{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,16]],"date-time":"2025-10-16T09:24:02Z","timestamp":1760606642597,"version":"3.40.4"},"reference-count":38,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"12","license":[{"start":{"date-parts":[[2021,3,26]],"date-time":"2021-03-26T00:00:00Z","timestamp":1616716800000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"name":"CAMS Initiative Fund for Medical Sciences","award":["2017-I2N-3-001"],"award-info":[{"award-number":["2017-I2N-3-001"]}]},{"name":"Capital Medical Research and Development Fund","award":["2016-4-40112"],"award-info":[{"award-number":["2016-4-40112"]}]},{"name":"National Key Research and Development Program of China","award":["2016YFC0901500"],"award-info":[{"award-number":["2016YFC0901500"]}]}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"abstract":"<jats:sec>\n            <jats:title>Abstract<\/jats:title>\n            <jats:p>To identify the prevalence and clinical characteristics of Sj\u00f6gren syndrome (SS) in a Chinese single-center cohort of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome.<\/jats:p>\n            <jats:p>Patients diagnosed with SS were screened out from a cohort of 164 cases of SAHPO syndrome. Information regarding the patients\u2019 gender, age at onset, clinical features, laboratory tests, bone scintigraphy, and treatment was reviewed.<\/jats:p>\n            <jats:p>Five patients were screened out. The prevalence of SS in SAPHO patients was 3.05% The mean onset age of SS was 48.0\u200a\u00b1\u200a12.0 years old and no apparent time order in the occurrence of SAPHO and SS was observed. Compared with the general SAPHO cohort, the 5 SS patients exhibited no significant difference in the SAPHO related clinical features or inflammatory markers, except for a higher prevalence of peripheral joints and bones involvement in bone scintigraphy. Objective evidence of dryness and positive salivary gland biopsy were found in all the patients. However, the positive rates of SSA and SSB antibody were only 20%. Anti-inflammatory treatment for SS was recorded in 3 patients (ESSDAI score: 3 in 2 patients; 12 in 1 patient) with extra-glandular manifestations, severe complications or poor response to the basic treatment.<\/jats:p>\n            <jats:p>The prevalence of SS is higher in the SAPHO cohort than in the general Chinese population. Objective tests or biopsy might be more indicative than the antibody detection for SS diagnosis. Anti-inflammatory treatment should be prescribed in consideration of both the severity of SS and the demand for disease activity control of SAPHO.<\/jats:p>\n          <\/jats:sec>","DOI":"10.1097\/md.0000000000023940","type":"journal-article","created":{"date-parts":[[2021,3,23]],"date-time":"2021-03-23T19:36:23Z","timestamp":1616528183000},"page":"e23940","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":2,"title":["Coexistence of Sj\u00f6gren syndrome in patients with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome"],"prefix":"10.1097","volume":"100","author":[{"given":"Lun","family":"Wang","sequence":"first","affiliation":[{"name":"Institute of Clinical Medicine"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yanying","family":"Yu","sequence":"additional","affiliation":[{"name":"Institute of Clinical Medicine"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shuo","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Rheumatology and Clinical Immunology, Peking Union Medical College Hospital"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wen","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Rheumatology and Clinical Immunology, Peking Union Medical College Hospital"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chen","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Traditional Chinese Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China."