Department of Hepatobiliary Surgery, General Surgery, Qilu Hospital of Shandong University, Ji'nan, China.
Department of Pathology, Qilu Hospital of Shandong University, Ji'nan, China.
Pathol Oncol Res. 2021 Mar 30;27:628323. doi: 10.3389/pore.2021.628323. eCollection 2021.
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 on abdominal ultrasonography. Abdominal contrast-enhanced computed tomography (CT) showed a multilocular cystic lesion in the segment VI, with mild enhancement in the septae and peripheral wall within the lesion. Only alanine transaminase (ALT) and carbohydrate antigen 19-9 (CA19-9) increased slightly above normal value. Preoperative tests suggested possibility of a benign mucinous cystic neoplasm (MCN) or intraductal papillary neoplasm of the bile duct (IPNB). Laparoscopic complete resection of the lesion was performed. Histopathological examination showed numerous variably sized ductal structures surrounded by periductal glands and fibrous connective tissues containing small blood vessels and smooth muscle bundles. Immunohistochemical staining (IHC) revealed that dilated ducts were positive for cytokeratin CK19, characteristic for biliary tract. Histopathological findings confirmed diagnosis of multicystic biliary hamartoma (MCBH). No recurrence occurred during 6 months follow-up. In conclusion, MCBH should be differentiating from hepatic cystic lesion and could be resected laparoscopically safely.
多囊性胆管错构瘤（MCBH）是一种极罕见的肝脏囊性病变。一名 37 岁男性患者因腹部超声偶然发现肝囊性病变而被收入我院。腹部增强 CT 显示 VI 段多房囊性病变，病变内分隔和外周壁轻度强化。仅丙氨酸转氨酶（ALT）和糖类抗原 19-9（CA19-9）略高于正常值。术前检查提示良性黏液性囊腺瘤（MCN）或胆管内乳头状肿瘤（IPNB）的可能性。行腹腔镜下病变完全切除术。组织病理学检查显示多个大小不一的导管结构，周围有导管周围腺体和纤维结缔组织，内含小血管和平滑肌束。免疫组织化学染色（IHC）显示扩张的导管 CK19 阳性，这是胆管的特征。组织病理学检查结果证实了多囊性胆管错构瘤（MCBH）的诊断。6 个月随访期间无复发。总之，MCBH 应与肝囊性病变相鉴别，并可安全行腹腔镜切除。
