{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T06:05:17Z","timestamp":1780380317782,"version":"3.54.1"},"reference-count":28,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T00:00:00Z","timestamp":1780358400000},"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 autoinflammatory disease characterized primarily by cutaneous manifestations and osteoarticular involvement, and remains challenging to treat. Its pathogenesis is not fully understood and may involve genetic, immunological and environmental factors. Environmental factors play a significant role in autoimmune diseases such as asthma, urticaria, and rheumatic heart disease. Avoiding repeated exposure to sensitizing environmental trigger (e.g., microorganism or allergens) shows considerable potential in the treatment of above immune mediated disorders. This article reports a case of a 60-year-old female admitted with recurrent chest pain for over two months accompanied by periodic plantar pustules. A diagnosis of SAPHO syndrome was made based on her medical history, clinical manifestations, and bull\u2019s head sign on bone scintigraphy findings, after excluding other potential causes. Notably, the patient\u2019s husband had ankylosing spondylitis, and neither the patient nor her husband had a family history of inherited immune diseases. This rare occurrence in cohabiting spouse suggests that they shared environmental exposures may contribute to the development of phenotypically overlapping immune diseases. Therefore, we advised her to try changing her residential environment (moving to a drier, better ventilated environment) and iodophor disinfection of soles. Subsequently, her plantar pustules and chest pain were significantly relieved and remained relapse free. We first reported a case of successful environmental modification as an adjunctive treatment for SAPHO syndrome, suggesting a potential role of environmental factors in pathogenesis and clinical management of this condition.<\/jats:p>","DOI":"10.3389\/fimmu.2026.1776452","type":"journal-article","created":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T05:42:44Z","timestamp":1780378964000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Case Report: Sapho syndrome complicated by chest pain successfully treated with relocation"],"prefix":"10.3389","volume":"17","author":[{"given":"Youjun","family":"Zhang","sequence":"first","affiliation":[{"name":"Department of Cardiology, Huadong Hospital, Fudan University","place":["Shanghai, China"]},{"name":"Shanghai Key Laboratory of Clinical Geriatric Medicine","place":["Shanghai, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hongjin","family":"Chen","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Huadong Hospital, Fudan University","place":["Shanghai, China"]},{"name":"Shanghai Key Laboratory of Clinical Geriatric Medicine","place":["Shanghai, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shaofeng","family":"Guan","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Huadong Hospital, Fudan University","place":["Shanghai, China"]},{"name":"Shanghai Key Laboratory of Clinical Geriatric Medicine","place":["Shanghai, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xinkai","family":"Qu","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Huadong Hospital, Fudan University","place":["Shanghai, China"]},{"name":"Shanghai Key Laboratory of Clinical Geriatric Medicine","place":["Shanghai, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2026,6,2]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","DOI":"10.1016\/j.berh.2011.01.017","article-title":"The SAPHO syndrome--are microbes involved","volume":"25","author":"Assmann","year":"2011","journal-title":"Best Pract Res Clin Rheumatol"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jinf.2016.04.030","article-title":"Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome - a challenging diagnosis not to be missed","volume":"72","author":"Zimmermann","year":"2016","journal-title":"J Infect"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.1684\/ejd.2016.2854","article-title":"Interstitial granulomatous dermatitis occurring in a patient with SAPHO syndrome one month after starting leflunomide, and subsequently disappearing with ustekinumab","volume":"26","author":"Cornillier","year":"2016","journal-title":"Eur J Dermatology: EJD"},{"key":"B4","doi-asserted-by":"publisher","DOI":"10.1016\/j.jbspin.2017.05.016","article-title":"IL-23\/Th17 targeted therapies in SAPHO syndrome. A case series","volume":"84","author":"Wendling","year":"2017","journal-title":"Joint Bone Spine"},{"key":"B5","doi-asserted-by":"publisher","DOI":"10.4049\/jimmunol.174.7.4295","article-title":"Requirement for TLR9 in the immunomodulatory activity of Propionibacterium acnes","volume":"174","author":"Kalis","year":"2005","journal-title":"J Immunol"},{"key":"B6","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2022.911922","article-title":"SAPHO syndrome complicated by ankylosing spondylitis successfully treated with tofacitinib: a case report","volume":"13","author":"Yuan","year":"2022","journal-title":"Front Immunol"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.1007\/s10067-020-05393-w","article-title":"Clinical characteristics of pediatric synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome: the first Chinese case series from a single center","volume":"40","author":"Wu","year":"2021","journal-title":"Clin Rheumatol"},{"key":"B8","doi-asserted-by":"publisher","DOI":"10.1093\/qjmed\/hcz125","article-title":"The SAPHO syndrome and the bull's head sign","volume":"113","author":"Liu","year":"2020","journal-title":"Qjm"},{"key":"B9","doi-asserted-by":"publisher","DOI":"10.1007\/s10067-010-1560-x","article-title":"The