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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

Case Report: Vanishing bile duct syndrome in Hodgkin's lymphoma: a case highlighting jaundice and lymphadenopathy as early clues.

作者信息

Ma Shiyu, Cai Dali, Miao Yuan, Deng Baocheng, Yan Xiaojing, Gao Ran

机构信息

Department of Hematology, The First Hospital of China Medical University, Shenyang, Liaoning, China.

Department of Pathology, The First Hospital of China Medical University, Shenyang, Liaoning, China.

出版信息

Front Immunol. 2025 May 21;16:1561110. doi: 10.3389/fimmu.2025.1561110. eCollection 2025.

DOI:10.3389/fimmu.2025.1561110
PMID:40469301
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12133851/
Abstract

Vanishing bile duct syndrome is a specific pathologic process characterized by ductopenia and intrahepatic cholestasis, which may be a unique paraneoplastic syndrome of Hodgkin's lymphoma with an unfavorable prognosis. We report a 34-year-old woman with acute jaundice and lymphadenopathy, which was subsequently confirmed to be Hodgkin's lymphoma with concurrent vanishing bile duct syndrome based on a liver biopsy. The patient agreed to combination chemotherapy with brentuximab vedotin and achieved a complete response. Liver function recovered within 4 months. This article reviews the literature and provides insight for addressing similar clinical challenges.

摘要

胆管消失综合征是一种以胆管减少和肝内胆汁淤积为特征的特定病理过程，可能是霍奇金淋巴瘤的一种独特的副肿瘤综合征，预后不良。我们报告一名34岁女性，出现急性黄疸和淋巴结病，随后经肝活检证实为霍奇金淋巴瘤并发胆管消失综合征。患者同意接受苯达莫司汀与维布妥昔单抗联合化疗并获得完全缓解。肝功能在4个月内恢复。本文回顾了相关文献，并为应对类似临床挑战提供了见解。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6077/12133851/da2f8ea82b0d/fimmu-16-1561110-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6077/12133851/279aaec908f7/fimmu-16-1561110-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6077/12133851/d26165920eee/fimmu-16-1561110-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6077/12133851/da2f8ea82b0d/fimmu-16-1561110-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6077/12133851/279aaec908f7/fimmu-16-1561110-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6077/12133851/d26165920eee/fimmu-16-1561110-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6077/12133851/da2f8ea82b0d/fimmu-16-1561110-g003.jpg

相似文献

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
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。
Turk J Gastroenterol. 2013;24(5):444-6.
3
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
World J Gastroenterol. 2017 Jan 14;23(2):366-372. doi: 10.3748/wjg.v23.i2.366.
4
Severe jaundice, due to vanishing bile duct syndrome, as presenting symptom of Hodgkin's lymphoma, fully reversible after chemotherapy.严重黄疸，由胆管消失综合征引起，作为霍奇金淋巴瘤的首发症状，化疗后完全可逆。
Eur J Gastroenterol Hepatol. 2008 Feb;20(2):145-7. doi: 10.1097/MEG.0b013e3282b9e6c2.
5
Complete remission of paraneoplastic vanishing bile duct syndrome after the successful treatment of Hodgkin's lymphoma: a case report and review of the literature.霍奇金淋巴瘤成功治疗后副肿瘤性胆管消失综合征完全缓解：一例报告并文献复习
BMC Res Notes. 2014 Aug 14;7:529. doi: 10.1186/1756-0500-7-529.
6
Vanishing bile duct syndrome as a manifestation of Hodgkin's lymphoma: a case report and review of the literature.霍奇金淋巴瘤表现为胆管消失综合征：一例报告并文献复习
Tumori. 2013 Jul-Aug;99(4):e164-8. doi: 10.1177/030089161309900426.
7
Paraneoplastic Intrahepatic Cholestasis in Supradiaphragmatic Classical Hodgkin Lymphoma Successfully Treated With Brentuximab Vedotin: A Case Report and Review of the Literature.膈上经典型霍奇金淋巴瘤伴副肿瘤性肝内胆汁淤积经 Brentuximab Vedotin 成功治疗：病例报告及文献复习。
In Vivo. 2021 Jul-Aug;35(4):1951-1957. doi: 10.21873/invivo.12462.
8
Tumour-directed radiotherapy as a successful bridge to curative chemotherapy in early stage Hodgkin's lymphoma-associated vanishing bile duct syndrome.肿瘤定向放疗作为早期霍奇金淋巴瘤相关消失性胆管综合征治愈性化疗的有效桥梁。
BMJ Case Rep. 2024 Sep 24;17(9):e261879. doi: 10.1136/bcr-2024-261879.
9
Vanishing bile duct syndrome: a possible mechanism for intrahepatic cholestasis in Hodgkin's lymphoma.消失胆管综合征：霍奇金淋巴瘤肝内胆汁淤积的一种可能机制。
Hepatology. 1993 Jan;17(1):70-7.
10
Cholestatic liver disease with ductopenia (vanishing bile duct syndrome) in Hodgkin's disease: report of a case.霍奇金病伴胆管减少（消失性胆管综合征）的胆汁淤积性肝病：病例报告
Tumori. 2004 Sep-Oct;90(5):517-20. doi: 10.1177/030089160409000516.

