{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T12:06:28Z","timestamp":1778760388576,"version":"3.51.4"},"reference-count":59,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,12,10]],"date-time":"2025-12-10T00:00:00Z","timestamp":1765324800000},"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. Med."],"abstract":"<jats:p>Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare aseptic inflammatory clinical syndrome characterized by widespread skin lesions and bone-joint lesions. The underlying mechanisms remain unclear, making it difficult to treat. Currently, drug-based symptomatic treatment is the main approach, but symptoms often recur or worsen when medications are reduced or discontinued. Recently, there have been increasing reports of the significant therapeutic effects of tonsillectomy in treating SAPHO syndrome. This paper reviews the past evidence on the use of tonsillectomy for SAPHO syndrome by searching PubMed, Google Scholar, Web of Science, Embase, Scopus, CNKI, and Wanfang databases using the keywords \u201cSAPHO syndrome and tonsillectomy.\u201d It also discusses the potential mechanisms of tonsillectomy in treating SAPHO syndrome, aiming to provide new treatment methods for SAPHO syndrome.<\/jats:p>","DOI":"10.3389\/fmed.2025.1695572","type":"journal-article","created":{"date-parts":[[2025,12,10]],"date-time":"2025-12-10T06:41:24Z","timestamp":1765348884000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":1,"title":["Evaluation of tonsillectomy in the treatment of SAPHO syndrome: past evidence and future directions"],"prefix":"10.3389","volume":"12","author":[{"given":"Wen-Yuan","family":"Gan","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ben-Hong","family":"Ren","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qing-Ping","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yi","family":"Wang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2025,12,10]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"118","DOI":"10.1007\/bf02030980","article-title":"Acute pseudoseptic arthritis and palmoplantar pustulosis.","volume":"5","author":"Chamot","year":"1986","journal-title":"Clin Rheumatol."},{"key":"B2","doi-asserted-by":"publisher","first-page":"254","DOI":"10.1016\/j.semarthrit.2012.05.006","article-title":"The SAPHO syndrome.","volume":"42","author":"Nguyen","year":"2012","journal-title":"Seminars Arthritis Rheumatism."},{"key":"B3","doi-asserted-by":"publisher","first-page":"401","DOI":"10.1016\/j.rdc.2013.02.009","article-title":"SAPHO syndrome.","volume":"39","author":"Carneiro","year":"2013","journal-title":"Rheumatic Dis Clin."},{"key":"B4","doi-asserted-by":"publisher","first-page":"937","DOI":"10.5114\/ada.2020.97394","article-title":"SAPHO syndrome: pathogenesis, clinical presentation, imaging, comorbidities and treatment: a review.","volume":"38","author":"Przepiera-B\u0119dzak","year":"2021","journal-title":"Adv Dermatol Allergol."},{"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":"Therapeutic Adv Musculoskeletal Dis."},{"key":"B6","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1007\/s11926-020-00928-1","article-title":"New insights into adult and paediatric chronic non-bacterial osteomyelitis CNO.","volume":"22","author":"Hedrich","year":"2020","journal-title":"Curr Rheumatol Rep."},{"key":"B7","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1016\/j.jbspin.2017.04.010","article-title":"SAPHO, autophagy, IL-1, FoxO1, and Propionibacterium (Cutibacterium) acnes.","volume":"85","author":"Berthelot","year":"2018","journal-title":"Joint Bone Spine."},{"key":"B8","doi-asserted-by":"publisher","first-page":"1255","DOI":"10.1136\/annrheumdis-2020-217250","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 Rheumatic Dis."