of herniation ().


A 15-year-old male patient referred to the Department of Oral and Maxillofacial surgery, presented with a chief complaint of swelling on right lower one-third of the jaw since 2 months, with a history of slowly progressing swelling. The approximately 5 × 4 cm facial swelling extended superiorly from right corner of mouth to 1 cm inferiorly beyond the lower border of the mandible, medially from left para-symphysis region to distally half of the body of the mandible []. Intraoral examination revealed missing right and left mandibular canines, while in the maxilla the right canine and left maxillary second molar were missing with the left maxillary canine in erupting phase, and with rotated left maxillary premolars. Intraoral swelling was present in relation to mandibular anterior teeth []. Based on the history and clinical findings, a provisional diagnosis of dentigerous cyst in relation to right mandibular canine was made. The patient was subjected to the following radiographic examination.
Orthopantomograph revealed multiple unilocular well-defined radiolucencies with sclerotic border in maxilla and mandible []. Mandibular occlusal radiograph [] revealed a well-defined radiolucent lesion with a sclerotic border extending from periapical region of lower anteriors extending to the lower border of the mandible in relation to 33 and 43.
The presence of multiple cysts in the jaws, in association with unerupted teeth, raised a suspicion of Gorlin–Goltz's syndrome. The patient was further evaluated for this. Lateral skull view revealed bridging of the sella turcica []. Computed tomographic (CT) images showed multiple cystic lesions in maxilla [] and mandible []. Postero-anterior [] chest radiograph revealed a bifid third rib in the right side and an unusual finding of dextrocardia. CT images of the brain showed areas of hyperdensities, suggestive of falx cerebri [] and tentorial cerebelli [] calcification. CT images of spine revealed bifid spine in relation to C6 C7 T1 []. Ultrasonographic images showed the presence of spleen in the right hypochondrium [] and liver in the left hypochondrium [] with transposition on great vessels []. The dorsum of the patient revealed the presence of sprengel deformity. There were multiple palmer pits [] measuring 0.2–0.3 mm in diameter, which were brownish black in color, and were present on the palms of both his hands. Excisional biopsy was done for one of the cysts and was taken for microscopic examination, which revealed a stratified squamous parakeratinized epithelium with palisading pattern of basal cells [], suggestive of odontogenic keratocyst. Enucleation was done under local anesthesia followed by chemical cauterization with carnoy's solution since odontogenic keratocyst has high recurrence rate due to the presence of daughter or satellite cysts and highly adherent epithelial layer. The patient was asked to have a regular follow up.
Since the patient fulfilled the three major criteria for multiple odontogenic keratocysts (palmer pits, falx cerebri and tentorial cerebelli calcification) with three minor criteria of bifid rib, spina bifida and sprengel deformity, the final diagnosis was made as Gorlin–Goltz syndrome with situs oppositus (also called situs transversus or situs inversus), which is a congenital condition in which the major visceral organs are reversed or mirrored from their normal positions.


A 32-year-old male patient reported to the Department of Oral and Maxillofacial Surgery in October 1993 with a chief complaint of swelling on the left side of lower jaw. The swelling was hard and had a duration of 6 months. It was gradually progressing in size and was associated with pain and paresthesia for the past 1 month. On I/O- Intraoral examination, the swelling extended from left lateral incisor upto second molar. There was more expansion of buccal cortex than lingual cortex. Expansion had caused the obliteration of buccal vestibule. On radiographic examination, well-defined multilocular radiolucency extending from lateral incisor to second molar was seen []. Incisional biopsy was done under local anesthesia. On h/p- histopathological examination, ameloblastoma was confirmed. Segmental resection of lesion with 1 cm of normal bone was done []. After 6 months of resection, reconstruction was done with nonvascularized iliac crest bone graft and stabilized with AO reconstruction plate []. The patient was kept under regular observation for a period of 6 months. In due course of time, mandibular prosthesis was given as replacement for missing teeth. No complications in the postoperative period were reported till 2008 [].
In December 2008, the patient reported to the department with a complaint of painless swelling in the labial vestibule, from the past 2 months, in relation to apical region of right lateral incisor and canine teeth. Aspiration revealed 2–3 ml of straw-colored fluid, after which the swelling subsided, which later recurred in 2 weeks. On radiographic examination [orthopantomogram (OPG)], multilocular radiolucency at the level of right symphysis region was seen with no involvement of the bone graft []. Because the lesion was completely contained within the healthy bone, marginal resection of the bone was done under local anesthesia with 1 cm margin []. On microscopic examination, recurrence of ameloblastoma was confirmed. Graft had consolidated markedly well with normal bone and to our surprise, relapse of the lesion was noticed on normal side and no recurrence was noticed on the bone graft []. The patient was kept under regular follow-up after every 3 months, no recurrence has been reported till date [].


