Cholangiocarcinoma in association with multiple biliary microhamartomas. The rarity of cholangiocarcinoma occurring in von Meyenburg complexes is well appreciated. We describe the case of a 35-year-old man found to have cholangiocarcinoma in association with multiple biliary microhamartomas of the liver. Areas of ductal proliferation and atypia within the bile duct hamartomas suggest malignant transformation to cholangiocarcinoma. 