},{"key":"B9","doi-asserted-by":"publisher","first-page":"e11149","DOI":"10.1097\/md.0000000000011149","article-title":"Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib.","volume":"97","author":"Yang","year":"2018","journal-title":"Medicine."},{"key":"B10","doi-asserted-by":"publisher","first-page":"50","DOI":"10.12144\/zgmfskin202401050","article-title":"Refractory SAPHO syndrome treated with tofacitinib: a case report.","volume":"40","author":"Wenwen","year":"2024","journal-title":"China J Leprosy Skin Dis."},{"key":"B11","doi-asserted-by":"publisher","first-page":"6201887","DOI":"10.1155\/2023\/6201887","article-title":"A case of SAPHO syndrome complicated by uveitis with good response to both TNF inhibitor and JAKinib.","volume":"2023","author":"Baisya","year":"2023","journal-title":"Case Rep Rheumatol."},{"key":"B12","doi-asserted-by":"publisher","first-page":"4527","DOI":"10.12998\/wjcc.v8.i19.4527","article-title":"Rapid remission of refractory synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome in response to the Janus kinase inhibitor tofacitinib: a case report.","volume":"8","author":"Li","year":"2020","journal-title":"World J Clin Cases."},{"key":"B13","doi-asserted-by":"publisher","first-page":"106210","DOI":"10.1201\/9781351042468-1","article-title":"The role of JAK\/STAT signaling pathway and its inhibitors in diseases.","volume":"80","author":"Xin","year":"2020","journal-title":"Int Immunopharmacol."},{"key":"B14","doi-asserted-by":"publisher","first-page":"4280","DOI":"10.12998\/wjcc.v8.i19.4280","article-title":"Role of monoclonal antibody drugs in the treatment of COVID-19.","volume":"8","author":"Ucciferri","year":"2020","journal-title":"World J Clin Cases."},{"key":"B15","doi-asserted-by":"publisher","first-page":"2365","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":"B16","doi-asserted-by":"publisher","first-page":"812","DOI":"10.1111\/1346-8138.14348","article-title":"Treatment outcome and prognostic factors of tonsillectomy for palmoplantar pustulosis and pustulotic arthro-osteitis: a retrospective subjective and objective quantitative analysis of 138 patients.","volume":"45","author":"Takahara","year":"2018","journal-title":"J Dermatol."},{"key":"B17","doi-asserted-by":"publisher","first-page":"519","DOI":"10.1111\/1756-185x.14064","article-title":"Tonsillitis as a possible predisposition to synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome.","volume":"24","author":"Xiang","year":"2021","journal-title":"Int J Rheumatic Dis."},{"key":"B18","doi-asserted-by":"publisher","first-page":"1671","DOI":"10.16352\/j.issn.1001-6325.2020.12.016","article-title":"Improvement of bone pain and rash symptoms in SAPHO patients after tonsillectomy.","volume":"40","author":"Wang","year":"2020","journal-title":"Basic Med Clin Pract."},{"key":"B19","doi-asserted-by":"publisher","first-page":"229","DOI":"10.1016\/j.jns.2014.12.020","article-title":"A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome presenting with hypertrophic pachymeningitis.","volume":"349","author":"Shiraishi","year":"2015","journal-title":"J Neurol Sci."},{"key":"B20","doi-asserted-by":"publisher","first-page":"508","DOI":"10.1016\/j.anl.2009.09.008","article-title":"Fluctuating mixed-type hearing loss associated with synovitis\u2013acne\u2013pustulosis\u2013hyperostosis\u2013osteomyelitis (SAPHO) syndrome.","volume":"37","author":"Shimizu","year":"2010","journal-title":"Auris Nasus Larynx."},{"key":"B21","doi-asserted-by":"publisher","first-page":"e186","DOI":"10.1111\/ijd.16492","article-title":"Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome associated with pyoderma gangrenosum: report of two patients.","volume":"62","author":"Ma","year":"2023","journal-title":"Int J Dermatol."