A 23-year-old female reported in the outpatient department with the chief complaint of an asymptomatic bony hard swelling in respect to the right posterior mandible, which gradually increased in size in the past 3 years to attain the present size of concern. Dental and medical history was insignificant. On clinical examination, there was evidence of a large non-tender, non-compressible, hard swelling measuring 2 × 2 cm on the right body of mandible, about 1 cm anterior to the posterior border of ramus, but no lymphadenopathy was noted. Overlying skin was normal in color, texture and consistency and was not adherent to the underlying swelling.
Intraoral examination revealed a painless swelling in the right posterior mandibular vestibule from right mandibular second premolar to the distal aspect of second molar on the same side. The swelling was firm and covered by normal mucous membrane []. In place of the right mandibular first molar, only root stumps were present, but molar and premolar adjacent to it tested vital.
Radiographically, it presented as a unilocular, radiolucent lesion, well circumscribed by a radiopaque border, about 3 × 2 cm, extending from the root of second molar posteriorly to the root of the first premolar anteriorly []. The lesion also caused a displacement of the right inferior alveolar canal toward the cortical inferior border of the mandible.
The lesion was perforated with a wide bore needle to rule out vascular lesion and aspirate the content. The aspirate was sent for pathological evaluation and the report stated it to be a cystic fluid with inflammatory exudates, confirming the clinical and radiographic diagnosis of radicular cyst in association with root stumps of 46.
So, enucleation of cyst with extraction of root stumps of first molar was planned. Under local anesthesia, an incision was made from distal of third molar to the medial of canine. Mucoperiosteal flap was raised and it was possible to see a thin expanded cortical plate which was removed with a scalpel. The cavity contained fibrous tissue wall full of liquid.
The lesion with all its fibrous tissue lining was enucleated carefully to ensure complete removal. The remaining bone tissue showed normal contour and consistency without any clinical signs of lesion. Root stump of first molar was extracted and mucoperiosteal flap sutured back after thorough curettage and lavage of bony cavity.
The histopathologic examination of the removed lining revealed UA contrary to the provisional diagnosis of radicular cyst []. Hence, root canal of 47 and 45 was done and radiologic observation of patient to monitor the relapse of lesion was chosen as the well-defined cystic envelope ensured the complete removal.
Presently, the patient is under follow-up since 1 year without any signs of relapse and with bony restoration in the affected area [].


