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儿童霍奇金淋巴瘤继发胆汁淤积综合征。

Vanishing Bile Duct Syndrome Secondary to Hodgkin Lymphoma in a Child.

机构信息

Pediatric Haematology Oncology Unit Department of Pediatrics.

Department of Pathology.

出版信息

J Pediatr Hematol Oncol. 2022 Aug 1;44(6):e945-e947. doi: 10.1097/MPH.0000000000002505. Epub 2022 Jul 6.

DOI:10.1097/MPH.0000000000002505
PMID:35796591
Abstract

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.

摘要

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。

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Vanishing Bile Duct Syndrome Secondary to Hodgkin Lymphoma in a Child.儿童霍奇金淋巴瘤继发胆汁淤积综合征。
J Pediatr Hematol Oncol. 2022 Aug 1;44(6):e945-e947. doi: 10.1097/MPH.0000000000002505. Epub 2022 Jul 6.
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Vanishing bile duct syndrome in Hodgkin's lymphoma: A case report and literature review.霍奇金淋巴瘤中的胆管消失综合征:一例报告及文献复习
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Vanishing bile duct syndrome in a Hodgkin's lymphoma patient with fatal outcome despite lymphoma remission.霍奇金淋巴瘤患者发生消失性胆管综合征,尽管淋巴瘤缓解但仍导致死亡。
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Vanishing bile duct syndrome: a rare cause of jaundice in Hodgkin's lymphoma.胆管消失综合征:霍奇金淋巴瘤中黄疸的罕见病因。
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Administration of brentuximab vedotin to a Hodgkin lymphoma patient with liver dysfunction due to vanishing bile duct syndrome resulting in a partial response without any severe adverse events.因先天性肝内胆管缺如综合征导致肝功能障碍的霍奇金淋巴瘤患者接受 Brentuximab vedotin 治疗后获得部分缓解,且无严重不良事件。
J Clin Exp Hematop. 2022 Sep 28;62(3):154-157. doi: 10.3960/jslrt.21035. Epub 2022 Jul 12.

引用本文的文献

1
Case Report: Vanishing bile duct syndrome in Hodgkin's lymphoma: a case highlighting jaundice and lymphadenopathy as early clues.病例报告:霍奇金淋巴瘤中的胆管消失综合征:一例以黄疸和淋巴结病为早期线索的病例
Front Immunol. 2025 May 21;16:1561110. doi: 10.3389/fimmu.2025.1561110. eCollection 2025.
2
Review of Paraneoplastic Syndromes in Children with Malignancy.儿童恶性肿瘤副肿瘤综合征综述
Med Sci Monit. 2025 Mar 31;31:e947393. doi: 10.12659/MSM.947393.
3
Hodgkin lymphoma associated vanishing bile duct syndrome treated successfully with a brentuximab based regimen.
成功用博来霉素、依托泊苷、多柔比星、长春新碱和达卡巴嗪方案治疗霍奇金淋巴瘤相关的进行性肝内胆汁淤积症。
BMJ Case Rep. 2023 Nov 21;16(11):e257211. doi: 10.1136/bcr-2023-257211.
4
Non-invasive biomarkers for identification of vanishing bile duct syndrome among children with acute cholestatic hepatitis.用于识别急性胆汁淤积性肝炎患儿中消失性胆管综合征的非侵入性生物标志物。
Transl Pediatr. 2023 Oct 30;12(10):1782-1790. doi: 10.21037/tp-23-305. Epub 2023 Oct 27.