{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2024,8,9]],"date-time":"2024-08-09T06:29:44Z","timestamp":1723184984494},"reference-count":21,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2024,5,15]],"date-time":"2024-05-15T00:00:00Z","timestamp":1715731200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,7,8]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Synovitis-acne-pustulosis-hyperostosis-osteomyelitis (SAPHO) syndrome is characterised by aseptic osteitis and is often complicated by pustular dermatitis, such as palmoplantar pustulosis or acne. Although bone lesions are most found in the anterior thoracic region or spine, femoral lesions are not well documented in the literature. There is no established treatment for this condition, and few reports have described its long-term course. Here, we describe two cases of SAPHO syndrome involving the femur and discuss their long-term follow-up. A 40-year-old man (Case 1) presented with right thigh pain. Fifteen years after the initial diagnosis, the pain could be controlled with minomycin, salazosulfapyridine, and methotrexate. X-rays of the femur showed gradual cortical thickening. Although there were waves of pain, it gradually improved with the adjustment of drugs 25\u2009years following the initial diagnosis. A 35-year-old man (Case 2) with right thigh pain was prescribed salazosulfapyridine and methotrexate; however, these were ineffective. Alendronate and guselkumab also proved ineffective. Ultimately, infliximab was started 9\u2009years following disease onset, and pain became manageable. X-rays of the femur showed cortical thickening. SAPHO syndrome can be managed with drug therapies, such as nonsteroidal anti-inflammatory drugs, methotrexate, and conventional synthetic disease-modifying antirheumatic drugs; however, there are occasional treatment-resistant cases.<\/jats:p>","DOI":"10.1093\/mrcr\/rxae024","type":"journal-article","created":{"date-parts":[[2024,5,15]],"date-time":"2024-05-15T15:16:03Z","timestamp":1715786163000},"page":"373-377","source":"Crossref","is-referenced-by-count":1,"title":["Long-term clinical course of two rare cases of synovitis-acne-pustulosis-hyperostosis-osteomyelitis syndrome involving only unilateral femur"],"prefix":"10.1093","volume":"8","author":[{"given":"Hiroki","family":"Ito","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery, Toyohashi Municipal Hospital , Toyohashi, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yuji","family":"Hirano","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Toyohashi Municipal Hospital , Toyohashi, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2024,5,15]]},"reference":[{"key":"2024070814455124200_R1","doi-asserted-by":"crossref","first-page":"554","DOI":"10.1136\/ard.40.6.554","article-title":"Incidence of arthro-osteitis in patients with pustulosis palmaris et plantaris","volume":"40","author":"Sonozaki","year":"1981","journal-title":"Ann Rheum Dis"},{"key":"2024070814455124200_R2","first-page":"187","article-title":"Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases","volume":"54","author":"Chamot","year":"1987","journal-title":"Rev Rhum Mal Osteoartic"},{"key":"2024070814455124200_R3","first-page":"109","article-title":"Synovitis-acne-pustulosis hyperostosis-osteomyelitis syndrome (SAPHO). A new syndrome among the spondyloarthropathies?","volume":"6","author":"Benhamou","year":"1988","journal-title":"Clin Exp Rheumatol"},{"key":"2024070814455124200_R4","doi-asserted-by":"crossref","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 Rheum"},{"key":"2024070814455124200_R5","doi-asserted-by":"crossref","first-page":"813","DOI":"10.1002\/art.24540","article-title":"Clinical and radiologic evolution of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a single center study of a cohort of 71 subjects","volume":"61","author":"Colina","year":"2009","journal-title":"Arthritis Rheum"},{"key":"2024070814455124200_R6","doi-asserted-by":"crossref","first-page":"245","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":"2024070814455124200_R7","doi-asserted-by":"crossref","first-page":"254","DOI":"10.1016\/j.semarthrit.2012.05.006","article-title":"The SAPHO syndrome","volume":"42","author":"Nguyen","year":"2012","journal-title":"Semin Arthritis Rheum"},{"key":"2024070814455124200_R8","doi-asserted-by":"crossref","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 