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一名患有儿童期起病的自身免疫性肝炎的27岁女性的暴发性原发性胆汁性胆管炎-自身免疫性肝炎(PBC-AIH)重叠综合征:类固醇难治性失代偿,需要紧急进行移植评估

Fulminant Primary Biliary Cholangitis-Autoimmune Hepatitis (PBC-AIH) Overlap Syndrome in a 27-Year-Old Woman With Childhood-Onset AIH: Steroid-Refractory Decompensation Necessitating Urgent Transplant Evaluation.

作者信息

Said Ebram, Tafesse Yordanos, Elbenawi Hossam R, Chalise Shyam, Atia George

机构信息

Internal Medicine, Ascension Saint Joseph Hospital, Chicago, USA.

Internal Medicine, Mansoura University School of Medicine, Mansoura, EGY.

出版信息

Cureus. 2025 Jul 8;17(7):e87519. doi: 10.7759/cureus.87519. eCollection 2025 Jul.

Abstract

Autoimmune hepatitis (AIH) and primary biliary cholangitis (PBC) represent distinct autoimmune liver diseases, each with characteristic clinical, serological, and histological features. Rarely, patients may exhibit an overlap syndrome, presenting diagnostic and therapeutic challenges. We describe a 27-year-old woman with a longstanding history of childhood-onset AIH who subsequently developed clinical and serologic features consistent with PBC, fulfilling the Paris criteria for AIH-PBC overlap syndrome. Despite aggressive treatment with high-dose corticosteroids, she experienced rapid clinical deterioration, developing acute hepatic encephalopathy and requiring intensive care unit admission. A liver biopsy confirmed the coexistence of interface hepatitis (typical of AIH) and florid bile duct lesions (characteristic of PBC). Notably, the patient had a steroid-refractory course, systemic autoimmune comorbidities including ulcerative colitis and pyoderma gangrenosum, and exhibited atypical early-onset disease progression. Ultimately, due to severe and refractory liver failure, she required urgent liver transplantation evaluation. This case highlights the aggressive clinical course and critical management complexities of AIH-PBC overlap syndrome, emphasizing the importance of early recognition, comprehensive histologic evaluation, combined immunosuppressive and cholestatic therapies, and expedited referral for transplantation in refractory cases.

摘要

自身免疫性肝炎(AIH)和原发性胆汁性胆管炎(PBC)是两种不同的自身免疫性肝病,各有其独特的临床、血清学和组织学特征。患者很少会出现重叠综合征,这给诊断和治疗带来了挑战。我们报告一例27岁女性,自幼起病,长期患有AIH,随后出现了符合PBC的临床和血清学特征,满足AIH-PBC重叠综合征的巴黎标准。尽管使用高剂量皮质类固醇进行了积极治疗,但她的临床病情仍迅速恶化,发展为急性肝性脑病,需要入住重症监护病房。肝活检证实存在界面性肝炎(AIH的典型表现)和明显的胆管病变(PBC的特征性表现)。值得注意的是,该患者对类固醇治疗无效,伴有包括溃疡性结肠炎和坏疽性脓皮病在内的全身性自身免疫性合并症,且疾病进展具有非典型的早发性特点。最终,由于严重的难治性肝衰竭,她需要紧急进行肝移植评估。该病例突出了AIH-PBC重叠综合征的侵袭性临床病程和关键管理复杂性,强调了早期识别、全面组织学评估、联合免疫抑制和胆汁淤积治疗以及在难治性病例中加快转诊进行移植的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0406/12332042/c720445ce026/cureus-0017-00000087519-i01.jpg

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