A rare cause of dilated bile duct incidentally detected on imaging

Abstract

Adenomas involving the extrahepatic bile ducts are rare benign tumours of the biliary tract. Given the asymptomatic nature of these tumours and the natural progression of biliary adenoma to carcinoma including cholangiocarcinoma, early detection is challenging. We present a case of an octogenarian woman with a remote history of gallstones who initially presented to the gastroenterology office for an incidental finding of biliary dilation, and whose subsequent imagings and biopsy suggested common bile adenoma. However, the patient was deemed a poor surgical candidate for pancreaticoduodenectomy, and unfortunately represented 5 months later with findings suggesting cholangiocarcinoma. This case provides a unique opportunity to study the natural progression of biliary adenoma to cholangiocarcinoma.

Adenomas involving the extrahepatic biliary tree are rare but have an aggressive clinical course

“…Three extrahepatic bile duct adenomas were diagnosed at our institution among a total of 10 559 bile duct pinch biopsies and surgical specimens (0.03 %) over 13 years. One of our cases has been reported previously 1 . On extensive review of the literature, we found another 36 cases making a total of 39 cases of extrahepatic biliary adenomas reported to date 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 ( Table1 and Fig.…”

“…There is little uniformity in the nomenclature applied to benign epithelial lesions and various classifications have been proposed. According to the WHO classification, they are divided into five different types: tubular, papillary (also known as papillomas), tubulopapillary, biliary cystadenoma, and papillomatosis 1 . Adenomas comprise two-thirds of benign biliary tumors 2 .…”

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“…Three extrahepatic bile duct adenomas were diagnosed at our institution among a total of 10 559 bile duct pinch biopsies and surgical specimens (0.03 %) over 13 years. One of our cases has been reported previously [1]. On extensive review of the literature, we found another 36 cases making a total of 39 cases of extrahepatic biliary adenomas reported to date [2 -28] (• " Table1 and…”

“…One of our cases presented with recurrent bacteremia in the setting of underlying primary sclerosing cholangitis. Two reported cases were asymptomatic with incidental findings of biliary dilation on imaging [1,8]. One case was found incidentally in a surgical resection specimen performed for duodenal adenocarcinoma [11].…”

“…Longterm follow-up (> 1 year) and short-term outcome (< 1 year) were reported in 8 [3,7,11,19] and 17 cases [4, 5, 8, 10, 11, 15 -17, 21, 24, 28], respectively. Five cases presented with interval malignancy including cholangiocarcinoma, and small-bowel adenocarcinoma was noted at follow-up [1,4,17]. The longest followup was reported to be 7 years with the patient still alive [4].…”