Rheum"},{"key":"2024070814455124200_R9","first-page":"2135","article-title":"SAPHO syndrome: a followup study of 19 cases with special emphasis on enthesis involvement","volume":"22","author":"Maugars","year":"1995","journal-title":"J Rheumatol"},{"key":"2024070814455124200_R10","doi-asserted-by":"crossref","first-page":"100","DOI":"10.2174\/1874325000903010100","article-title":"SAPHO syndrome diagnosis and treatment: report of five cases and review of the literature","volume":"3","author":"Matzaroglou","year":"2009","journal-title":"Open Orthop J"},{"key":"2024070814455124200_R11","doi-asserted-by":"crossref","first-page":"277","DOI":"10.1097\/00003086-200509000-00044","article-title":"Aseptic femoral osteitis and sternocostal hyperostosis from SAPHO syndrome","volume":"438","author":"Franz","year":"2005","journal-title":"Clin Orthop Relat Res"},{"key":"2024070814455124200_R12","doi-asserted-by":"crossref","first-page":"243","DOI":"10.3109\/2000-1967-045","article-title":"SAPHO syndrome with a tumour-like bony proliferative lesion in distal femur. A case report","volume":"111","author":"Watanuki","year":"2006","journal-title":"Ups J Med Sci"},{"key":"2024070814455124200_R13","doi-asserted-by":"crossref","first-page":"1131","DOI":"10.1007\/s10067-020-05322-x","article-title":"The effectiveness of treatments for patients with SAPHO syndrome: a follow-up study of 24 cases from a single center and review of literature","volume":"40","author":"Huang","year":"2021","journal-title":"Clin Rheumatol"},{"key":"2024070814455124200_R14","doi-asserted-by":"crossref","first-page":"713","DOI":"10.1007\/s00296-008-0760-z","article-title":"Successful treatment of SAPHO syndrome with an oral bisphosphonate","volume":"29","author":"Ichikawa","year":"2009","journal-title":"Rheumatol Int"},{"key":"2024070814455124200_R15","first-page":"663","article-title":"Efficacy of bisphosphonates in patients with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a prospective open study","volume":"37","author":"Li","year":"2019","journal-title":"Clin Exp Rheumatol"},{"key":"2024070814455124200_R16","doi-asserted-by":"crossref","first-page":"1153","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":"2024070814455124200_R17","doi-asserted-by":"crossref","first-page":"2318","DOI":"10.1111\/jdv.16355","article-title":"Efficacy of guselkumab in a subpopulation with pustulotic arthro-osteitis through week 52: an exploratory analysis of a phase 3, randomized, double-blind, placebo-controlled study in Japanese patients with palmoplantar pustulosis","volume":"34","author":"Yamamoto","year":"2020","journal-title":"J Eur Acad Dermatol Venereol"},{"key":"2024070814455124200_R18","doi-asserted-by":"crossref","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 Inflamm Res"},{"key":"2024070814455124200_R19","doi-asserted-by":"crossref","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":"2024070814455124200_R20","doi-asserted-by":"crossref","first-page":"e206","DOI":"10.1111\/ijd.12675","article-title":"Etanercept in the treatment of SAPHO syndrome","volume":"54","author":"S\u00e1ez-Mart\u00edn","year":"2015","journal-title":"Int J Dermatol"},{"key":"2024070814455124200_R21","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1016\/j.rdc.2013.02.009","article-title":"SAPHO syndrome","volume":"39","author":"Carneiro","year":"2013","journal-title":"Rheum Dis Clin North Am"}],"container-title":["Modern Rheumatology Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/mrcr\/advance-article-pdf\/doi\/10.1093\/mrcr\/rxae024\/57701441\/rxae024.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/8\/2\/373\/58470454\/rxae024.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/8\/2\/373\/58470454\/rxae024.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,7,8]],"date-time":"2024-07-08T14:49:02Z","timestamp":1720450142000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/mrcr\/article\/8\/2\/373\/7674126"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,5,15]]},"references-count":21,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2024,5,15]]},"published-print":{"date-parts":[[2024,7,8]]}},"URL":"https:\/\/doi.org\/10.1093\/mrcr\/rxae024","relation":{},"ISSN":["2472-5625"],"issn-type":[{"value":"2472-5625","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2024,7]]},"published":{"date-parts":[[2024,5,15]]}}}