},{"key":"B22","doi-asserted-by":"publisher","first-page":"S719","DOI":"10.1097\/rhu.0000000000001546","article-title":"Tonsillectomy leads to remission of bone marrow edema and palmoplantar pustulosis in synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome.","volume":"27","author":"Wang","year":"2021","journal-title":"J Clin Rheumatol."},{"key":"B23","doi-asserted-by":"publisher","first-page":"rjaa288","DOI":"10.1093\/jscr\/rjaa288","article-title":"Tonsillectomy as an effective treatment for arthralgia of SAPHO syndrome.","volume":"2020","author":"Horiguchi","year":"2020","journal-title":"J Surg Case Rep."},{"key":"B24","doi-asserted-by":"publisher","first-page":"159","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 Rheumatism."},{"key":"B25","doi-asserted-by":"publisher","first-page":"639","DOI":"10.3899\/jrheum.090863","article-title":"Prevalence of autoantibodies in SAPHO syndrome: a single-center study of 90 patients.","volume":"37","author":"Grosjean","year":"2010","journal-title":"J Rheumatol."},{"key":"B26","doi-asserted-by":"publisher","first-page":"2301","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":"B27","doi-asserted-by":"publisher","first-page":"1152","DOI":"10.1111\/1756-185x.13899","article-title":"Axial skeletal lesions and disease duration in SAPHO syndrome: A retrospective review of computed tomography findings in 81 patients.","volume":"23","author":"Li","year":"2020","journal-title":"Int J Rheumatic Dis."},{"key":"B28","doi-asserted-by":"publisher","first-page":"758","DOI":"10.1684\/ejd.2010.1109","article-title":"A pilot study of medium-dose cyclosporine for the treatment of palmoplantar pustulosis complicated with pustulotic arthroosteitis.","volume":"20","author":"Hayama","year":"2010","journal-title":"Eur J Dermatol."},{"key":"B29","doi-asserted-by":"publisher","first-page":"618","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 Rheumatism."},{"key":"B30","doi-asserted-by":"publisher","first-page":"1699","DOI":"10.3899\/jrheum.091086","article-title":"Tumor necrosis factor-\u03b1 blockers in SAPHO syndrome.","volume":"37","author":"Abdelghani","year":"2010","journal-title":"J Rheumatol."},{"key":"B31","doi-asserted-by":"publisher","first-page":"281","DOI":"10.1007\/s00296-012-2606-y","article-title":"The lobster sign in SAPHO syndrome: unusually extensive osteitis of the anterior chest wall partially responsive to infliximab.","volume":"34","author":"Ani\u0107","year":"2014","journal-title":"Rheumatol Int."},{"key":"B32","doi-asserted-by":"publisher","first-page":"1505","DOI":"10.1093\/rheumatology\/keq122","article-title":"Biologic therapy in refractory chronic non-bacterial osteomyelitis of childhood.","volume":"49","author":"Eleftheriou","year":"2010","journal-title":"Rheumatology."},{"key":"B33","doi-asserted-by":"publisher","first-page":"1965","DOI":"10.1002\/art.10539","article-title":"Sustained response to tumor necrosis factor \u03b1\u2013blocking agents in two patients with SAPHO syndrome.","volume":"46","author":"Wagner","year":"2002","journal-title":"Arthritis Rheumatism."},{"key":"B34","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":"B35","doi-asserted-by":"publisher","first-page":"44","DOI":"10.1016\/j.reumae.2015.11.022","article-title":"SAPHO syndrome presenting as an osteolytic lesion of the neck.","volume":"13","author":"Mateo","year":"2017","journal-title":"Reumatol Cl\u00edn."},{"key":"B36","doi-asserted-by":"publisher","first-page":"730","DOI":"10.1093\/rheumatology\/kei221","article-title":"In SAPHO syndrome anti-TNF-\u03b1 therapy may induce persistent amelioration of osteoarticular complaints, but may exacerbate cutaneous manifestations.","volume":"45","author":"Massara","year":"2006","journal-title":"Rheumatology."