Development of bile duct cancer in a 26-year-old man after resection of infantile choledochal cyst

Development of bile duct cancer in a 26-year-old man after resection of infantile choledochal cyst
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DOI:
10.1016/j.jpedsurg.2008.01.073
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发表时间:
2008-06-01
影响因子:
2.4
通讯作者:
Iwai, Naomi
Iwai, Naomi
中科院分区:
医学3区
文献类型:
--
作者:
Ono, Shigeru;Sakai, Kohei;Iwai, Naomi

文献摘要

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胰胆管的异常排列被认为是胆道恶性肿瘤的高危因素。我们报告一位26岁男性,在5个月大时因胆总管囊肿全切除合并胰胆管排列异常而发生肝内胆管癌。他在胆总管囊肿全切除术后一直表现良好;然而,在26岁时,他突然出现右上腹部的尖状发热和腹痛。电脑断层扫描及经皮经肝胆管镜检查发现肝管及左肝内胆管有多处结石及狭窄。从狭窄部位获得的活检标本的组织学检查显示肝内胆管腺癌。行左肝叶切除术,右肝管空肠吻合重建,术后病程顺利。然而,手术后一年,他死于癌性腹膜炎和胆管癌复发。本报告提醒我们,即使在婴儿胆总管囊肿全切除术后,由于肝内胆管狭窄和反复胆管炎伴多发结石引起的胆汁淤积也是肝内胆管癌变的高危因素。(c)2008年爱思唯尔公司All rights reserved.
Anomalous arrangement of the pancreaticobiliary duct is considered to be a high-risk factor for biliary tract malignancy. We report a case of intrahepatic cholangiocarcinoma in a 26-year-old man after total resection of choledochal cyst with anomalous arrangement of the pancreaticobiliary duct at the age of 5 months. He had been doing well after total resection of the choledochal cyst; however, he suddenly presented with a spiky fever and abdominal pain in the right upper quadrant at the age of 26 years. Computed tomographic scan and percutaneous transhepatic cholangioscope revealed multiple stones and stenosis of the hepatic duct and the left intrahepatic bile duct. Histologic examination of a biopsy specimen obtained from the stenotic site showed adenocarcinoma of the intrahepatic bile duct. Left lobectomy with re-reconstruction by right hepaticojejunostomy was performed, and his postoperative course was uneventful. One year after the operation, however, he died of carcinomatous peritonitis with recurrence of cholangiocarcinoma. This report warns us that bile stasis owing to stenosis of the intrahepatic bile duct and repeated cholangitis with multiple stones are high-risk factors for carcinogenesis of the intrahepatic bile duct even after total resection of the infantile choledochal cyst. (c) 2008 Elsevier Inc. All rights reserved.