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We report two cases of biliary papillomatosis, a rare disease which is characterized by multiple, papillary adenomas of the intrahepatic and/or extrahepatic bile ducts. Cases 1 and 2 were a 45-year-old woman and a 61-year-old woman, respectively, with upper abdominal pain. Abdominal computed tomography scan of case 1 revealed a 4-cm sized cystic and solid mass within the dilatated intrahepatic bile duct. Hepatobiliary ultrasonography of case 2 showed dilatation of the main intrahepatic and common bile duct with nonshadowing echogenic material. Soft, friable papillary masses were noted grossly in the dilated intrahepatic bile duct. Microscopically, the tumor was composed of papillary structures, most of which were lined by a layer of tall columnar cells. Complex papillary fronds with pseudostratification, cellular atypia, structural atypia and foci of invasion were noted in case 2. The p53 expression increased in the dysplastic and carcinomatous area and was correlated with the Ki-67 labeling index.


We report two cases of biliary papillomatosis, a rare disease which is characterized by multiple, papillary adenomas of the intrahepatic and/or extrahepatic bile ducts. Cases 1 and 2 were a 45-year-old woman and a 61-year-old woman, respectively, with upper abdominal pain. Abdominal computed tomography scan of case 1 revealed a 4-cm sized cystic and solid mass within the dilatated intrahepatic bile duct. Hepatobiliary ultrasonography of case 2 showed dilatation of the main intrahepatic and common bile duct with nonshadowing echogenic material. Soft, friable papillary masses were noted grossly in the dilated intrahepatic bile duct. Microscopically, the tumor was composed of papillary structures, most of which were lined by a layer of tall columnar cells. Complex papillary fronds with pseudostratification, cellular atypia, structural atypia and foci of invasion were noted in case 2. The p53 expression increased in the dysplastic and carcinomatous area and was correlated with the Ki-67 labeling index.


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Liver-Papillomatosis-Biliary tract-Adenocarcinoma-p53