{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,16]],"date-time":"2026-09-16T07:58:48Z","timestamp":1789545528783,"version":"build-2803163510"},"reference-count":23,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2024,7,31]],"date-time":"2024-07-31T00:00:00Z","timestamp":1722384000000},"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":[[2025,1,16]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome is a rare autoinflammatory disease characterised by bone inflammation and skin manifestations including acne, palmoplantar pustulosis, psoriasis, or hidradenitis suppurativa. SAPHO syndrome is considered on the same spectrum as chronic nonbacterial osteomyelitis\/chronic recurrent multifocal osteomyelitis, the former often being the nomenclature in adults and the latter in children. The diagnosis is made on patterns of clinical manifestations and is a diagnosis of exclusion. While skin and bone manifestations are commonly described with SAPHO syndrome, pleural involvement is rare, and few cases have been described in the literature, especially in paediatric patients. Herein we present a 14-year-old female with a past medical history of hidradenitis supprtiva, eczema, psoriasis, and a prior episode of culture-negative osteomyelitis who presented to the emergency room with chief complaints of right-sided pain with inspiration and back pain. Exam revealed palmoplantar pustulosis, hidradenitis supprativa, psoriasis, and tenderness of vertebrae. Imaging showed a right-sided pleural effusion and multiple sites of osteitis. Laboratory evaluation revealed elevated inflammatory markers, an exudative pleural effusion with neutrophilic predominance, and no evidence of malignancy, infection, or immunodeficiency. The patient was diagnosed with SAPHO syndrome and treated with naproxen, methotrexate, and golimumab with significant improvement including resolution of the pleural effusion. Paediatric SAPHO syndrome is a rare disease that classically causes osteitis and skin manifestations. This case highlights that pleural effusion can be a rare manifestation of paediatric SAPHO syndrome. Patients with suspected SAPHO syndrome with respiratory symptoms should be evaluated for pleural effusion.<\/jats:p>","DOI":"10.1093\/mrcr\/rxae038","type":"journal-article","created":{"date-parts":[[2024,8,1]],"date-time":"2024-08-01T01:35:46Z","timestamp":1722476146000},"page":"168-173","source":"Crossref","is-referenced-by-count":2,"title":["Paediatric SAPHO syndrome with pleural effusion: Case report of a unique finding in a rare disease"],"prefix":"10.1093","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0735-1204","authenticated-orcid":false,"given":"Christopher","family":"Kruger","sequence":"first","affiliation":[{"name":"Division of General Pediatrics, University of Michigan C. S. Mott Children\u2019s Hospital , Ann Arbor, MI, 48109","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christine","family":"Wang","sequence":"additional","affiliation":[{"name":"Division of Pediatric Rheumatology, University of Michigan C. S. Mott Children\u2019s Hospital , Ann Arbor, MI, 48109","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-1426-5322","authenticated-orcid":false,"given":"Andrew","family":"Grim","sequence":"additional","affiliation":[{"name":"Division of Pediatric Rheumatology, University of Michigan C. S. Mott Children\u2019s Hospital , Ann Arbor, MI, 48109","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2024,7,31]]},"reference":[{"key":"2025011615475056200_R1","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":"2025011615475056200_R2","doi-asserted-by":"crossref","first-page":"1487","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":"2025011615475056200_R3","doi-asserted-by":"crossref","DOI":"10.1016\/j.jtauto.2021.100095","article-title":"Chronic nonbacterial osteomyelitis (CNO) and chronic recurrent multifocal osteomyelitis (CRMO)","volume":"4","author":"Zhao","year":"2021","journal-title":"J Transl Autoimmun"},{"key":"2025011615475056200_R4","doi-asserted-by":"crossref","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":"2025011615475056200_R5","doi-asserted-by":"crossref","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\u0229dzak","year":"2021","journal-title":"Adv Dermatol Allergol"},{"key":"2025011615475056200_R6","doi-asserted-by":"crossref","DOI":"10.1378\/chest.120.5.1752","article-title":"SAPHO with pleural effusion 61 YO M","volume":"120","author":"Fernandez-Campillo","year":"2001","journal-title":"Chest"},{"key":"2025011615475056200_R7","doi-asserted-by":"crossref","first-page":"2677","DOI":"10.2169\/internalmedicine.8473-21","article-title":"Synovitis-acne-pustulosis-hyperostosis-osteitis syndrome with bilateral pleural effusion","volume":"61","author":"Adachi-Katayama","year":"2022","journal-title":"Intern Med"},{"key":"2025011615475056200_R8","doi-asserted-by":"crossref","first-page":"2779","DOI":"10.2169\/internalmedicine.8250-16","article-title":"Synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome with significant bilateral pleural effusions","volume":"56","author":"Hasegawa","year":"2017","journal-title":"Intern Med"},{"key":"2025011615475056200_R9","doi-asserted-by":"crossref","first-page":"599","DOI":"10.1111\/1756-185X.13821","article-title":"Pleural effusion: an uncommon manifestation of SAPHO syndrome?","volume":"23","author":"Mor\u00e1n-\u00c1lvarez","year":"2020","journal-title":"Int J Rheum Dis"},{"key":"2025011615475056200_R10","doi-asserted-by":"crossref","first-page":"463","DOI":"10.1093\/rheumatology\/38.5.463","article-title":"SAPHO syndrome or psoriatic arthritis? 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