An 11-year-old male child reported to the Department of Pedodontics and Preventive Dentistry, Saraswati Dental College, with the chief complaint of swelling in right back region of upper jaw. His past medical and family histories were non-contributory. The swelling had been first noticed 18 months back which gradually increased to the present size. Initially swelling was asymptomatic but had become painful on closure of jaw due to the contact pressure from lower molars. On extra-oral examination, swelling was evident on right side of the face leading to slight facial asymmetry []. Patient presented with adequate mouth opening and intraoral examination revealed swelling of size approximately 2 × 2 cm distal to 16 with buccal as well as lingual expansion and indentation imprint of lower molar []. Swelling was accompanied by marked buccal displacement of 16 and missing/unerupted 17. The overlying oral mucosa was intact and normal in appearance. Swelling was non-tender and bony hard in consistency with well-defined margin. There were no palpable cervical or submandibular lymph nodes.
Panoramic radiographical examination [] revealed a well-defined homogenous radiopaque mass distal to the tooth 16 and coronal to un-erupted tooth 17 with radiolucent margin. There was distal and superior displacement of tooth 17 with its root dilacerations. Tooth follicle of 18 was also present. CT scan [Figure –] showed a well-circumscribed and sharply defined lesion consists of a mixture of radiolucency and radiodensity with sclerotic border.
Excisional biopsy of the mass was planned under local anesthesia. After achieving adequate anesthesia, incision was given directly over the alveolar ridge swelling that extended anteriorly in the crevice of maxillary first molar to its mesial aspect and from here a vertical releasing incision was given. A full-thickness mucoperiosteal flap was reflected buccally and palatally. On exploration, the mass was well-demarcated from its surrounding bone and easily separated from the bony bed. The lesion was enucleated and was taken out in toto and sent for histopathological examination. Tooth 17 was also extracted during the procedure because of its extreme buccal malposition [] and to facilitate the complete enucleation of the lesion. The extracted 17 was presented with root dilacerations with its palatal root curved sharply in the palatal direction while mesiobuccal and distobuccal roots curved distally [Figure and ]. The wound was closed in single layer with 3-0 black silk suture. Healing was uneventful on subsequent postoperative follow-up.
Grossly, the specimen was a well-circumscribed mass measuring about 2.5×2.2×1.6 cm [] with areas of hardness. Radiograph of the excised specimen revealed radiolucency in the peripheral region and mixed radiopacity and radiolucency in the central region []. On cutting, the mass was hard and gritty in consistency. Microscopical examination of decalcified sections revealed a well-circumscribed fibrous connective tissue mass showing areas of mineralization. The hard tissue was present in the form of trabeculae of mature lamellar bone, immature woven bone and areas of unmineralized osteoid []. The connective tissue adjacent to the bone was delicately fibrillar composed of spindle-shaped cells. The trabeculae of woven bone and osteoid showed brush border at the periphery and were lined by plump osteoblasts []. Focal areas of osteoclastic activity in form of multinucleated osteoclasts and prominent reversal lines were also obvious. No cellular atypia or increased mitosis was seen. Based on the above histopathological features and correlating it with the clinical and radiological findings, a final diagnosis of central ossifying fibroma was made.


