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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.

DOI:10.7759/cureus.84770
PMID:40557016
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12186570/
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
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/350a/12186570/543098cbc921/cureus-0017-00000084770-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/350a/12186570/543098cbc921/cureus-0017-00000084770-i01.jpg

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本文引用的文献

1
EULAR/PReS recommendations for the diagnosis and management of Still's disease, comprising systemic juvenile idiopathic arthritis and adult-onset Still's disease.EULAR/PReS 关于斯蒂尔病(包括全身型幼年特发性关节炎和成人斯蒂尔病)的诊断和治疗建议。
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JAK inhibitors in difficult-to-treat adult-onset Still's disease and systemic-onset juvenile idiopathic arthritis.
JAK抑制剂在难治性成人斯蒂尔病和全身型幼年特发性关节炎中的应用
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Clinical Phenotypes of Adult-Onset Still's Disease: New Insights from Pathophysiology and Literature Findings.成人斯蒂尔病的临床表型:来自病理生理学和文献研究结果的新见解
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[Successful treatment of adult Still's disease with tofacitinib in a HIV-2 positive female patient].[托法替布成功治疗一名HIV-2阳性成年女性患者的成人斯蒂尔病]
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Tofacitinib in refractory adult-onset Still's disease: 14 cases from a single centre in China.托法替布治疗难治性成人斯蒂尔病:来自中国单中心的14例病例
Ann Rheum Dis. 2020 Jun;79(6):842-844. doi: 10.1136/annrheumdis-2019-216699. Epub 2020 Feb 20.
8
Racial/Ethnic variations in morbidity and mortality in Adult Onset Still's Disease: An analysis of national dataset.成人Still 病发病率和死亡率的种族/民族差异:国家数据集分析。
Semin Arthritis Rheum. 2019 Dec;49(3):469-473. doi: 10.1016/j.semarthrit.2019.04.004. Epub 2019 Apr 25.
9
Long-Term Outcomes in Juvenile Idiopathic Arthritis: Eighteen Years of Follow-Up in the Population-Based Nordic Juvenile Idiopathic Arthritis Cohort.青少年特发性关节炎的长期结局:基于人群的北欧青少年特发性关节炎队列的 18 年随访。
Arthritis Care Res (Hoboken). 2020 Apr;72(4):507-516. doi: 10.1002/acr.23853.
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A JAK1 Selective Kinase Inhibitor and Tofacitinib Affect Macrophage Activation and Function.JAK1 选择性激酶抑制剂和托法替尼对巨噬细胞激活和功能的影响。
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