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<p>Vanishing bile duct syndrome (VBDS) is a rare, and often fatal complication of Hodgkin lymphoma (HL), characterized by progressive intrahepatic bile duct loss and severe cholestasis. Management remains ill-defined, particularly in patients with refractory HL and significant hepatic dysfunction. We present a case of a young woman with biopsy-proven VBDS who experienced disease progression and worsening cholestasis despite second-line chemotherapy, corticosteroids, and brentuximab vedotin. Salvage therapy with pembrolizumab was initiated, resulting in a complete metabolic remission of HL and normalization of liver function. Notably, the patient did not experience immune-related hepatic adverse events. To our knowledge, this is the first report of HL-related VBDS successfully treated with programmed death-1 blockade. This case suggests that immune checkpoint inhibitors may be a viable therapeutic option for patients with HL-related VBDS, even in the setting of severe hepatic dysfunction.</p>

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Resolution of vanishing bile duct syndrome in a patient associated with refractory hodgkin lymphoma following Anti–PD-1 therapy: a case report and literature review

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2026年2月26日

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  • Yi-Ching Lin,
  • Wan-Chen Hsieh,
  • Yu-Hsuan Tuan,
  • Hsu-Hua Tseng,
  • Jia-Huei Tsai,
  • Tung-Hung Su,
  • Tai-Chung Huang

摘要

Vanishing bile duct syndrome (VBDS) is a rare, and often fatal complication of Hodgkin lymphoma (HL), characterized by progressive intrahepatic bile duct loss and severe cholestasis. Management remains ill-defined, particularly in patients with refractory HL and significant hepatic dysfunction. We present a case of a young woman with biopsy-proven VBDS who experienced disease progression and worsening cholestasis despite second-line chemotherapy, corticosteroids, and brentuximab vedotin. Salvage therapy with pembrolizumab was initiated, resulting in a complete metabolic remission of HL and normalization of liver function. Notably, the patient did not experience immune-related hepatic adverse events. To our knowledge, this is the first report of HL-related VBDS successfully treated with programmed death-1 blockade. This case suggests that immune checkpoint inhibitors may be a viable therapeutic option for patients with HL-related VBDS, even in the setting of severe hepatic dysfunction.