Introduction Biliary cystadenoma is a rare, benign neoplasm of the bile ducts with malignant potential. Symptoms, predominantly right hypochondrial pain and the feeling of a lump or fullness are usually due to the mass effect. Jaundice is rare. This is the fifth reported patient with an intrahepatic biliary cystadenoma giving rise to episodic biliary obstruction, which is usually caused by choledocholithiasis or periampullary carcinoma. Considering the mean age of previous similar patients (53.5, standard deviation 14.6 years), the early age of presentation is very unusual in our patient. Case presentation A 25-year-old Asian woman presented with right hypochondrial pain and episodic biliary obstruction. Contrast enhanced computed tomography revealed a cystic mass in segment 4B and protruding into and along the left hepatic duct. Laparotomy confirmed the contrast enhanced computed tomography findings and histology revealed an intrahepatic mucinous biliary cystadenoma. Conclusion Biliary cystadenoma should be considered as a differential diagnosis in patients with cystic liver lesions who present with episodic biliary obstruction. Due to the reported malignant potential, radical surgery such as wide local excision of the lesion or hepatic resection is needed to minimize the risk of local recurrence.
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