Abstract

Intraductal papillary neoplasm of the bile duct (IPNB) is classified as a biliary cystic tumor with a tendency of causing obstruction. Neoplastic cases involving hemobilia are rarely reported. We herein describe a case of biliary cystic tumor with repeated hemobilia. A 57-year-old woman was histologically diagnosed with cavernous hemangioma. During the follow-up period after transcatheter arterial embolization (TAE), she experienced repeated hemobilia, and multiple other TAE sessions were performed for hemostasis. She was referred to our hospital 8 years after the first surgery owing to a growing tumor. Histopathological examination after extended right hepatectomy and caudate lobectomy indicated IPNB with an associated invasive carcinoma. Six months thereafter, computed tomography revealed a recurrent liver tumor and a nodule in the abdominal cavity. She died 36 months after the second surgery, despite chemotherapy. Our experience suggests that IPNB should be considered during differential diagnosis of dilated hepatobiliary tumors with hemobilia.

Highlights

  • Hemobilia is usually caused by trauma, iatrogenic events, cholangitis, and cholelithiasis, but rarely associated with a tumor [1]

  • intraductal papillary neoplasms of the bile duct (IPNB) is identified as a premalignant lesion or intraepithelial neoplasm of the bile duct, and it is relatively rare among all bile duct cancers

  • If hemorrhage occurs from an artery, transcatheter arterial embolization (TAE) is the first choice of treatment and often effective

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Summary

Background

Hemobilia is usually caused by trauma, iatrogenic events, cholangitis, and cholelithiasis, but rarely associated with a tumor [1]. She received transcatheter arterial embolization (TAE) for hemostasis of the right lobe tumor. During the follow-up period thereafter, she experienced six episodes of hemobilia, and four other TAE sessions were performed She was referred to our hospital 8 years after the diagnostic surgery owing to the growing tumor in the right lobe. The patient's medical history included an appendectomy at 9 years of age, Cesarean section at the age of 26 and 29 years, and right lower lobectomy with partial resection of the lungs' left upper lobe for adenocarcinomas at the age of 53 years She had no history of liver disease, including hepatolithiasis. Histological examination indicated the biliary cystic lesion was a well-differentiated adenocarcinoma (Figure 4). The final diagnosis was IPNB with an associated invasive carcinoma

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