本文引用的文献

1
Vanishing Bile Duct Syndrome in the Presence of Hodgkin Lymphoma.霍奇金淋巴瘤患者中的胆管消失综合征
Cureus. 2022 Jul 14;14(7):e26842. doi: 10.7759/cureus.26842. eCollection 2022 Jul.
2
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.
3
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.
4
Vanishing Bile Duct Syndrome Associated With Non-Hodgkin's Lymphoma and Hepatitis E Virus Infection.与非霍奇金淋巴瘤和戊型肝炎病毒感染相关的胆管消失综合征
Cureus. 2022 Jan 17;14(1):e21328. doi: 10.7759/cureus.21328. eCollection 2022 Jan.
5
Paraneoplastic Intrahepatic Cholestasis in Supradiaphragmatic Classical Hodgkin Lymphoma Successfully Treated With Brentuximab Vedotin: A Case Report and Review of the Literature.膈上经典型霍奇金淋巴瘤伴副肿瘤性肝内胆汁淤积经 Brentuximab Vedotin 成功治疗：病例报告及文献复习。
In Vivo. 2021 Jul-Aug;35(4):1951-1957. doi: 10.21873/invivo.12462.
6
Hodgkin Lymphoma-related Vanishing Bile Duct Syndrome Cholestasis Resolved After Chemotherapy.霍奇金淋巴瘤相关的消失性胆管综合征化疗后胆汁淤积缓解。
J Pediatr Hematol Oncol. 2022 Apr 1;44(3):e728-e732. doi: 10.1097/MPH.0000000000002223.
7
[Hodgkin’s lymphoma-related vanishing bile duct syndrome].[霍奇金淋巴瘤相关的胆管消失综合征]
Orv Hetil. 2021 May 30;162(22):884-888. doi: 10.1556/650.2021.32093.
8
Vanishing Bile Duct Syndrome Preceding the Diagnosis of Hodgkin Lymphoma.霍奇金淋巴瘤诊断前的胆管消失综合征
ACG Case Rep J. 2020 Mar 2;7(2):e00336. doi: 10.14309/crj.0000000000000336. eCollection 2020 Feb.
9
Vanishing bile duct syndrome related to DILI and Hodgkin lymphoma overlap: A rare and severe case.与 DILI 和霍奇金淋巴瘤重叠相关的消失胆管综合征：一例罕见且严重的病例。
Ann Hepatol. 2020 Jan-Feb;19(1):107-112. doi: 10.1016/j.aohep.2019.06.010. Epub 2019 Aug 19.
10
Brentuximab vedotin in combination with sequential procarbazine, cyclophosphamide and prednisolone for the management of Hodgkin's lymphoma-associated vanishing bile duct syndrome (VBDS) with severe obstructive liver failure.本妥昔单抗联合序贯使用丙卡巴肼、环磷酰胺和泼尼松龙治疗霍奇金淋巴瘤相关的伴有严重梗阻性肝衰竭的消失性胆管综合征（VBDS）。
BMJ Case Rep. 2019 Feb 21;12(2):e227676. doi: 10.1136/bcr-2018-227676.