A 34-year-old, gravida 2 para 1 parturient was scheduled for Cesarean section at 38 weeks of gestational age. At age 19, she presented with paresthesias, headaches, neck pain, and poor balance, and Type I ACM with an extensive syringomyelia from the cervical to the lumbar area. Over the next several years, she had undergone surgical treatments, including suboccipital craniectomy, duraplasty with fascia lata graft, and decompressive C1–C6 laminectomies with placement of a syringopleural shunt. As a consequence of the laminectomies, the patient had residual cervical xyphosis with limited extension and continued to have symptoms related to Type I ACM, such as headaches, neck pain, stiffness, poor balance, and bilateral paresthesias of the upper and lower extremities (left side worse than right). Her symptoms were worsened by straining, coughing, and neck movements. Due to her residual Type I ACM, her neurosurgeon recommended avoiding normal vaginal delivery. The preoperative anesthetic evaluation showed a Mallampati grade III airway, limited mouth opening to 4 cm, and limited neck movement, with an anticipated difficult airway intubation. Recent MRI imaging of the cervical spine demonstrated worsening of the post-laminectomy cervical kyphosis, spinal cord atrophy, and a residual syrinx. The spinal cord was deviated posteriorly, the MRI of the thoracic spine demonstrated a syrinx in the entire thoracic cord, and the MRI of the brain showed structures of the hindbrain protruding through the foramen magnum [Figures –].
The anesthesiology team, in conjunction with the obstetrical team, decided to perform awake fiberoptic intubation and utilize a multimodal general anesthesia. The patient and her husband were informed of an anticipated difficult orotracheal intubation. On the day of surgery, the patient was brought to the operating room with fetal heart rate monitoring in place. She received 0.2 mg IV glycopyrrolate, famotidine 20 mg IV, and 30 ml Shol's solution pre-operatively. Standard American Society of Anesthesiologists (ASA) monitors and a Bispectral Index (BIS) monitor were applied. The neck was maintained in a neutral position. Adequate local anesthetic topicalization with Cetacaine (Cetylite Industries, Inc., Pennsauken, NJ, USA) topical oral spray (benzocaine 14%, butamben 2%, and tetracaine 2%) and 5 ml of lidocaine 2% jelly (Uro-Jet, International Medical Systems, Ltd., South El Monte, CA, USA) as a gargle solution were given in preparation of awake fiberoptic intubation. The patient received butorphanol 1.5 mg IV for sedation. Transtracheal 4% lidocaine 4 ml was administered. As the operative field was prepped and sterilely draped, the patient was properly prepared to undergo awake fiberoptic intubation with the head in the neutral position and with no neck movement. Utilizing an Ovassapian fiberoptic airway (Hudson RCI, Research Triangle Park, NC, USA), a pediatric bronchoscope (preloaded with a 6.5 endotracheal tube (ETT)) connected to an O2 source was advanced into the trachea. It was noticed that the epiglottis was “floppy,” angulated, the glottic opening kinked, and was cephalad and anterior due to the significant cervical kyphosis. No coughing or gagging was encountered, and the ETT was advanced to the supracarinal position and was secured.
General anesthesia with the neck in the neutral position was induced with propofol (2 mg/kg) IV as soon as proper ETT placement was confirmed by identifying breath sounds plus end-tidal CO2 (EtCO2) in addition to the fiberoptic visualization. Sevoflurane 2.0% end-tidal was initially used until the baby was delivered and then anesthesia was maintained with sevoflurane 1.0% end-tidal, plus an intravenous infusion of propofol (100 mcg/kg/min), midazolam 5 mg, and fentanyl (3 mcg/kg). A deep balanced anesthetic level was used without neuromuscular blockade. A healthy baby boy with Apgar scores at 1 and 5 min of 9/9 was delivered. The remainder of the case was without incident. The surgical team noted excellent uterine tone, and hemorrhage was less than average estimated blood loss ((EBL) < 700 ml). Post-intubation neurological assessment was performed and a prolonged in-house neurological observation was conducted. The patient had no complaints of headache or pain in the post-anesthesia care unit (PACU) or evidence of recall of the intraoperative events. She was discharged home after 4 days. Neurological exam showed no change in the patient's strength, sensation, or gross coordination. The patient remained stable at the outpatient clinic visit (2 weeks after Cesarean section), and she was advised to follow-up with her neurosurgeon with regards to progressive cervical kyphosis.


A female aged 62 years presented with painful swelling in the right breast since two years, which was gradually increasing in size since six months. Clinical examination revealed a hard mobile lump measuring about 5 × 4 cm2 in the upper outer quadrant of the right breast. There was no ulceration of overlying skin and no nipple discharge.
Fine needle aspiration smears revealed loose clusters and isolated population of tumor cells showing abundant finely granular and vacuolated cytoplasm []. The tumor cells had eccentrically placed nuclei with mild pleomorphism and inconspicuous nucleoli []. There was no colloid material in the background and no evidence of mitotic activity in the tumor cells. A possibility of secretory carcinoma of the breast was suggested.
The patient underwent modified radical mastectomy and the specimen was sent to pathology department for histopathological examination. Cut section of the specimen revealed circumscribed firm grey-white lesion with tiny cystic areas measuring 5 × 3 × 2.5 cm3 in size []. Axillary pad of fat showed twelve lymphnodes.
On microscopic examination, the sections revealed tumor cells arranged in solid sheets, ducts, tubular and microcystic pattern []. The cystic spaces showed eosinophilic secretions. The tumor cells were round to polygonal exhibiting mild atypia. Some of the cells showed intracytoplasmic vacuolation. The secretions in the lumen of microcysts were PAS positive. The tumor cells showed carcinoembryonic antigen (CEA) positivity on immunohistochemistry []. The sections from the lymph nodes showed reactive hyperplasia without tumor deposits.


