{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,1]],"date-time":"2026-08-01T16:12:38Z","timestamp":1785600758431,"version":"3.56.0"},"reference-count":26,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,4,24]],"date-time":"2025-04-24T00:00:00Z","timestamp":1745452800000},"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:sec><jats:title>Introduction<\/jats:title><jats:p>Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is an extremely rare condition with nonspecific clinical signs and symptoms.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case description<\/jats:title><jats:p>Here, we present the case of a 54-year-old Chinese woman with an 8-year history of recurrent furuncles and a 6-year history of clavicular pain. Initially, computed tomography (CT) showed nonspecific changes. The patient was treated with nonsteroidal anti-inflammatory drugs for symptomatic relief; however, clavicular symptoms recurred intermittently. Two years before the current presentation, the patient experienced ocular discomfort from bilateral lacrimal gland enlargement and extraocular muscle thickening. She exhibited elevated IgG4 levels and was diagnosed with IgG4-related ophthalmic disease. Treatment with low-dose glucocorticoids slightly improved her clavicular symptoms. However, over the past month, her clavicular pain worsened. Recent CT and magnetic resonance imaging revealed a deformity of the right clavicular cortex near the end, with poor bone quality and continuity, and bone scintigraphy revealed intense radiotracer uptake in the sternoclavicular region. Consequently, the patient was diagnosed with SAPHO syndrome.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>While clinical manifestations and imaging are helpful in narrowing the differential diagnosis, biopsy and histopathological examinations are necessary to confirm SAPHO syndrome. Regulation of TNF-\u03b1 may be a therapeutic option for bone pain in this patient. In patients with an initial presentation of abnormal IgG4 levels, physicians must maintain a high index of suspicion to ensure appropriate treatment.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fimmu.2025.1563542","type":"journal-article","created":{"date-parts":[[2025,4,24]],"date-time":"2025-04-24T05:29:56Z","timestamp":1745472596000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":4,"title":["SAPHO syndrome complicated by IgG4-related ophthalmic disease: a case report and literature review"],"prefix":"10.3389","volume":"16","author":[{"given":"Chenxiao","family":"Liu","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tingting","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yanyan","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qi","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hao","family":"Hu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Huanhuan","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2025,4,24]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/keab760","article-title":"SAPHO syndrome","volume":"61","author":"Xu","year":"2022","journal-title":"Rheumatol (Oxford)"},{"key":"B2","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","year":"2024","journal-title":"Rheumatol Int"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.1111\/1756-185X.14779","article-title":"Extracutaneous\/osteoarticular manifestations in patients with SAPHO syndrome","volume":"26","author":"Hu","year":"2023","journal-title":"Int J Rheum Dis"},{"key":"B4","doi-asserted-by":"publisher","first-page":"216","DOI":"10.1186\/s13075-020-02309-6","article-title":"Huo J: Anterior chest wall in SAPHO syndrome: magnetic resonance imaging findings","volume":"22","author":"Yu","year":"2020","journal-title":"Arthritis Res Ther"},{"key":"B5","doi-asserted-by":"publisher","DOI":"10.4103\/0366-6999.172607","article-title":"Sacroiliitis and spondylitis with sternoclavicular hyperostosis: SAPHO or an ankylosing spondylitis variant","volume":"129","author":"Shen","year":"2016","journal-title":"Chin Med J (Engl)"},{"key":"B6","doi-asserted-by":"publisher","first-page":"26","DOI":"10.1007\/s13730-015-0184-z","article-title":"A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome complicated by IgA nephropathy with nephrotic syndrome","volume":"5","author":"Morimoto","year":"2016","journal-title":"CEN Case Rep"},{"key":"B7","doi-asserted-by":"publisher","first-page":"3558853","DOI":"10.1155\/2024\/3558853","article-title":"Enteropathic SAPHO syndrome in ulcerative colitis responsive to bisphosphonates","author":"Phillipps","year":"2024","journal-title":"Case Rep Rheumatol"},{"key":"B8","doi-asserted-by":"publisher","DOI":"10.2169\/internalmedicine.3309-19","article-title":"IgG4-related ophthalmic disease with proptosis","volume":"59","author":"Matsuhisa","year":"2020","journal-title":"Intern Med"},{"key":"B9","doi-asserted-by":"publisher","first-page":"101782","DOI":"10.1016\/j.ajoc.2022.101782","article-title":"IgG4-related disease presenting with profound bilateral orbital and adnexal inflammation","volume":"29","author":"Kutzscher","year":"2023","journal-title":"Am