}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"276","published-online":{"date-parts":[[2021,3,26]]},"reference":[{"key":"R1-20250504","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"},{"key":"R2-20250504","doi-asserted-by":"crossref","first-page":"658","DOI":"10.1097\/BOR.0b013e328363eb08","article-title":"Autoinflammatory bone disorders: update on immunologic abnormalities and clues about possible triggers","volume":"25","author":"Sharma","year":"2013","journal-title":"Curr Opin Rheumatol"},{"key":"R3-20250504","doi-asserted-by":"crossref","first-page":"2138","DOI":"10.1007\/s10620-013-2653-6","article-title":"A systematic review of SAPHO syndrome and inflammatory bowel disease association","volume":"58","author":"Naves","year":"2013","journal-title":"Dig Dis Sci"},{"key":"R4-20250504","doi-asserted-by":"crossref","first-page":"e5724","DOI":"10.1097\/MD.0000000000005724","article-title":"The coexistence of SAPHO syndrome and rheumatoid arthritis: a case report","volume":"96","author":"Xu","year":"2017","journal-title":"Medicine (Baltimore)"},{"key":"R5-20250504","doi-asserted-by":"crossref","first-page":"674","DOI":"10.1016\/j.jbspin.2013.03.020","article-title":"Coexistence of seropositive rheumatoid arthritis and SAPHO syndrome","volume":"80","author":"Nissen","year":"2013","journal-title":"Joint Bone Spine"},{"key":"R6-20250504","doi-asserted-by":"crossref","first-page":"18026","DOI":"10.1038\/bdjteam.2018.26","article-title":"Facilitating timely diagnosis of Sj\u00f6gren's syndrome","volume":"5","author":"Douglas","year":"2018","journal-title":"BDJ Team"},{"key":"R7-20250504","doi-asserted-by":"crossref","first-page":"1023","DOI":"10.1093\/rheumatology\/kew015","article-title":"Synovitis, acne, pustulosis, hyperostosis and osteitis syndrome: a single centre study of a cohort of 164 patients","volume":"55","author":"Li","year":"2016","journal-title":"Rheumatology (Oxford)"},{"key":"R8-20250504","doi-asserted-by":"crossref","first-page":"333","DOI":"10.1016\/S0950-3579(94)80022-7","article-title":"The SAPHO syndrome","volume":"8","author":"Kahn","year":"1994","journal-title":"Baillieres Clin Rheumatol"},{"key":"R9-20250504","doi-asserted-by":"crossref","first-page":"554","DOI":"10.1136\/ard.61.6.554","article-title":"Classification criteria for Sj\u00f6gren's syndrome: a revised version of the European criteria proposed by the American-European Consensus Group","volume":"61","author":"Vitali","year":"2002","journal-title":"Ann Rheum Dis"},{"key":"R10-20250504","doi-asserted-by":"crossref","first-page":"475","DOI":"10.1002\/acr.21591","article-title":"American College of Rheumatology classification criteria for Sj\u00f6gren's syndrome: a data-driven, expert consensus approach in the Sj\u00f6gren's International Collaborative Clinical Alliance cohort","volume":"64","author":"Shiboski","year":"2012","journal-title":"Arthritis Care Res (Hoboken)"},{"key":"R11-20250504","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1002\/art.39859","article-title":"2016 American College of Rheumatology\/European League Against Rheumatism Classification Criteria for Primary Sj\u00f6gren's Syndrome: a Consensus and Data-Driven Methodology Involving Three International Patient Cohorts","volume":"69","author":"Shiboski","year":"2017","journal-title":"Arthritis Rheumatol"},{"key":"R12-20250504","doi-asserted-by":"crossref","first-page":"e000022","DOI":"10.1136\/rmdopen-2014-000022","article-title":"EULAR Sj\u00f6gren's syndrome disease activity index (ESSDAI): a user guide","volume":"1","author":"Seror","year":"2015","journal-title":"RMD Open"},{"key":"R13-20250504","first-page":"125","article-title":"Synovitis-acne-pustulosis-hyperostosis-osteitis syndrome and psoriatic arthritis exhibit a different immunogenetic profile","volume":"26","author":"Queiro","year":"2008","journal-title":"Clin Exp Rheumatol"},{"key":"R14-20250504","doi-asserted-by":"crossref","first-page":"20","DOI":"10.1186\/s13023-017-0573-9","article-title":"High