SAPHO syndrome: a clinical and imaging study","volume":"30","author":"Sall\u00e9s","year":"2011","journal-title":"Clin Rheumatol"},{"key":"B10","doi-asserted-by":"publisher","DOI":"10.1016\/j.jbspin.2006.06.006","article-title":"Valuable lessons from SAPHO syndrome","volume":"74","author":"Hayem","year":"2007","journal-title":"Joint Bone Spine"},{"key":"B11","doi-asserted-by":"publisher","first-page":"195","DOI":"10.1016\/j.crad.2011.08.014","article-title":"SAPHO: what radiologists should know","volume":"67","author":"Depasquale","year":"2011","journal-title":"Clin Radiol"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.7759\/cureus.71897","article-title":"Challenges in diagnosing SAPHO syndrome: a multidisciplinary perspective","volume":"16","author":"Zahid","year":"2024","journal-title":"Cureus"},{"key":"B13","doi-asserted-by":"publisher","DOI":"10.1097\/mnm.0000000000000451","article-title":"Is the bullhead sign on bone scintigraphy really common in the patient with SAPHO syndrome? A single-center study of a 16-year experience","volume":"37","author":"Fu","year":"2016","journal-title":"Nucl Med Commun"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2025.1683214","article-title":"Misdiagnosed with spinal infection instead of SAPHO syndrome: a case report and literature review","volume":"16","author":"Wang","year":"2025","journal-title":"Front Immunol"},{"key":"B15","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.immunol.25.022106.141627","article-title":"Horror autoinflammaticus: the molecular pathophysiology of autoinflammatory disease (*)","volume":"27","author":"Masters","year":"2009","journal-title":"Annu Rev Immunol"},{"key":"B16","doi-asserted-by":"publisher","DOI":"10.1016\/j.autrev.2008.07.030","article-title":"SAPHO syndrome and infections","volume":"8","author":"Govoni","year":"2008","journal-title":"Autoimmun Rev"},{"key":"B17","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2024.01.016","article-title":"Impact of climate change on immune responses and barrier defense","volume":"153","author":"Skevaki","year":"2024","journal-title":"J Allergy Clin Immunol"},{"key":"B18","doi-asserted-by":"publisher","DOI":"10.1038\/s41569-020-00435-x","article-title":"Myocarditis and inflammatory cardiomyopathy: current evidence and future directions","volume":"18","author":"Tsch\u00f6pe","year":"2020","journal-title":"Nat Rev Cardiol"},{"key":"B19","doi-asserted-by":"publisher","DOI":"10.1038\/s41569-019-0258-2","article-title":"Genetics of rheumatic fever and rheumatic heart disease","volume":"17","author":"Muhamed","year":"2019","journal-title":"Nat Rev Cardiol"},{"key":"B20","doi-asserted-by":"publisher","DOI":"10.1016\/j.jacc.2018.06.063","article-title":"Rheumatic heart disease worldwide: JACC scientific expert panel","volume":"72","author":"Watkins","year":"2018","journal-title":"J Am Coll Cardiol"},{"key":"B21","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2024.1429591","article-title":"Oleic acid attenuates asthma pathogenesis via Th1\/Th2 immune cell modulation, TLR3\/4-NF-\u03baB-related inflammation suppression, and intrinsic apoptotic pathway induction","volume":"15","author":"Lee","year":"2024","journal-title":"Front Immunol"},{"key":"B22","doi-asserted-by":"publisher","DOI":"10.1385\/criai:30:1:003","article-title":"Pathophysiology of urticaria","volume":"30","author":"Hennino","year":"2006","journal-title":"Clin Rev Allergy Immunol"},{"key":"B23","doi-asserted-by":"publisher","DOI":"10.1186\/ar2812","article-title":"Efficacy of antibiotic therapy for SAPHO syndrome is lost after its discontinuation: an interventional study","volume":"11","author":"Assmann","year":"2009","journal-title":"Arthritis Res Ther"},{"key":"B24","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2024.1427784","article-title":"The role of cytokines in the pathogenesis of SAPHO syndrome","volume":"15","author":"Yang","year":"2024","journal-title":"Front Immunol"},{"key":"B25","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/ken185","article-title":"Characterization of the immune response in the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome","volume":"47","author":"Hurtado-Nedelec","year":"2008","journal-title":"Rheumatol (Oxford)"},{"key":"B26","doi-asserted-by":"publisher","DOI":"10.1007\/s00296-009-1138-6","article-title":"Mucosal lesions may be a minor complication of SAPHO syndrome: a study of 11 Japanese patients with SAPHO syndrome","volume":"30","author":"Yabe","year":"2010","journal-title":"Rheumatol Int"},{"key":"B27","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2025.1560398","article-title":"Therapeutic approaches for SAPHO syndrome from the perspective of pathogenesis: a review of the literature","volume":"16","author":"Wang","year":"2025","journal-title":"Front Immunol"},{"key":"B28","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2025.1662675","article-title":"Upadacitinib in the treatment of SAPHO syndrome: a case report","volume":"16","author":"Yang","year":"2025","journal-title":"Front Immunol"}],"container-title":["Frontiers in Immunology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fimmu.2026.1776452\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T05:42:47Z","timestamp":1780378967000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fimmu.2026.1776452\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6,2]]},"references-count":28,"alternative-id":["10.3389\/fimmu.2026.1776452"],"URL":"https:\/\/doi.org\/10.3389\/fimmu.2026.1776452","relation":{},"ISSN":["1664-3224"],"issn-type":[{"value":"1664-3224","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,6,2]]},"article-number":"1776452"}}