{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T23:10:27Z","timestamp":1784675427425,"version":"3.55.0"},"reference-count":29,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2018,11,12]],"date-time":"2018-11-12T00:00:00Z","timestamp":1541980800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"Assistance Publique- H\u00f4pitaux de Paris (D\u00e9l\u00e9gation \u00e0 la Recherche Clinique et \u00e0 l'Innovation), French Ministry of Health","award":["AOM09123 - NI08014 - IDR-CB 2010-A00512-37"],"award-info":[{"award-number":["AOM09123 - NI08014 - IDR-CB 2010-A00512-37"]}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Acta Ophthalmologica"],"published-print":{"date-parts":[[2019,6]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Purpose<\/jats:title><jats:p>To better characterize IgG4\u2010related disease (RD) in the setting of idiopathic orbital inflammation syndrome (IOIS).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>National, multicentre, prospective, observational cohort study. Among the patients consecutively included in the French multicentre <jats:italic>SIOI<\/jats:italic> cohort, we selected those who underwent orbital and\/or adnexal biopsy. Clinical, morphological and pathological findings at diagnosis were blindly analysed. Serum IgG4 levels at inclusion were measured and all available biopsy specimens were immunostained for IgG4 and IgG. Biopsy samples with more than 10 IgG4\u2010positive plasma cells per high\u2010power field and a IgG4+\/IgG+ plasma cell ratio above 40% were scored as positive. IgG4\u2010positive patients were then screened for comprehensive diagnostic criteria for IgG4\u2010RD.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of the 87 patients included, 35 had histologically documented IOIS. Thirteen patients (37%) with a mean age at onset of 27\u00a0years (range 21\u201378) had IgG4\u2010positive biopsies, among which 10 patients (77%) and 3 (23%, with IgG4 serum levels &gt;1.35\u00a0g\/L) were considered as having probable and definite IgG4\u2010RD, respectively. The latter 13 patients more frequently fulfilled histological criteria for IgG4\u2010RD (including plasmacytic infiltrate (p\u00a0=\u00a00.006), fibrosis (p\u00a0=\u00a00.0025) and periphlebitis (p\u00a0=\u00a00.075)) than IgG4\u2010negative patients. Storiform fibrosis was exclusively found in orbital tissues from IgG4\u2010positive patients (<jats:italic>n<\/jats:italic>\u00a0=\u00a03, 23%). Eosinophilia associated with recurrent sinusitis or asthma was a prominent feature in patients with definite IgG4\u2010RD.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>More than one\u2010third of patients with biopsy\u2010proven IOIS satisfied criteria for IgG4\u2010RD, but only a few had a definite type.<\/jats:p><\/jats:sec>","DOI":"10.1111\/aos.13968","type":"journal-article","created":{"date-parts":[[2018,11,13]],"date-time":"2018-11-13T05:46:07Z","timestamp":1542087967000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":17,"title":["IgG4\u2010related disease in patients with idiopathic orbital inflammation syndrome: data from the French <i>SIOI<\/i> prospective cohort"],"prefix":"10.1111","volume":"97","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6401-7088","authenticated-orcid":false,"given":"S\u00e9bastien","family":"Abad","sequence":"first","affiliation":[{"name":"Service de 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Paris Cit\u00e9 Universit\u00e9 Paris 13 Bobigny France"},{"name":"Service d'Anatomopathologie AP\u2010HP H\u00f4pital Avicenne Bobigny France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9183-4235","authenticated-orcid":false,"given":"Fr\u00e9d\u00e9ric","family":"Mouriaux","sequence":"additional","affiliation":[{"name":"Service d'Ophthalmologie Centre Hospitalier Universitaire de Rennes Rennes France"},{"name":"Facult\u00e9 de M\u00e9decine Universit\u00e9 Rennes 1 Rennes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emmanuel","family":"H\u00e9ron","sequence":"additional","affiliation":[{"name":"Service de M\u00e9decine Interne H\u00f4pital des Quinze\/vingts Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Neila","family":"S\u00e9dira","sequence":"additional","affiliation":[{"name":"Service de M\u00e9decine Interne H\u00f4pital des Quinze\/vingts Paris 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