prevalence of autoimmune disease in the rare inflammatory bone disorder sternocostoclavicular hyperostosis: survey of a Dutch cohort","volume":"12","author":"Valkema","year":"2017","journal-title":"Orphanet J Rare Dis"},{"key":"R15-20250504","doi-asserted-by":"crossref","first-page":"481","DOI":"10.1007\/s00296-008-0809-z","article-title":"Prevalence of rheumatic diseases and disability in China","volume":"29","author":"Xiang","year":"2009","journal-title":"Rheumatol Int"},{"key":"R16-20250504","doi-asserted-by":"crossref","first-page":"R17","DOI":"10.1186\/ar2368","article-title":"Rheumatic diseases in China","volume":"10","author":"Zeng","year":"2008","journal-title":"Arthritis Res Ther"},{"key":"R17-20250504","doi-asserted-by":"crossref","first-page":"191","DOI":"10.7150\/ijms.17718","article-title":"Reviewing primary Sj\u00f6gren's syndrome: beyond the dryness - From pathophysiology to diagnosis and treatment","volume":"14","author":"Both","year":"2017","journal-title":"Int J Med Sci"},{"key":"R18-20250504","doi-asserted-by":"crossref","first-page":"785705","DOI":"10.1155\/2015\/785705","article-title":"Serum IL-6 and IL-23 levels and their correlation with angiogenic cytokines and disease activity in ankylosing spondylitis, psoriatic arthritis, and SAPHO syndrome","volume":"2015","author":"Przepiera-B\u0119dzak","year":"2015","journal-title":"Mediators Inflamm"},{"key":"R19-20250504","doi-asserted-by":"crossref","first-page":"2898","DOI":"10.4049\/jimmunol.181.4.2898","article-title":"Identification of IL-18 and Th17 cells in salivary glands of patients with Sj\u00f6gren's syndrome, and amplification of IL-17-mediated secretion of inflammatory cytokines from salivary gland cells by IL-18","volume":"181","author":"Sakai","year":"2008","journal-title":"J Immunol"},{"key":"R20-20250504","doi-asserted-by":"crossref","first-page":"389","DOI":"10.3109\/08916934.2014.906582","article-title":"TH17 cells are increased in the peripheral blood of patients with SAPHO syndrome","volume":"47","author":"Firinu","year":"2014","journal-title":"Autoimmunity"},{"key":"R21-20250504","doi-asserted-by":"crossref","first-page":"665","DOI":"10.1002\/art.22087","article-title":"Conundrums in nosology: synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome and spondylarthritis","volume":"55","author":"Rohekar","year":"2006","journal-title":"Arthritis Rheum"},{"key":"R22-20250504","doi-asserted-by":"crossref","first-page":"383","DOI":"10.1016\/S1297-319X(02)00414-1","article-title":"Is the relationship between spondyloarthropathy and Sj\u00f6gren's syndrome in women coincidental? A study of 13 cases","volume":"69","author":"Scotto di Fazano","year":"2002","journal-title":"Joint Bone Spine"},{"key":"R23-20250504","first-page":"718","article-title":"Increased frequency of Sj\u00f6gren's syndrome in patients with spondyloarthropathy","volume":"25","author":"Brandt","year":"1998","journal-title":"J Rheumatol"},{"key":"R24-20250504","doi-asserted-by":"crossref","first-page":"173","DOI":"10.1007\/s10067-006-0255-9","article-title":"Sj\u00f6gren's syndrome in patients with ankylosing spondylitis","volume":"26","author":"Kobak","year":"2007","journal-title":"Clin Rheumatol"},{"key":"R25-20250504","article-title":"Ankylosing spondylitis coexists with rheumatoid arthritis and Sj\u00f6gren's syndrome: a case report with literature review","author":"Zhao","year":"2020","journal-title":"Clin Rheumatol"},{"key":"R26-20250504","doi-asserted-by":"crossref","first-page":"1164","DOI":"10.1093\/rheumatology\/keq047","article-title":"Articular manifestations in primary Sj\u00f6gren's syndrome: clinical significance and prognosis of 188 patients","volume":"49","author":"Fauchais","year":"2010","journal-title":"Rheumatology (Oxford, England)"},{"key":"R27-20250504","doi-asserted-by":"crossref","first-page":"9","DOI":"10.1016\/j.autrev.2018.06.015","article-title":"Arthritis in primary Sj\u00f6gren's