{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T04:16:01Z","timestamp":1787026561214,"version":"build-2736575974"},"reference-count":34,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,7,10]],"date-time":"2025-07-10T00:00:00Z","timestamp":1752105600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Immunol."],"abstract":"<jats:p>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.<\/jats:p>","DOI":"10.3389\/fimmu.2025.1628279","type":"journal-article","created":{"date-parts":[[2025,7,10]],"date-time":"2025-07-10T04:12:20Z","timestamp":1752120740000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":5,"title":["Case Report: Successful treatment of refractory synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome and palmoplantar pustulosis with ustekinumab"],"prefix":"10.3389","volume":"16","author":[{"given":"Luxia","family":"Chen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qichang","family":"Liang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sijia","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hao","family":"Cheng","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2025,7,10]]},"reference":[{"key":"B1","doi-asserted-by":"crossref","first-page":"1366","DOI":"10.3390\/jcm14041366","article-title":"What is new and what is next for SAPHO syndrome management: A narrative review","volume":"14","author":"Ferraioli","year":"2025","journal-title":"J Clin Med"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.3899\/jrheum.181393","article-title":"Copy number variation of multiple genes in SAPHO syndrome","volume":"47","author":"Guo","year":"2020","journal-title":"J Rheumatol"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2024.1427784\/full","article-title":"The role of cytokines in the pathogenesis of SAPHO syndrome","volume":"15","author":"Yang","year":"2024","journal-title":"Front Immunol"},{"key":"B4","doi-asserted-by":"publisher","DOI":"10.3899\/jrheum.090456","article-title":"Genetic susceptibility factors in a cohort of 38 patients with SAPHO syndrome: a study of PSTPIP2, NOD2, and LPIN2 genes","volume":"37","author":"Hurtado-Nedelec","year":"2010","journal-title":"J Rheumatol"},{"key":"B5","doi-asserted-by":"publisher","first-page":"1759720X20912865","DOI":"10.1177\/1759720X20912865","article-title":"Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: review and update","volume":"12","author":"Liu","year":"2020","journal-title":"Ther Adv Musculoskelet Dis"},{"key":"B6","doi-asserted-by":"crossref","DOI":"10.1016\/j.semarthrit.2018.04.003","article-title":"Biologics in SAPHO syndrome: A systematic review","volume":"48","author":"Daoussis","year":"2019","journal-title":"Semin Arthritis Rheum"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.1007\/s00296-023-05491-3","article-title":"SAPHO syndrome: current clinical, diagnostic and treatment approaches","volume":"44","author":"Demirci Yildirim","year":"2024","journal-title":"Rheumatol Int"},{"key":"B8","doi-asserted-by":"crossref","DOI":"10.1016\/S0950-3579(94)80022-7","article-title":"The SAPHO syndrome","volume":"8","author":"Kahn","year":"1994","journal-title":"Bailli\u00e8res Clin Rheumatol"},{"key":"B9","doi-asserted-by":"crossref","DOI":"10.1016\/S0049-0172(99)80027-4","article-title":"SAPHO syndrome: A long-term follow-up study of 120 cases","volume":"29","author":"Hayem","year":"1999","journal-title":"Semin Arthritis Rheum"},{"key":"B10","doi-asserted-by":"publisher","first-page":"38","DOI":"10.1111\/jdv.13846","article-title":"What do we know about palmoplantar pustulosis","volume":"31","author":"Misiak-Galazka","year":"2017","journal-title":"J Eur Acad Dermatol Venereol JEADV"},{"key":"B11","doi-asserted-by":"crossref","first-page":"492","DOI":"10.1097\/BOR.0b013e32825f5492","article-title":"Autoinflammatory bone disorders","volume":"19","author":"Ferguson","year":"2007","journal-title":"Curr Opin Rheumatol"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa0807865","article-title":"An autoinflammatory disease with deficiency of the interleukin-1-receptor antagonist","volume":"360","author":"Aksentijevich","year":"2009","journal-title":"N Engl J Med"},{"key":"B13","doi-asserted-by":"publisher","DOI":"10.1111\/jdv.15849","article-title":"Successful treatment of co-existent SAPHO syndrome and hidradenitis suppurativa with adalimumab and methotrexate","volume":"33","author":"Genovese","year":"2019","journal-title":"J Eur Acad Dermatol Venereol"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.7759\/cureus.42791","article-title":"Paradoxical tumor necrosis factor-alpha (TNF-\u03b1) inhibitor-induced psoriasis: A systematic review of pathogenesis, clinical presentation, and treatment","volume":"15","author":"Chokshi","year":"2023","journal-title":"Cureus"},{"key":"B15","doi-asserted-by":"publisher","first-page":"53","DOI":"10.1007\/s10067-018-4083-5","article-title":"Paradoxical skin lesions induced by anti-TNF-\u03b1 agents in SAPHO syndrome","volume":"38","author":"Li","year":"2019","journal-title":"Clin Rheumatol"},{"key":"B16","doi-asserted-by":"publisher","DOI":"10.2147\/CCID.S454057","article-title":"Successful treatment of refractory synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome and paradoxical psoriasis with secukinumab: A case report","volume":"17","author":"Fan","year":"2024","journal-title":"Clin Cosmet Investig Dermatol"},{"key":"B17","doi-asserted-by":"crossref","first-page":"3394","DOI":"10.3390\/ijms20143394","article-title":"Th17 cells and the IL-23\/IL-17 axis in the pathogenesis of periodontitis and immune-mediated inflammatory diseases","volume":"20","author":"Bunte","year":"2019","journal-title":"Int J Mol Sci"},{"key":"B18","doi-asserted-by":"publisher","DOI":"10.1111\/1346-8138.15826","article-title":"Similarity and difference between palmoplantar pustulosis and pustular