Aggressive hilar inflammatory myofibroblastic tumor with hilar bile duct carcinoma in situ

Inflammatory myofibroblastic tumor (IMT) of the biliary tree is extremely rare and is generally a benign condition, though malignant change is possible. Making a differential diagnosis between this lesion and other malignant conditions is very difficult on preoperative imaging studies. Hence, the final diagnosis of IMT may be made during or after operation depending on the pathologic examination. We treated a 63-year-old woman who received right hepatectomy with caudate lobectomy under the suspicion of hilar cholangiocarcinoma. Frozen biopsy during the operation showed carcinoma in situ and there were stromal cells in the bile duct's resection margins. The postoperative hospital course was uneventful except for minor bile leakage. At postoperative month 4, she developed jaundice, ascites and pleural effusion. Computed tomography images showed a mass-like lesion in the porta hepatis with portal vein thrombosis and a right chest wall mass. Excisional biopsy was done and the pathology report was malignant spindle cell tumor suggestive of an aggressive form of IMT. Her condition rapidly deteriorated regardless of the best supportive care and she expired at postoperative month 5. Further investigation is necessary to clarify the reasons for recurrence and infiltration of this disease

A Bile Based Study of Clonorchis sinensis Infections in Patients with Biliary Tract Diseases in Ulsan, Korea

Stool examination is believed to be the most reliable method for detecting Clonorchis sinensis (CS) eggs. However, it has limited value for diagnosing clonorchiasis when the biliary tract is obstructed or when there is a light infection. We evaluated the infection states of CS in patients with biliary tract diseases using a bile sample. From January 2001 to August 2003, 238 patients who had undergone endoscopic biliary drainage were prospectively included in the study. The patients' bile samples were obtained directly from the nasobiliary drainage tube and then analyzed to detect CS eggs. The overall CS egg positive rate was 28.2% (35.4% in males, 19.4% in females). The egg positive rate was similar in all age groups examined: 26.7% in 30-39 years, 25.0% in 40-49 years, 24.4% in 50-59 years, 30.2% in 60-69 years, 35.3% in 70-79 years, and 25.0% in 80 years of age and over. There were no significant differences in the egg positive rate between the disease groups: 32.6% in bile duct cancer, 38.5% in gallbladder cancer, and 26.4% in gallstone diseases. Our results show that the CS infection rate was very high, regardless of the age, gender, and type of diseases of the patients. Although the study population was limited to patients with biliary tract diseases, it is assumed that clonorchiasis is still an endemic disease in Ulsan, Korea

Eosinophilic Gastroenteritis with Eosinophilic Dermatitis

Eosinophilic gastroenteritis (EG) is characterized by eosinophilic infiltration of the bowel wall and variable gastrointestinal manifestations. Clinicians should have a high index of suspicion for EG when faced with gastrointestinal symptoms and peripheral eosinophilia to avoid incorrect diagnosis and inappropriate treatments. A 24-year-old woman was admitted to our hospital complaining of acute right lower quadrant abdominal pain and a laparoscopic appendectomy performed for a presumed diagnosis of an acute appendicitis. However, the procedure revealed bowel edema and a moderate amount of ascites without evidence of a suppurative appendicitis. Postoperatively, she showed persistent and progressive eosinophilia, exudative eosinophilic ascites, eosinophilic infiltration of the resected appendix wall, and eosinophilic infiltration of gastroduodenal mucosa. A punch biopsy of the abdominal skin also revealed inflammation with marked eosinophilic infiltration of the skin. She recovered after the treatment with a low dose of steroid for the EG with eosinophilic dermatitis. EG with eosinophilic dermatitis has not been reported yet and is considered fortuitous in this case

Unusual Presentation of Cystic Lymphangioma of the Gallbladder

Cystic lymphangioma of the gallbladder is quite a rare tumor with only a few cases having been reported in the literature. We describe here a rare case of cystic lymphangioma of the gallbladder, which was unusual in that the patient presented with biliary pain and an abnormal liver test. Ultrasonography and computed tomography of the abdomen showed a multi-septated cystic mass in the gallbladder fossa and an adjacent compressed gallbladder. Endoscopic retrograde cholangiography showed there was no communication between the bile tract and the lesion, and there were no other abnormal findings with the exception of a laterally compressed gallbladder. After performing endoscopic sphincterotomy, a small amount of sludge was released from the bile duct. The histological findings were consistent with a cystic lymphangioma originating from the subserosal layer of the gallbladder. This unusual clinical presentation of a gallbladder cystic lymphangioma was attributed to biliary sludge, and this was induced by gallbladder dysfunction that was possibly from compression of the gallbladder due to the mass

A Case of Santorinicele without Pancreas Divisum: Diagnosis with Multi-detector Row Computed Tomography

A santorinicele is defined as a focal cystic dilatation of the terminal portion of the dorsal pancreatic duct at the minor papilla. Most cases reported previously were associated with pancreas divisum and a santorinicele without pancreas divisum is known to be rare. We recently experienced a typical case of a santorinicele without pancreas divisum in a 67-yr-old woman with abdominal pain and hematochezia, subsequently proven to be the result of an ischemic colitis. The santorinicele was diagnosed incidentally with multi-detector row computed tomography using a minimum intensity projection technique, which clearly showed a cystic dilatation of the terminal portion of the dorsal pancreatic duct and a communication between the ventral and dorsal pancreatic ducts. This finding was also confirmed by a magnetic resonance cholangiopancreatography

A Case Report with Lymphangiomatosis of the Colon

The incidence of lymphangiomas in the gastrointestinal tract is low, particularly in the colon and rectum, and most cases are solitary. Lymphangiomatosis of the colon are encountered infrequently with only one report in the English literature, and polypectomy was performed for the diagnosis in that case report. However, trends in the diagnosis of lymphangiomatosis of colon have been changing since the development of endoscopic ultrasonography (EUS), and this case is the first in that lymphangiomatosis of the colon was diagnosed without invasive procedures. Here we describe the case of 31-yr-old woman with lymphangiomatosis of the colon with numerous polyposis-like appearing lesions diagnosed by endoscopic ultrasonography and a colonoscopy