A 27-year-old male presented with an eight-month history of painful anterior abdominal wall swelling. Physical examination showed a superficial tumor, measuring 4 × 3 × 2 cm in size with ulceration. FNAC was performed.
The smears were moderately cellular. The cells were arranged singly and in clusters, were polygonal in shape, with moderate amount of vacuolated cytoplasm and enlarged hyperchromatic nuclei. Few cells were multinucleated. Intranuclear inclusions were noted in some cells []. There was a myxoid stromal component in a necrotic background, with abundant neutrophils. There were no clear-cut intracytoplasmic lumina within the erythrocytes. A provisional diagnosis of poorly differentiated carcinoma, possibly metastatic, was made, and a biopsy was advised.
The biopsy showed a tumor composed of round to spindle-shaped cells in solid nests. The cells were embedded in a light blue to deep pink myxohyaline matrix. The cells were seen extending centrifugally from large vessels into the soft tissue. The tumor had a blistered, vacuolated appearance due to the intracytoplasmic lumina, many of which contained erythrocytes. The cells had moderate-to-abundant cytoplasm, almost uniform with a vesicular nucleus and prominent nucleoli in some. There was no increase in mitotic activity.
CD 34 was positive revealing the endothelial nature of the cells.
A diagnosis of epithelioid hemangioendothelioma was made.


A 13-year-old girl presented to the Ear, Nose and Throat (ENT) outpatient department (OPD) with unilateral enlargement of the right tonsil. A computed tomography (CT) scan showed a vascular mass around the carotid artery extending up to the base of the skull. Some degree of bone destruction was also seen at the base of the skull. Ultrasonography (USG) guided FNAC was done.
The smears were moderately cellular and diluted with blood. The cells were small and round, with scanty cytoplasm. They were arranged in small clusters as well as singly. Occasional acini-like structures were also seen []. The chromatin was finely granular. A provisional diagnosis of a ‘small round cell tumor, could be consistent with alveolar rhabdomyosarcoma,’ was made.
An effort was made to remove the tumor. Sections showed a tumor mass composed of intra-vascular papillary structures lined by hobnail endothelial cells. Some glomeruloid bodies were also noted. The cells had scanty eosinophilic cytoplasm with a prominent nucleus and little or no cytological atypia. Mitotic figures were rare.
CD 34 was positive revealing the endothelial nature of the cells.
A diagnosis of papillary intralymphatic angioendothelioma was made.


