CASE REPORT article

Front. Immunol.

Sec. Autoimmune and Autoinflammatory Disorders : Autoimmune Disorders

Volume 16 - 2025 | doi: 10.3389/fimmu.2025.1628279

Case Report: Successful Treatment of Refractory Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Syndrome and Palmoplantar Pustulosis with Ustekinumab

Provisionally accepted
Luxia  ChenLuxia Chen1Qichang  LiangQichang Liang2Sijia  ChenSijia Chen1Hao  ChengHao Cheng1*
  • 1Sir Run Run Shaw Hospital, School of Medicine, Graduate School, Zhejiang University, Hangzhou, China
  • 2Children's hospital, zhejiang university school of medicine, Hangzhou, China

The final, formatted version of the article will be published soon.

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.

Keywords: SAPHO syndrome, Palmoplantar pustulosis, Anti-IL-12/23 antibody, Ustekinumab, immune-mediated inflammatory diseases

Received: 14 May 2025; Accepted: 23 Jun 2025.

Copyright: © 2025 Chen, Liang, Chen and Cheng. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

* Correspondence: Hao Cheng, Sir Run Run Shaw Hospital, School of Medicine, Graduate School, Zhejiang University, Hangzhou, China

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