psoriasis","volume":"48","author":"Yamamoto","year":"2021","journal-title":"J Dermatol"},{"key":"B19","unstructured":"Anti-TNF antibody-induced psoriasiform skin lesions in patients with inflammatory bowel disease are characterised by interferon-\u03b3-expressing Th1 cells and IL-17A\/IL-22-expressing Th17 cells and respond to anti-IL-12\/IL-23 antibody treatment\n          \n          \n            \n              Tillack\n              C\n            \n            \n              Ehmann\n              LM\n            \n            \n              Friedrich\n              M\n            \n            \n              Laubender\n              RP\n            \n            \n              Papay\n              P\n            \n            \n              Vogelsang\n              H\n            \n          \n          23468464\n          \n          2014"},{"key":"B20","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2009-04-216085","article-title":"Evidence for a cross-talk between human neutrophils and Th17 cells","volume":"115","author":"Pelletier","year":"2010","journal-title":"Blood"},{"key":"B21","doi-asserted-by":"crossref","first-page":"114339","DOI":"10.1016\/j.intimp.2025.114339","article-title":"The safety assessment of ustekinumab on psoriasis and psoriatic arthritis: A real-world analysis based on the FDA adverse event reporting system database","volume":"151","author":"Zheng","year":"2025","journal-title":"Int Immunopharmacol"},{"key":"B22","doi-asserted-by":"publisher","first-page":"656","DOI":"10.3899\/jrheum.211155","article-title":"Off-label use of secukinumab: A potential therapeutic option for SAPHO syndrome","volume":"49","author":"Wang","year":"2022","journal-title":"J Rheumatol"},{"key":"B23","doi-asserted-by":"crossref","DOI":"10.1016\/j.rhum.2017.07.055","article-title":"Ciblage de l\u2019axe IL-23\/Th17 dans le traitement du syndrome SAPHO","volume":"85","author":"Wendling","year":"2018","journal-title":"Rev Rhum"},{"key":"B24","doi-asserted-by":"publisher","DOI":"10.2147\/JIR.S353539","article-title":"New insights in the treatment of SAPHO syndrome and medication recommendations","volume":"15","author":"Cheng","year":"2022","journal-title":"J Inflammation Res"},{"key":"B25","doi-asserted-by":"publisher","first-page":"35","DOI":"10.1007\/s11926-016-0583-y","article-title":"SAPHO syndrome: current developments and approaches to clinical treatment","volume":"18","author":"Firinu","year":"2016","journal-title":"Curr Rheumatol Rep"},{"key":"B26","unstructured":"SAPHO syndrome in crohn\u2019s disease successfully treated with ustekinumab: case report and review of the literature\n          \n          \n            \n              Caporuscio\n              S\n            \n            \n              Maggi\n              D\n            \n            \n              Aratari\n              A\n            \n            \n              Papi\n              C\n            \n            \n              Festa\n              S\n            \n          \n          \n          2023"},{"key":"B27","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2133.2010.09897.x","article-title":"Ustekinumab in the treatment of palmoplantar pustulosis","volume":"163","author":"Gerdes","year":"2010","journal-title":"Br J Dermatol"},{"key":"B28","article-title":"Rapid and sustained remission of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome with IL-23p19 antagonist (risankizumab)","volume":"14","author":"Flora","year":"2021","journal-title":"JAAD Case Rep"},{"key":"B29","doi-asserted-by":"publisher","DOI":"10.1155\/2024\/9076852","article-title":"Risankizumab efficacy in synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) remission: A case report on rheumatologic and dermatologic disease manifestations with literature review","volume":"2024","author":"Ferraioli","year":"2024","journal-title":"Case Rep Immunol"},{"key":"B30","doi-asserted-by":"publisher","DOI":"10.1111\/dth.14758","article-title":"SAPHO syndrome successful treated with tildrakizumab","volume":"34","author":"Licata","year":"2021","journal-title":"Dermatol Ther"},{"key":"B31","doi-asserted-by":"publisher","DOI":"10.1001\/jamadermatol.2019.1394","article-title":"Efficacy and safety of guselkumab in Japanese patients with palmoplantar pustulosis: A phase 3 randomized clinical trial","volume":"155","author":"Terui","year":"2019","journal-title":"JAMA Dermatol"},{"key":"B32","article-title":"Efficacy of tofacitinib in synovitis, acne, pustulosis, hyperostosis and osteitis syndrome: a pilot study with clinical and MRI evaluation","volume":"79","author":"Li","year":"2020","journal-title":"Ann Rheum Dis"},{"key":"B33","doi-asserted-by":"publisher","DOI":"10.1001\/jamadermatol.2020.3095","article-title":"Tofacitinib for the treatment of nail lesions and palmoplantar pustulosis in synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome","volume":"157","author":"Li","year":"2021","journal-title":"JAMA Dermatol"},{"key":"B34","doi-asserted-by":"publisher","DOI":"10.1111\/1756-185X.14628","article-title":"SAPHO syndrome with Takayasu arteritis successfully treated with tofacitinib","volume":"26","author":"Ru","year":"2023","journal-title":"Int J Rheum Dis"}],"container-title":["Frontiers in Immunology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fimmu.2025.1628279\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,7,10]],"date-time":"2025-07-10T04:12:24Z","timestamp":1752120744000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fimmu.2025.1628279\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,7,10]]},"references-count":34,"alternative-id":["10.3389\/fimmu.2025.1628279"],"URL":"https:\/\/doi.org\/10.3389\/fimmu.2025.1628279","relation":{},"ISSN":["1664-3224"],"issn-type":[{"value":"1664-3224","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,7,10]]},"article-number":"1628279"}}