syndrome: Characteristics, outcome and treatment from French multicenter retrospective study","volume":"18","author":"Mirouse","year":"2019","journal-title":"Autoimmun Rev"},{"key":"R28-20250504","doi-asserted-by":"crossref","first-page":"e59868","DOI":"10.1371\/journal.pone.0059868","article-title":"Serum levels of beta2-microglobulin and free light chains of immunoglobulins are associated with systemic disease activity in primary Sj\u00f6gren's syndrome. Data at enrollment in the prospective ASSESS cohort","volume":"8","author":"Gottenberg","year":"2013","journal-title":"PLoS One"},{"key":"R29-20250504","doi-asserted-by":"crossref","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 advances in musculoskeletal disease"},{"key":"R30-20250504","doi-asserted-by":"crossref","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 (Baltimore)"},{"key":"R31-20250504","doi-asserted-by":"crossref","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 Rheumat Dis"},{"key":"R32-20250504","doi-asserted-by":"crossref","first-page":"2814","DOI":"10.3389\/fimmu.2019.02814","article-title":"JAK inhibitors and oxidative stress control","volume":"10","author":"Charras","year":"2019","journal-title":"Front Immunol"},{"key":"R33-20250504","first-page":"2057","article-title":"JAK-1 inhibition suppresses interferon-induced BAFF Production in Human Salivary Gland: potential therapeutic strate for primary Sj\u00f6gren's syndrome","volume":"70","author":"Lee","year":"2018","journal-title":"Arth Rheumatol (Hoboken, NJ)"},{"key":"R34-20250504","doi-asserted-by":"crossref","first-page":"605","DOI":"10.1080\/13543784.2019.1631796","article-title":"Small-molecule inhibitors and the salivary gland epithelium in Sj\u00f6gren's syndrome","volume":"28","author":"Pringle","year":"2019","journal-title":"Expert opinion on investigational drugs"},{"key":"R35-20250504","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1136\/annrheumdis-2019-216114","article-title":"EULAR recommendations for the management of Sj\u00f6gren's syndrome with topical and systemic therapies","volume":"79","author":"Ramos-Casals","year":"2020","journal-title":"Ann Rheu Dis"},{"key":"R36-20250504","doi-asserted-by":"crossref","first-page":"1990","DOI":"10.3389\/fimmu.2019.01990","article-title":"Systematic review of safety and efficacy of rituximab in treating immune-mediated disorders","volume":"10","author":"Kaegi","year":"2019","journal-title":"Front Immunol"},{"key":"R37-20250504","doi-asserted-by":"crossref","first-page":"2235","DOI":"10.3899\/jrheum.121149","article-title":"Is there a role for rituximab in the treatment of spondyloarthritis and psoriatic arthritis?","volume":"39","author":"Mease","year":"2012","journal-title":"J Rheumatol"},{"key":"R38-20250504","doi-asserted-by":"crossref","first-page":"ii38","DOI":"10.1093\/rheumatology\/kew349","article-title":"New treatment targets for axial spondyloarthritis","volume":"55","author":"Sieper","year":"2016","journal-title":"Rheumatology (Oxford, England)"}],"container-title":["Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.lww.com\/10.1097\/MD.0000000000023940","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,5,4]],"date-time":"2025-05-04T06:27:09Z","timestamp":1746340029000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.lww.com\/10.1097\/MD.0000000000023940"}},"subtitle":["A retrospective observational study"],"short-title":[],"issued":{"date-parts":[[2021,3,26]]},"references-count":38,"journal-issue":{"issue":"12","published-online":{"date-parts":[[2021]]}},"URL":"https:\/\/doi.org\/10.1097\/md.0000000000023940","relation":{},"ISSN":["0025-7974","1536-5964"],"issn-type":[{"type":"print","value":"0025-7974"},{"type":"electronic","value":"1536-5964"}],"subject":[],"published":{"date-parts":[[2021,3,26]]},"assertion":[{"value":"2020-03-30","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-12-01","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}