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A 61-year-old man with asthma, diabetes mellitus, cirrhosis secondary to chronic hepatitis C virus (HCV) infection, and hepatocellular carcinoma (HCC), was admitted to our hospital because of progressive abdominal pain and jaundice of 7 days duration. On clinical examination, the patient had abdominal distention, tenderness in the right upper quadrant, and jaundice. His hemoglobin was 8.7mg/dL; hematocrit, 25%; white blood cells, 1.7×109/L; international normalized ratio (INR), 1.4; total bilirubin, 15mg/dL; direct bilirubin, 7.6mg/dL; indirect bilirubin, 7.4mg/dL; and alkaline phosphatase, 65 IU/L. The remaining laboratory parameters were within reference ranges. A computed tomography scan (CT) revealed cirrhosis, HCC, portal vein thrombosis and choledocholithiasis with intrahepatic filling defects and ductal dilation (●" Fig.1). During endoscopic retrograde cholangiopancreatography (ERCP) marked portal hypertensive gastropathy and duodenopathy was evident. ERCP showed irregular filling defects within the entire common bile duct (●" Fig.2). During standard cannulation the wirewould not advance into the bile duct because of the presence of the large impacted filling defect. Therefore, a biliary precut-fistulotomy was performed with subsequent extraction of a worm-shaped structure (“biliary cast”) (●" Fig.3). Upon removal of the biliary cast, hemobilia was observed, which was successfully treated by inserting a fully covered self-expanding metal stent (fcSEMS; Wallstent 10mm diameter×80mm length, Boston Scientific, Massachusetts, United States) (●" Fig.4). The length of the stent was chosen based on the diffuse characteristics of the portal biliopathy. Two-days later, a direct cholangioscopy (DC) using Spyglass (Boston Scientific, Massachusetts, United States) for biliary cleansing was performed. During the DC, the inner mucosal lining of the bile duct was observed, demonstrating a typical snake-skin mucosal pattern consistent with portal hypertensive biliopathy (●" Fig.5, ●" Video 1). The histology showed a biliary cast (●" Fig.3c). The clinical condition stabilized and the patient was discharged home one-week later. The stent was removed uneventfully four-weeks later. At the four-month follow-up, he did not have any further bleeding.

Biliary cast syndrome in portal hypertensive biliopathy: direct cholangioscopic findings and endoscopic therapy with metal stent