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病例报告:用优特克单抗成功治疗难治性滑膜炎、痤疮、脓疱病、骨肥厚和骨炎综合征及掌跖脓疱病。

Case Report: Successful treatment of refractory synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome and palmoplantar pustulosis with ustekinumab.

作者信息

Chen Luxia, Liang Qichang, Chen Sijia, Cheng Hao

机构信息

Department of Dermatology and Venereology, Sir Run Run Shaw Hospital, Zhejiang University School of Medicine, Hangzhou, China.

Department of Dermatology, Children's Hospital, Zhejiang University School of Medicine, Hangzhou, China.

出版信息

Front Immunol. 2025 Jul 10;16:1628279. doi: 10.3389/fimmu.2025.1628279. eCollection 2025.

DOI:10.3389/fimmu.2025.1628279
PMID:40709177
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12286785/
Abstract

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare chronic inflammatory disease mainly manifested as skin and osteoarticular lesions. We describe a male patient with SAPHO syndrome who exhibited primary palmoplantar pustulosis (PPP). Notably, his condition worsened during treatment with adalimumab and other immunosuppressants. After switching to ustekinumab, the patient obtained significant improvement in both skin lesions and osteoarticular pain. These findings suggest that ustekinumab may represent an appropriate option for patients with refractory SAPHO syndrome.

摘要

滑膜炎、痤疮、脓疱病、骨肥厚和骨炎(SAPHO)综合征是一种罕见的慢性炎症性疾病,主要表现为皮肤和骨关节病变。我们描述了一名患有SAPHO综合征的男性患者,他表现为原发性掌跖脓疱病(PPP)。值得注意的是,在使用阿达木单抗和其他免疫抑制剂治疗期间,他的病情恶化。改用优特克单抗后,患者的皮肤病变和骨关节疼痛均有显著改善。这些发现表明,优特克单抗可能是难治性SAPHO综合征患者的一个合适选择。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/553aa8fcb52c/fimmu-16-1628279-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/6e7ee3ac5a58/fimmu-16-1628279-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/5df58af1aad9/fimmu-16-1628279-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/75c9638fd300/fimmu-16-1628279-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/553aa8fcb52c/fimmu-16-1628279-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/6e7ee3ac5a58/fimmu-16-1628279-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/5df58af1aad9/fimmu-16-1628279-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/75c9638fd300/fimmu-16-1628279-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d5bd/12286785/553aa8fcb52c/fimmu-16-1628279-g004.jpg

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本文引用的文献

1
What Is New and What Is Next for SAPHO Syndrome Management: A Narrative Review.SAPHO综合征管理的新进展与未来方向:一篇叙述性综述
J Clin Med. 2025 Feb 18;14(4):1366. doi: 10.3390/jcm14041366.
2
The safety assessment of ustekinumab on psoriasis and psoriatic arthritis: A real-world analysis based on the FDA adverse event reporting system database.优特克单抗治疗银屑病和银屑病关节炎的安全性评估:基于美国食品药品监督管理局不良事件报告系统数据库的真实世界分析
Int Immunopharmacol. 2025 Apr 4;151:114339. doi: 10.1016/j.intimp.2025.114339. Epub 2025 Feb 22.
3
The role of cytokines in the pathogenesis of SAPHO syndrome.
细胞因子在 SAPHO 综合征发病机制中的作用。
Front Immunol. 2024 Sep 2;15:1427784. doi: 10.3389/fimmu.2024.1427784. eCollection 2024.
4
Risankizumab Efficacy in Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Remission: A Case Report on Rheumatologic and Dermatologic Disease Manifestations with Literature Review.司库奇尤单抗治疗滑膜炎、痤疮、脓疱病、骨肥厚和骨炎(SAPHO)缓解的疗效:1例关于风湿性和皮肤病表现的病例报告及文献综述
Case Reports Immunol. 2024 Mar 19;2024:9076852. doi: 10.1155/2024/9076852. eCollection 2024.
5
Successful Treatment of Refractory Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Syndrome and Paradoxical Psoriasis with Secukinumab: A Case Report.司库奇尤单抗成功治疗难治性滑膜炎、痤疮、脓疱病、骨肥厚和骨炎(SAPHO)综合征及反常性银屑病:一例报告
Clin Cosmet Investig Dermatol. 2024 Mar 5;17:547-552. doi: 10.2147/CCID.S454057. eCollection 2024.
6
SAPHO syndrome: current clinical, diagnostic and treatment approaches.SAPHO 综合征:当前的临床、诊断和治疗方法。
Rheumatol Int. 2024 Nov;44(11):2301-2313. doi: 10.1007/s00296-023-05491-3. Epub 2023 Oct 27.
7
Paradoxical Tumor Necrosis Factor-Alpha (TNF-α) Inhibitor-Induced Psoriasis: A Systematic Review of Pathogenesis, Clinical Presentation, and Treatment.矛盾性肿瘤坏死因子-α(TNF-α)抑制剂诱导的银屑病:发病机制、临床表现及治疗的系统评价
Cureus. 2023 Aug 1;15(8):e42791. doi: 10.7759/cureus.42791. eCollection 2023 Aug.
8
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J Inflamm Res. 2022 Apr 13;15:2365-2380. doi: 10.2147/JIR.S353539. eCollection 2022.
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Off-label Use of Secukinumab: A Potential Therapeutic Option for SAPHO Syndrome.司库奇尤单抗的超说明书用药:SAPHO综合征的一种潜在治疗选择
J Rheumatol. 2022 Jun;49(6):656. doi: 10.3899/jrheum.211155. Epub 2021 Dec 15.