Korean J Gastroenterol.  2009 Jul;54(1):55-59. 10.4166/kjg.2009.54.1.55.

A Case of Early Bile Duct Cancer Arising from Villous Adenoma in Choledochal Cyst

• Lee TS
• Kim HK
• Ahn HM
• Lee UJ
• Choi YC
• John BM
• Park TI
• Koo JH

• KMID: 1792768
• DOI: http://doi.org/10.4166/kjg.2009.54.1.55

Abstract

Choledochal cyst is an uncommon premalignant anomaly. The morphology and pathogenesis of the premalignant lesion of cholangiocarcinoma arising from the choledochal cyst has not been well described. Herein, we report a rare case of bile duct adenoma arising from choledochal cyst with anomalous union of pancreaticobiliary duct (AUPBD). 50-year-old woman was admitted to our hospital with the complaint of epigastric pain. She had received common bile duct (CBD) exploration and choledocholithotomy and cholecystectomy 3 months earlier under the diagnosis of multiple CBD stones. Intraoperalive cholangiogram was not remarkable except CBD dilatation at that time. Endoscopic retrograde cholangiopancreatography revealed choledochal cyst with AUPBD and round filling defect which disappeared easily on the balloon cholaniogram. On magnetic resonance cholangiopancreatography, the filling defect was confirmed as 2 cm polypoid mass attached to the distal bile duct wall. At laparotomy, a soft whitish mass was palpable on the lower CBD. On histological examination, adenoma with focal carcinoma change arising from choledochal cyst was diagnosed.

Keyword

Choledochal cyst; Cholangiocarcinoma; Bile duct adenoma

MeSH Terms

Adenoma, Villous/*diagnosis/pathology/radiography
Bile Duct Neoplasms/*diagnosis/pathology/radiography
Cholangiopancreatography, Magnetic Resonance
Choledochal Cyst/*radiography/secretion/surgery
Female
Humans
Middle Aged
Tomography, X-Ray Computed

Figure

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  Fig. 1. Abdominal CT showed multiple bile duct stones with marked common bile duct dilatation.

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  Fig. 2. ERCP shows multiple filling defect of common bile duct and proximal duct dilatation. But, basket extraction and balloon sweeping retrieved nothing. Pancreatic duct was not cannulated.

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  Fig. 3. ERCP showed round filling defect which disappeared easily on the balloon occlusive cholangiogram (A) and revealed choledochal cyst with anomalous union of pancreaticobiliary duct (AUPBD) (B).

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  Fig. 4. MRCP showed fusiform dilatation of extrahepatic duct (A) and a soft tissue signal mass measured 2×2 cm within a choledochal cyst (B). Combined AUPBD with choledochal cyst was seen (C).

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  Fig. 5. (A) Microscopic findings of the specimen showed protruding polypoid lesion into lumen, consisting of tubulovillous adenoma (H&E stain, ×20). (B) Focal adenocarcinoma change arising from adenoma was lim-ited to bile duct mucosa (H&E stain, ×200).

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