Biliary papillomatosis with malignant transformation: A case report and review of the literature

Abstract

Biliary papillomatosis is a rare benign tumor that possesses a high potential for malignant transformation, and is frequently misdiagnosed. In the present report, a case of biliary papillomatosis with malignant transformation in a 63-year-old female, with a 19-year history of cholelithiasis, is presented. The patient presented with right epigastric pain and magnetic resonance imaging revealed a diffuse mass in the right liver lobe, which extended through the common bile duct. Definitive preoperative diagnosis was unable to be established; however, following surgical resection of the tumor, a diagnosis of biliary papillomatosis with malignant transformation was confirmed by pathological examination. The patient was followed up seven months later and no recurrence was detected. A supplementary review of previously published literature is additionally presented.

Keywords: biliary papillomatosis; intraductal papillary neoplasm of the bile duct; malignant; tumor.

Figure 1.

MRI demonstrates a diffuse and complex solid neoplasm, extending from the right intrahepatic bile duct into the common bile duct. (A) Contrast T1-weighted MRI revealed a hypointense lesion, and (B) contrast T2-weighted MRI revealed a hyperintense lesion. MRI, magnetic resonance imaging.

Figure 2.

Evaluation of the resected tumor. (A) The resected specimen comprised the right lobe of the liver and measured 15×13×7 cm. The bile ducts were distinctly dilated. (B) The common hepatic duct and bile ducts of the liver were encased by a diffuse villous tumor.

Figure 3.

Microscopically, tumor cells were observed to be papillary hyperplasia, possessing cellular characteristics of high-grade dysplasia and marked nuclear pleomorphism (magnification, ×400).