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An 84-year-old man was admitted with epigastralgia. Computed tomography showed contrast-enhanced wall thickness in the cystic duct. An endoscopic examination revealed short irregular stricture in the cystic duct, and per-oral cholangioscopy revealed a reddish papillary tumor at the stricture site. Surgical resection revealed high-grade biliary intraepithelial neoplasia (BilIN) at the stricture site of the cystic duct. To our knowledge, this is the first case of a solitary high-grade BilIN epithelium in the cystic duct detected by per-oral cholangioscopy.

Pancreatic serous cystic neoplasms (SCNs), such as serous cystadenoma (SCA), are generally recognized as benign because malignant counterparts of SCNs have been extremely rare. In clinical practice, pancreatic cystic neoplasms diagnosed as SCNs have been managed by conservative observation, as long as the patients remained asymptomatic. We herein report a case of metachronous ductal adenocarcinoma that was discovered during long-term follow-up of SCN and review the related literature. To our knowledge, this was the first reported case of the local presence of ductal adenocarcinoma adjacent to SCA that was preoperatively diagnosed by endoscopic ultrasound-guided fine-needle aspiration.

We herein report a case of a 56-year-old man with IgG4-related sclerosing cholangitis (IgG4-SC) with no biliary stricture, but with a severely thickened bile duct wall. Contrast-enhanced computed tomography showed diffuse swelling of the pancreas and thickening of the common bile duct (CBD) wall with delayed enhancement. Obvious diffuse wall thickening of the CBD was observed on endoscopic ultrasonography. However, endoscopic retrograde cholangiography showed no biliary stricture in the CBD that had thickened. Although IgG4-SC has been classified by a stenotic lesion on cholangiography, we should be aware of some IgG4-SC cases showing only bile duct wall thickness without any biliary stricture.

An 80-year-old man was admitted with anorexia, a high fever and general icterus. Laboratory examinations revealed remarkable inflammation and elevation of liver/biliary enzymes. Computed tomography (CT) showed a high-density lesion in the bile duct, and endoscopic retrograde cholangiopancreatography (ERCP) disclosed an oval filling defect mimicking choledocholithiasis. Plastic stents were inserted to treat the acute cholangitis; however, the patient's symptoms recurred. An erythematous hypervascular mass obtained with a balloon catheter contained numerous pleomorphic histiocytic cells with eosinophilic cytoplasm, remarkable anisonucleosis and occasional mitoses. A diagnosis of histiocytic sarcoma (HS) was made based on the results of intensive immunohistochemistry. Monoclonal rearrangement of the IGH and TCRG genes, IGH split and IGH/BCL2 fusion was negative, although polysomy 8, 14, and 18 was detected. The patient was treated conservatively and died of the disease 20 months after the initial diagnosis. To the best of our knowledge, this is the first case of bile duct HS. This case, which involved numerical alterations of chromosomes, presented with CT and ERCP findings similar to those of choledocholithiasis.