J Ophthalmol Case Rep"},{"key":"B10","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1007\/s10384-014-0352-2","article-title":"Diagnostic criteria for IgG4-related ophthalmic disease","volume":"59","author":"Goto","year":"2015","journal-title":"Jpn J Ophthalmol"},{"key":"B11","first-page":"35","article-title":"Serum IgG4 elevation in SAPHO syndrome: does it unmask a disease activity marker","volume":"38","author":"Li","year":"2020","journal-title":"Clin Exp Rheumatol"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.1155\/2020\/3757015","article-title":"Increased circulating Th1 and Tfh1 cell numbers are associated with disease activity in glucocorticoid-treated patients with IgG4-related disease","volume":"2020","author":"Xia","year":"2020","journal-title":"J Immunol Res"},{"key":"B13","doi-asserted-by":"publisher","DOI":"10.1007\/s12026-023-09381-5","article-title":"SAPHO syndrome after COVID-19 vaccination complicated with thyroid function abnormalities: a case report and literature review","volume":"71","author":"Lin","year":"2023","journal-title":"Immunol Res"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.1038\/s41591-021-01419-1","article-title":"COVID-19 vaccination and immune thrombocytopenia","volume":"27","author":"Pishko","year":"2021","journal-title":"Nat Med"},{"key":"B15","doi-asserted-by":"publisher","DOI":"10.5114\/pedm.2020.95618","article-title":"Anti-tumour necrosis factor alpha therapy - Does it increase the risk of thyroid disease or protect against its development","volume":"26","author":"Furtak","year":"2020","journal-title":"Pediatr Endocrinol Diabetes Metab"},{"key":"B16","doi-asserted-by":"publisher","DOI":"10.1016\/bs.vh.2017.07.001","article-title":"Thyroid autoimmunity: An interplay of factors","volume":"106","author":"Merrill","year":"2018","journal-title":"Vitam Horm"},{"key":"B17","doi-asserted-by":"publisher","first-page":"1770","DOI":"10.1093\/rheumatology\/keae380","article-title":"TNF-alpha blockade in primary chronic non-bacterial osteomyelitis of the mandible","volume":"64","author":"de La Rochefoucauld","year":"2025","journal-title":"Rheumatol (Oxford)"},{"key":"B18","doi-asserted-by":"publisher","DOI":"10.1136\/rmdopen-2023-003688","article-title":"Treatment and monitoring of SAPHO syndrome: a systematic review","volume":"9","author":"Li","year":"2023","journal-title":"RMD Open"},{"key":"B19","doi-asserted-by":"publisher","DOI":"10.1177\/0300060518778190","article-title":"The relationship between inflammatory factors, oxidative stress and DIO-1 concentration in patients with chronic renal failure accompanied with or without euthyroid sick syndrome","volume":"46","author":"Qin","year":"2018","journal-title":"J Int Med Res"},{"key":"B20","doi-asserted-by":"publisher","DOI":"10.1007\/s10067-007-0767-y","article-title":"Infliximab can induce a prolonged clinical remission and a decrease in thyroid hormonal requirements in a patient with SAPHO syndrome and hypothyroidism","volume":"27","author":"Sabugo","year":"2008","journal-title":"Clin Rheumatol"},{"key":"B21","doi-asserted-by":"publisher","DOI":"10.7150\/jca.53349","article-title":"The expression of transcription factors is different in papillary thyroid cancer cells during TNF - alpha induced EMT","volume":"12","author":"Lv","year":"2021","journal-title":"J Cancer"},{"key":"B22","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":"B23","first-page":"89","article-title":"Effect of cream, prepared with Tripterygium wilfordii Hook F and other four medicinals, on joint pain and swelling in patients with rheumatoid arthritis: a double-blinded, randomized, placebo controlled clinical trial","volume":"39","author":"Jiao","year":"2019","journal-title":"J Tradit Chin Med"},{"key":"B24","doi-asserted-by":"publisher","DOI":"10.3389\/fphar.2022.938849","article-title":"Tripterygium wilfordii glycosides ameliorates collagen-induced arthritis and aberrant lipid metabolism in rats","volume":"13","author":"Zhu","year":"2022","journal-title":"Front Pharmacol"},{"key":"B25","doi-asserted-by":"publisher","DOI":"10.1007\/s10067-020-05562-x","article-title":"Tripterygium wilfordii Hook F. in the treatment of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a clinical trial","volume":"40","author":"Wang","year":"2021","journal-title":"Clin Rheumatol"},{"key":"B26","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"}],"container-title":["Frontiers in Immunology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fimmu.2025.1563542\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,24]],"date-time":"2025-04-24T05:29:57Z","timestamp":1745472597000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fimmu.2025.1563542\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,4,24]]},"references-count":26,"alternative-id":["10.3389\/fimmu.2025.1563542"],"URL":"https:\/\/doi.org\/10.3389\/fimmu.2025.1563542","relation":{},"ISSN":["1664-3224"],"issn-type":[{"value":"1664-3224","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,4,24]]},"article-number":"1563542"}}