},{"key":"B37","doi-asserted-by":"publisher","first-page":"bcr1020081145","DOI":"10.1136\/bcr.10.2008.1145","article-title":"Response to infliximab in SAPHO syndrome.","volume":"2009","author":"Fruehauf","year":"2009","journal-title":"Case Rep."},{"key":"B38","doi-asserted-by":"publisher","first-page":"0300060520949100","DOI":"10.1177\/0300060520949100","article-title":"Successful treatment of synovitis, acne, pustulosis, hyperostosis, and osteitis and paradoxical skin lesions by Tripterygium wilfordii hook f: a case report.","volume":"48","author":"Zhang","year":"2020","journal-title":"J Int Med Res."},{"key":"B39","doi-asserted-by":"publisher","first-page":"bcr1120115197","DOI":"10.1136\/bcr.11.2011.5197","article-title":"The dark side of SAPHO syndrome.","volume":"2011","author":"Henriques","year":"2011","journal-title":"Case Rep."},{"key":"B40","doi-asserted-by":"publisher","first-page":"2151","DOI":"10.1007\/s10067-017-3751-1","article-title":"Clinical heterogeneity of SAPHO syndrome: challenging diagnose and treatment.","volume":"36","author":"Cianci","year":"2017","journal-title":"Clin Rheumatol."},{"key":"B41","doi-asserted-by":"publisher","first-page":"1337","DOI":"10.1007\/s00431-011-1501-y","article-title":"Lifethreatening disseminated tuberculosis as a complication of TNF-\u03b1 blockade in an adolescent.","volume":"170","author":"Hess","year":"2011","journal-title":"Eur J Pediatrics."},{"key":"B42","doi-asserted-by":"publisher","first-page":"476","DOI":"10.1111\/1756-185x.12525","article-title":"Development of aseptic subcutaneous abscess after tocilizumab therapy in a patient with SAPHO syndrome complicated by amyloid A amyloidosis.","volume":"18","author":"Fujita","year":"2015","journal-title":"Int J Rheumatic Dis."},{"key":"B43","doi-asserted-by":"publisher","first-page":"959","DOI":"10.1111\/bjd.16071","article-title":"Successful treatment of SAPHO syndrome with apremilast.","volume":"179","author":"Adamo","year":"2018","journal-title":"Br J Dermatol."},{"key":"B44","doi-asserted-by":"publisher","first-page":"944","DOI":"10.13201\/j.issn.2096-7993.2021.10.018","article-title":"SAPHO syndrome with chronic tonsillitis: a case report and literature review.","volume":"35","author":"Jia","year":"2021","journal-title":"Lin Chuang er bi yan hou tou Jing wai ke za zhi."},{"key":"B45","doi-asserted-by":"publisher","first-page":"1102","DOI":"10.3760\/cma.j.cn115330-20201217-00929","article-title":"Tonsillectomy as a treatment for SAPHO syndrome: a case report.","volume":"56","author":"Xie","year":"2021","journal-title":"Zhonghua Er Bi Yan Hou Tou Jing Wai Ke Za Zhi."},{"key":"B46","doi-asserted-by":"publisher","first-page":"333","DOI":"10.1016\/s0950-3579(94)80022-7","article-title":"The SAPHO syndrome.","volume":"8","author":"Kahn","year":"1994","journal-title":"Bailliere\u2019s Clin Rheumatol."},{"key":"B47","doi-asserted-by":"publisher","first-page":"86","DOI":"10.1002\/iid3.248","article-title":"Recent advances in the immunological understanding of association between tonsil and immunoglobulin a nephropathy as a tonsil-induced autoimmune\/inflammatory syndrome.","volume":"7","author":"Harabuchi","year":"2019","journal-title":"Immunity Inflammation Dis."},{"key":"B48","doi-asserted-by":"publisher","first-page":"931","DOI":"10.1111\/1346-8138.15100","article-title":"Pathogenic role of palatine tonsils in palmoplantar pustulosis: a review.","volume":"46","author":"Harabuchi","year":"2019","journal-title":"J Dermatol."},{"key":"B49","doi-asserted-by":"publisher","first-page":"736","DOI":"10.1080\/03655230410003387","article-title":"Tonsillectomy and steroid pulse therapy significantly impact on clinical remission in patients with IgA nephropathy.","volume":"38","author":"Hotta","year":"2001","journal-title":"Am J Kidney Dis."