Pediatric Haematology Oncology Unit Department of Pediatrics.
Department of Pathology.
J Pediatr Hematol Oncol. 2022 Aug 1;44(6):e945-e947. doi: 10.1097/MPH.0000000000002505. Epub 2022 Jul 6.
Vanishing bile duct syndrome (VBDS) is a condition resulting from progressive destruction and loss of intrahepatic bile ducts leading to cholestasis, biliary cirrhosis, and liver failure. It occurs secondary to various pathologic conditions like autoimmune diseases, graft versus host disease, drug reactions, and as a paraneoplastic syndrome in malignancies. We here described a 9-year-old girl who presented with cervical lymphadenopathy and jaundice. This child was diagnosed as a case of Hodgkin lymphoma. All other causes of cholestasis were ruled out by appropriate investigations (particularly autoimmune, metabolic, infections, and drug-induced possibilities). On liver biopsy, her diagnosis was established as VBDS. In view of hepatic dysfunction, alternative chemotherapy with dexamethasone, high-dose cytarabine, and cisplatin (DHAP) was given, and she was started on hepatoprotective measures with ursodeoxycholic acid. Hepatic function gradually improved after the initiation of chemotherapy. VBDS is considered a dismal paraneoplastic syndrome with a high-case fatality. This case report highlights the importance of early recognition and initiation of appropriate full-dose chemotherapy as the only way to achieve complete resolution of VBDS.
进行性肝内胆管消失综合征(VBDS)是一种由肝内胆管进行性破坏和丧失导致胆汁淤积、胆汁性肝硬化和肝功能衰竭的疾病。它继发于各种病理情况,如自身免疫性疾病、移植物抗宿主病、药物反应,以及恶性肿瘤中的副肿瘤综合征。我们在此描述了一名 9 岁女孩,她因颈部淋巴结病和黄疸就诊。该患儿被诊断为霍奇金淋巴瘤。通过适当的检查排除了所有其他原因引起的胆汁淤积(特别是自身免疫、代谢、感染和药物诱导的可能性)。在肝活检中,她的诊断为 VBDS。鉴于肝功能障碍,给予替代化疗方案,即地塞米松、高剂量阿糖胞苷和顺铂(DHAP),并开始使用熊去氧胆酸进行保肝治疗。化疗开始后,肝功能逐渐改善。VBDS 被认为是一种预后不良的副肿瘤综合征,病死率很高。本病例报告强调了早期识别和启动适当的全剂量化疗的重要性,这是唯一能够完全缓解 VBDS 的方法。