Department of Surgery and Department of Pathology and Biodefense, Saga University Faculty of Medicine, Saga, Japan.
Hepatol Res. 2008 Jun;38(6):629-34. doi: 10.1111/j.1872-034X.2007.00314.x. Epub 2008 Jan 2.
Multicystic biliary hamartoma is a very rare hamartomatous nodule in the liver, which has recently been described as a new category of hepatic nodular lesion. We herein report the case of a 55-year-old man histopathologically diagnosed with this entity following surgery. A solitary multilocular lesion in the liver was pointed out by ultrasonography during a systemic examination for a positive HBs antigen. This nodule could not be definitively diagnosed by radiologic modalities, including computed tomography, magnetic resonance imaging and arteriography. The patient underwent a partial resection of the posterior segment of the liver. The nodule was a localized lesion which measured 5 x 3 cm at the widest point and displayed a honeycomb appearance. Histologically, it consisted of ductal structures, periductal glands, fibrous connective tissues containing blood vessels, and bile-like materials and xanthogranulomatous inflammation within some ducts. Liver parenchyma was not present in the nodule and the bile ducts were not dilated in the background liver. The ductal epithelium expressed biliary type cytokeratins (CK7 and 19) in immunohistochemical studies. These histopathological features were consistent with multicystic biliary hamartoma, and we discuss this rare case in detail in this report.
多囊性胆管错构瘤是肝脏中非常罕见的错构瘤性结节，最近被描述为一种新的肝结节病变类别。本文报告了 1 例 55 岁男性患者的病例，该患者在因 HBs 抗原阳性进行全身检查时超声检查发现肝脏有单发的多囊性病变。该结节的影像学检查（包括 CT、MRI 和动脉造影）均无法明确诊断。患者接受了肝后段的部分切除术。该结节为局限性病变，最宽处大小为 5 x 3 cm，呈蜂窝状外观。组织学上，由胆管结构、管周腺体、富含血管的纤维结缔组织和胆管内的胆汁样物质及黄肉芽肿性炎症组成。结节内无肝实质，背景肝内胆管无扩张。胆管上皮在免疫组织化学研究中表达胆管型细胞角蛋白（CK7 和 19）。这些组织病理学特征符合多囊性胆管错构瘤，本文详细讨论了这一罕见病例。