},{"key":"B50","doi-asserted-by":"publisher","first-page":"1546","DOI":"10.1093\/ndt\/gfu020","article-title":"A multicenter randomized controlled trial of tonsillectomy combined with steroid pulse therapy in patients with immunoglobulin A nephropathy.","volume":"29","author":"Kawamura","year":"2014","journal-title":"Nephrol Dialysis Transpl."},{"key":"B51","doi-asserted-by":"publisher","first-page":"94","DOI":"10.1007\/s10157-015-1138-7","article-title":"Significance of tonsillectomy combined with steroid pulse therapy for IgA nephropathy with mild proteinuria.","volume":"20","author":"Komatsu","year":"2016","journal-title":"Clin Exp Nephrol."},{"key":"B52","doi-asserted-by":"publisher","first-page":"384","DOI":"10.1080\/00016489950181431","article-title":"Increased Interleukin-6, interferon-? and tumour necrosis Factor-a production by tonsillar mononuclear cells stimulated with alpha-streptococci in patients with pustulosis palmaris et plantaris.","volume":"119","author":"Murakata","year":"1999","journal-title":"Acta Otolaryngol."},{"key":"B53","doi-asserted-by":"publisher","first-page":"949","DOI":"10.3950\/jibiinkoka.93.949","article-title":"Investigation of the immune response in tonsillar lymphocytes against streptococci in patients with pustulosis palmaris et plantaris.","volume":"93","author":"Kukuminato","year":"1990","journal-title":"Nippon Jibiinkoka Gakkai Kaiho."},{"key":"B54","doi-asserted-by":"publisher","first-page":"786","DOI":"10.3950\/jibiinkoka.93.786","article-title":"Investigation of the bacterial flora in the tonsillar lacunae and serum levels of streptococcal antigen-specific antibodies in patients with pustulosis palmaris et plantaris.","volume":"93","author":"Kukuminato","year":"1990","journal-title":"Nippon Jibiinkoka Gakkai Kaiho."},{"key":"B55","first-page":"201","article-title":"Cytokine production by tonsillar lymphocytes stimulated with alpha-streptococci in patients with pustulosis palmaris et plantaris.","volume":"523","author":"Murakata","year":"1996","journal-title":"Acta oto-laryngologica. Supplementum."},{"key":"B56","doi-asserted-by":"publisher","first-page":"237","DOI":"10.1111\/j.1365-2133.2012.11215.x","article-title":"The role of the palatine tonsils in the pathogenesis and treatment of psoriasis.","volume":"168","author":"Sigurdardottir","year":"2013","journal-title":"Br J Dermatol."},{"key":"B57","doi-asserted-by":"publisher","first-page":"1705","DOI":"10.1001\/jama.1910.04330200015004","article-title":"Acute nephritis following acute tonsillitis.","volume":"55","author":"Loeb","year":"1910","journal-title":"J Am Med Assoc."},{"key":"B58","doi-asserted-by":"publisher","first-page":"1027","DOI":"10.1056\/nejm193505302122203","article-title":"Arthritis and tonsillar infection.","volume":"212","author":"Nissen","year":"1935","journal-title":"N Engl J Med."},{"key":"B59","doi-asserted-by":"publisher","first-page":"264","DOI":"10.1126\/science.1110267","article-title":"Professional antigen-presentation function by human \u03b3\u03b4 T cells.","volume":"309","author":"Brandes","year":"2005","journal-title":"Science."}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1695572\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,12,10]],"date-time":"2025-12-10T06:41:28Z","timestamp":1765348888000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1695572\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,12,10]]},"references-count":59,"alternative-id":["10.3389\/fmed.2025.1695572"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2025.1695572","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,12,10]]},"article-number":"1695572"}}