BACKGROUND

Cystadenomas are uncommon benign cystic neoplasms of the biliary system, of unknown aetiology and account for approximately 5 % of all the hepatobiliary cystic masses. 1 Most cystadenomas arise within the intrahepatic bile ducts. 2 3 Very rarely these lesions are found in the extrahe-patic biliary system and gallbladder. 4 Women are more commonly affected with a mean age at presentation of 45 years. 4 Preoperative diagnosis is chal-lenging, since the radiologic features are non-specifi c. We present a rare case of biliary cystadenoma with both intra and extrahepatic bile duct involvement. The literature on the radiologic and pathologic features of biliary cysta-denomas, as well as approach to management, will be discussed. CASE PRESENTATION A 62-year-old woman with a remote history of breast cancer and hypothyroidism presented to her primary care physician with 1 week history of dysuria and hyperpig-mentation of her urine. Her physical examination was within the normal limits except for sclero-icterus. Thyroid replacement hormone therapy was the only medication she was on and her family and social history were non-contributory. INVESTIGATIONS Her physical examination was unremarkable. Liver func-tion tests were abnormal, with an alkaline phosphatase o