A 60-year-old female patient was admitted to the inpatient treatment section from the outpatient therapy wing of the physical medicine and rehabilitation department for the evaluation and treatment of impaired walking due to gradual weakening of the bilateral lower extremities that had lasted for three months. The patient seemed to have cognitive impairment, and did not have accurate memory of the trauma. However, according to the report of a family member, the patient had been medicated for cerebral infarction, hypertension, and osteoporosis, with no problems in ambulation and other activities of daily living before slipping down three months ago. After that, she had been in a bed ridden state and showed gait disturbance. Based on neurological examination, her mental status was alert, but her orientation, memory, calculation and other cerebral functions were generally impaired, whereas her speech was intact. In terms of the cerebellar function, there were no specific findings on both of the upper extremities, and the lower extremities could not be checked due to muscle weakness. Although an accurate evaluation of motor function was challenging during the manual muscle test, the upper extremities were found to be in normal range. For the lower extremities, the flexor and extensor of the hip and knee of both sides were in poor grade, and the dorsiflexor and plantarflexor of both ankle joints were degraded to trace grade level. There was no limitation in passive range of motion. In both lower extremities, the spasticity corresponded to the Modified Ashworth Scale I grade. Although accurate evaluation for sensory function was also difficult, the patient showed a relatively intact result in the pinprick test, but the proprioception and vibration sensation were impaired. Deep tendon reflex was normal for both upper extremities but increased in both lower extremities, and Barbinski's signs and ankle clonus were found on both sides. Results of the functional evaluation showed impossibility of independent ambulation. Although the activities of daily living using the upper extremities were possible, the patient needed considerable assistance in other areas. In terms of urinary function, urinary retention was observed, so we inserted a foley catheter since the patient was incapable of self-voiding. In addition, due to severe constipation, medication was provided and we helped defection with the use of glycerine enema, 2-3 per day as needed. The first lumbar vertebral body bursting fracture was found in the simple film taken at admission. From the magnetic resonance imaging (MRI) of the lumbar spine, the spinal dural arteriovenous fistula was found in the first lumbar vertebral body bursting fracture in addition to the shades of dilated vein in the periphery of the spines starting from the fifth thoracic vertebra to the cornus medullaris. As a result, the spinal cord edema was accompanied, and the specific findings of high signal, light shade of the spine were observed from the T2 weighted image (). Although electromyography and somatosensory evoked potential test were considered, we could not conduct them because of the patient's transfer to the neurosurgical department due to urgent need of arterial embolization. For more accurate location mapping of the spinal arteriovenous fistula, we conducted spinal vessel angiography and found that the arteriovenous fistula originated from the twelfth intercostal artery and first lumbar radicular artery. In addition, the spinal vein located on the anterior and posterior spine that continued to the cervical vertebra region was observed (). Since the artery of interest was not that large, we planned the embolization procedure employing glue (n-butyl cyanoacrylate) rather than the operation. After embolization of the number twelve right thoracic vertebrate intercostal artery and first lumber, the embolization of all fistula was followed. As a result, there was no venous plexus observed around the spine (), and the neurologic symptoms of the patient improved. The hip flexor and knee extensor improved to 'fair grade' and the ankle plantar flexor improved to 'poor grade'. Although we were not able to check the sensory function due to the patient's impaired cognition, big changes were not observed after the treatment. As a result of the post therapy follow-up using MRI to see the condition of spinal cord edema, we found that the spinal vein was dilated due to arteriovenous fistula not being found, as well as the spinal cord edema around the artery. Therefore, we were able to observe normal low signal intensity of the spinal cord from the T2 weighted image (). With the help of rehabilitation therapy including strengthening and endurance exercise of the lower extremities, gait training, and functional electrical stimulation to both tibialis anterior, the patient showed gradual improvement and could ambulate about 2-3 m. The urinary symptoms also improved and the patient was able to self-void after removal of the foley catheter following normal functioning results based on the urodynamic study. Bowel function also improved and the patient was capable of daily defecation.


A 45-year-old female was referred to our hospital for evaluation of an asymptomatic swelling of 6 months duration. The patient did not have any pain or discomfort other than occasional watery discharge from the lesion.
Clinical examination revealed a swelling on the labial gingiva in relation to the mandibular right canine and mandibular right first premolar. It was about 0.5 cm x 0.5 cm in size, pale yellow in color and firm in consistency []. Intra - oral periapical radiograph showed no osseous involvement [] and the teeth were vital.
A complete oral prophylaxis was performed. The lesion was completely excised and sent for histopathological examination. The patient was placed under follow-up.
Microscopically, hematoxylin and eosin - stained sections at multiple levels showed numerous salivary gland lobules composed of normal - appearing serous acini and ducts. The salivary gland lobules exhibited a sparse chronic inflammatory infiltrate. [].


A 77-year-old woman (height, 150 cm; weight, 50 kg) received spine surgery under general anesthesia in a local hospital. No other medical history and specific abnormal laboratory results were recorded except the arterial hypertension for about 20 years.
On postoperative day 2, the patient complained of discomfort on the anterior chest and neck with dyspnea. The chest X-ray showed pneumomediastinum and subcutaneous emphysema. The patient was then referred to our hospital. A computed tomography (CT) scan of the chest visualized the injury of the posterior tracheal wall due to the rupture of the pars membranosa ().
An emergent operation was decided, and was initiated as induction of anesthesia by propofol and remifentanil target controlled infusion (TCI). Before tracheal intubation, a fiberoptic bronchoscopy revealed a longitudinal hole with sharp margins at the posterior part of the membranous portion, with a total length of 6 cm approximately 3 cm from the carina. Tracheal intubation was performed by 35 Fr double lumen tube for one lung ventilation. Tracheal laceration was repaired without any specific event.


