Biliary
hamartomas are
tumor -like malformations of the
liver . Biliary
hamartomas are a type of fibrocystic disorder originating from ductal plate malformation and are typically considered benign, but with the
risk of malignant transformation. In this case
report , we present a rare occurrence of
intrahepatic cholangiocarcinoma (ICC) that developed from biliary
hamartomas , along with a
literature review . A 76-year-old man with a
diagnosis of biliary
hamartomas had a
history of recurrent
cholangitis for 12 years, necessitating
cholecystectomy ,
ERCP , and repeated
antibiotic treatments . During his last episode, imaging studies revealed a hypervascular infiltrative mass in the right posterior
liver segment. A
liver biopsy confirmed
adenocarcinoma and subsequent
surgical pathology revealed ICC originating from biliary
hamartomas . Chronic
inflammation in the
bile duct associated with biliary
hamartomas may serve as a potential trigger for malignant transformation, as observed in this case. Therefore, close
surveillance is essential for
patients with biliary
hamartomas presenting with infectious
complications .