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托法替布治疗合并乳糜泻的难治性成人斯蒂尔病:一例报告

Tofacitinib in the Treatment of Refractory Adult-Onset Still's Disease Co-diagnosed With Celiac Disease: A Case Report.

作者信息

Alhayali Mustafa

机构信息

Internal Medicine/Rheumatology, Ibn Sina University of Medical and Pharmaceutical Sciences, Baghdad, IRQ.

Rheumatology and Medical Rehabilitation, Center of Spine and Joint Diseases, Baghdad, IRQ.

出版信息

Cureus. 2025 May 25;17(5):e84770. doi: 10.7759/cureus.84770. eCollection 2025 May.

Abstract

Adult-onset Still's disease (AOSD) is an autoinflammatory disease characterized by systemic and musculoskeletal manifestations driven by dysregulated cytokine activity. Fevers, evanescent salmon-colored rash, and transaminitis are hallmark features of the systemic subset of AOSD. Macrophage activation syndrome (MAS) represents the most feared and potentially life-threatening complication of AOSD. Tofacitinib, a Janus kinase (JAK) inhibitor, is approved for the management of several autoimmune conditions. We present a case of a 31-year-old woman with AOSD co-diagnosed with celiac disease, who was refractory to high-dose corticosteroids and tocilizumab. Initiation of tofacitinib led to marked clinical and laboratory improvement, enabling complete cessation of prednisolone. This case contributes to the limited body of literature supporting the use of tofacitinib in refractory AOSD and represents only the second reported instance of overlap between AOSD and celiac disease.

摘要

成人斯蒂尔病(AOSD)是一种自身炎症性疾病,其特征为细胞因子活性失调导致的全身和肌肉骨骼表现。发热、一过性鲑鱼色皮疹和转氨酶升高是AOSD全身型的标志性特征。巨噬细胞活化综合征(MAS)是AOSD最可怕且可能危及生命的并发症。托法替布,一种Janus激酶(JAK)抑制剂,已被批准用于治疗多种自身免疫性疾病。我们报告一例31岁女性,同时诊断为AOSD和乳糜泻,对高剂量糖皮质激素和托珠单抗治疗无效。使用托法替布后临床和实验室指标显著改善,得以完全停用泼尼松龙。该病例为支持托法替布用于难治性AOSD的有限文献增添了内容,且是AOSD与乳糜泻重叠的第二例报道。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/350a/12186570/543098cbc921/cureus-0017-00000084770-i01.jpg

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