A 52-year-old woman (height, 158 cm; weight, 60 kg) underwent spine surgery due to spinal stenosis at the same hospital as case 1. She was transferred to our ER due to dyspnea and subcutaneous emphysema approximately 3 hours after the operation.
Her medical history was recorded as arterial hypertension and diabetes 18 years ago, and she was treated by administration of angiotensin receptor antagonist and insulin pump. When she was admitted at our hospital, pneumomediastinum, subcutaneous emphysema and pneumothorax were found on chest X-ray () and chest CT. Bronchoscopy confirmed a linear laceration of 3-4 cm in length on the posterior membranous wall of the mid-trachea (). An operation was decided after chest tubing on the left side.
Anesthesia was induced by propofol and remifentanil TCI, and tracheal intubation was taken by an ID 7.5 mm reinforced tube through the injured mid-trachea. The tube was advanced into the left main bronchus by fiberoptic bronchoscopy for one lung ventilation after the operation had begun. The trachea was repaired successfully, and the patient was discharged in good condition from the hospital.


A 73-year-old woman (height, 153 cm; weight, 53 kg), who underwent spine surgery at the same hospital as case 1 and 2, had mild subcutaneous emphysema on postoperative day 2. Pneumomediastinum and subcutaneous emphysema were presented on chest X-ray. Chest CT revealed a laceration on the posterior membranous wall of the mid-trachea (). However, the patient's symptoms were mild enough to recover by close observation with conservative treatment.


A 53-year-old man with end-stage renal disease was referred for urological evaluation of massive scrotal edema of 1 month duration. Continuous ambulatory peritoneal dialysis had been performed for the preceding 8 months. The patient had no trauma, hematuria, or pain and no signs of overhydration. Blood urea nitrogen and serum creatinine levels were 71.1 and 13.3, respectively. Potassium was slightly elevated. On physical examination, the abdomen was soft and nontender. The peritoneal catheter was functioning normally. Both the scrotum and the penis were swollen and nontender (). The scrotum transmitted light. Ultrasonography was performed to exclude an inguinal hernia. On ultrasonography, patency between the peritoneal cavity and the right inguinal canal was revealed and there was a large hydrocele (). However, there was no sign of bowel hernia. Peritoneal dialysis was temporarily replaced by hemodialysis to allow the edema to subside. Surgical repair was done. During the operation, the inguinal sac was found and high ligation was performed. The patient had complete resolution and returned to ambulatory treatment uneventfully at 1 month after the operation. However, the patient revisited the clinic for recurrent scrotal and penile swelling at 2 months after the operation. Although we tried intermittent peritoneal dialysis with a low dialysate volume, the genital edema was resolved only after hemodialysis was performed about 3 months later.


A 43-year-old male visited the Department of Family Medicine with a history of mild headache, neck pain and nausea which had been ongoing for a day. He had a history of hypertension for 2 years and his blood pressure was 145/95 mmHg on his first visit. The patient had a chief complaint of a headache in both the occipital and temporal areas and tightness in the posterior neck which were aggravated by standing, coughing and shaking of the head. On the other hand the symptoms were relieved by lying down. Simple analgesics and muscle relaxants were prescribed, but they did not ease the headache. On the sixth day since the onset of headache, the patient experienced blurred and double vision and this led him to the hospital again. On neurologic examination, he had horizontal diplopia which became worse when gazing to the right side (). Ophthalmologic consultation confirmed right sixth cranial nerve palsy, and the consulting neurologist recommended performing brain and orbit MRI. The brain MRI showed diffuse pachymeningeal thickening and enhancement (). The patient was admitted on the supposition that intracranial hypotension might have caused the headache and sixth nerve palsy. Spinal tap was performed and revealed an opening pressure of 11 cmH2O in the right decubitus position. A CT myelogram was also taken, and contrast leakage was found in the posterior epidural space from C4 to C6 (). The patient's symptoms, MRI finding, and CT myelogram could all be explained by spontaneous intracranial hypotension in spite of the normal CSF pressure. He was referred to the pain clinic for EBP 10 days after the onset of the diplopia. We performed the EBP at the C4/5 level under fluoroscopic guidance by injecting 8 ml of autologous blood (). The headache was nearly relieved by the following day, but the diplopia was only partially improved. After discharge from the hospital, the patient was observed for 2 months and we confirmed his complete recovery from the diplopia ().