AUTHOR=Mu Wentao , Su Peng , Ning Shanglei TITLE=Case Report: Incidentally Discovered a Rare Cystic Lesion of Liver: Multicystic Biliary Hamartoma JOURNAL=Pathology and Oncology Research VOLUME=Volume 27 - 2021 YEAR=2021 URL=https://www.por-journal.com/journals/pathology-and-oncology-research/articles/10.3389/pore.2021.628323 DOI=10.3389/pore.2021.628323 ISSN=1532-2807 ABSTRACT=Multicystic biliary hamartoma (MCBH) is an extremely rare cystic lesion of the liver. A 37-year old male patient was admitted to our hospital for incidentally discovered hepatic cystic lesions during routine physical examination. Abdominal contrast-enhanced computed tomography (CT) showed a multilocular cystic lesion in the segment VI, with mild enhancement in the septa and peripheral wall within the lesion. Only alanine transaminase (ALT) and carbohydrate antigen 19–9 (CA19–9) increased slightly above normal value. We suspected that the diagnosis was benign mucinous cystic neoplasm (MCN) or intraductal papillary neoplasm of the bile duct (IPNB) before operation. Then we performed compete resection of the lesion laparoscopically. Further pathological findings revealed many ductal structures with different size surrounded with periductal glands and fibrous connective tissues containing small blood vessels and smooth muscle bundles. Immunohistochemical staining (IHC) revealed that dilated ducts was positive for biliary-type cytokeratin CK19. Finally, pathologists made the diagnosis of multicystic biliary hamartoma (MCBH). There was no signs of recurrence 6 months after surgery. In conclusion, MCBH should be differentiating from hepatic cystic lesion and could be resected laparoscopically safely.