{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T12:22:38Z","timestamp":1756383758075,"version":"3.37.3"},"reference-count":17,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2021,12,27]],"date-time":"2021-12-27T00:00:00Z","timestamp":1640563200000},"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":[[2022,6,24]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Cogan\u2019s syndrome (CS) is a rare autoimmune vasculitis of unknown aetiology characterised by non-syphilitic interstitial keratitis, audiovestibular symptoms, sometimes systemic symptoms, and multi-organ involvement. Atypical CS has other ocular features, such as scleritis, episcelritis, retinitis, and optic neuritis. Diagnosis of CS is purely clinical without a confirmatory test. Hereby, we report a case of atypical CS presenting with features of encephalitis who was treated successfully with intravenous pulse methylprednisolone with cyclophosphamide. It is important to consider CS in the differential diagnosis of encephalitis with ocular and vestibular symptoms in young patients, as high morbidity and mortality rates are effectively lowered by early immunosuppressive treatment.<\/jats:p>","DOI":"10.1093\/mrcr\/rxab055","type":"journal-article","created":{"date-parts":[[2021,12,27]],"date-time":"2021-12-27T09:00:28Z","timestamp":1640595628000},"page":"305-308","source":"Crossref","is-referenced-by-count":6,"title":["A rare case of atypical Cogan\u2019s syndrome presenting as encephalitis"],"prefix":"10.1093","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8093-8303","authenticated-orcid":false,"given":"Debashis","family":"Maikap","sequence":"first","affiliation":[{"name":"Department of Clinical Immunology and Rheumatology, Kalinga Institute of Medical Sciences, KIIT University , Bhubaneswar, Odisha, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amrita","family":"Pradhan","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, S.C.B. Medical College , Cuttack, Odisha, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2545-9032","authenticated-orcid":false,"given":"Prasanta","family":"Padhan","sequence":"additional","affiliation":[{"name":"Department of Clinical Immunology and Rheumatology, Kalinga Institute of Medical Sciences, KIIT University , Bhubaneswar, Odisha, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2021,12,27]]},"reference":[{"key":"2022062410203618300_R1","doi-asserted-by":"crossref","first-page":"1007","DOI":"10.1093\/rheumatology\/keh228","article-title":"Typical and atypical Cogan\u2019s syndrome: 32 cases and review of the literature","volume":"43","author":"Grasland","year":"2004","journal-title":"Rheumatology"},{"key":"2022062410203618300_R2","doi-asserted-by":"crossref","first-page":"549","DOI":"10.1016\/0002-9343(76)90722-1","article-title":"Cogan\u2019s syndrome: a systemic vasculitis","volume":"60","author":"Cheson","year":"1976","journal-title":"Am J Med"},{"key":"2022062410203618300_R3","first-page":"913","article-title":"Manifestations cardiovasculaires du syndrome de Cogan. 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Cogan syndrome in children","volume":"125","author":"Pollard","year":"2007","journal-title":"Arch Ophthalmol"},{"key":"2022062410203618300_R11","doi-asserted-by":"crossref","first-page":"396","DOI":"10.1016\/j.autrev.2012.07.012","article-title":"Cogan\u2019s syndrome: an autoimmune inner ear disease","volume":"12","author":"Greco","year":"2013","journal-title":"Autoimmun Rev"},{"key":"2022062410203618300_R12","doi-asserted-by":"crossref","first-page":"1219","DOI":"10.1016\/j.autrev.2017.10.005","article-title":"Cogan syndrome: characteristics, outcome and treatment in a French nationwide retrospective study and literature review","volume":"16","author":"Durtette","year":"2017","journal-title":"Autoimmun Rev"},{"key":"2022062410203618300_R13","doi-asserted-by":"crossref","DOI":"10.1155\/2018\/5798103","article-title":"Audiovestibular symptoms in systemic autoimmune diseases","volume":"2018","author":"Ralli","year":"2018","journal-title":"J Immunol 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