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    Cogan's syndrome: present and future directions.

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    Authors
    Murphy, Grainne
    Sullivan, Miriam O
    Shanahan, Fergus
    Harney, Sinead
    Molloy, Michael
    Affiliation
    Department of Rheumatology, Cork University Hospital, Wilton, Cork, Ireland. grainne927@yahoo.com
    Issue Date
    2009-08
    MeSH
    Adrenal Cortex Hormones
    Adult
    Azathioprine
    Dose-Response Relationship, Drug
    Fatal Outcome
    Female
    Hearing Loss, Sensorineural
    Humans
    Keratitis
    Practice Guidelines as Topic
    Prognosis
    Randomized Controlled Trials as Topic
    Syndrome
    Uveitis
    Vascular Diseases
    Show allShow less
    
    Metadata
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    Citation
    Cogan's syndrome: present and future directions. 2009, 29 (10):1117-21 Rheumatol. Int.
    Journal
    Rheumatology international
    URI
    http://hdl.handle.net/10147/200965
    DOI
    10.1007/s00296-009-0945-0
    PubMed ID
    19471934
    Abstract
    Cogan's syndrome, typified by the combination of interstitial keratitis and immune-mediated sensorineural hearing loss, is a rare condition, and commonly associated with a diagnostic delay. Using a standard search protocol, we review the literature to date, focusing on a number of key areas pertaining to diagnosis, presentation and treatment. Using a case illustration of atypical disease which led to fulminant aortic regurgitation, we highlight the need for continued and collaborative research in order to identify negative prognostic factors and thus tailor therapeutic regimens. Atypical Cogan's syndrome is more commonly associated with systemic manifestations than typical disease, and may be refractory to immunosuppressive treatment. We discuss the application of laboratory (e.g antibodies targeting inner ear antigens) and radiological (PET-CT) aids to disease confirmation and detection of sub-clinical vascular inflammation. As illustrated by the included case description, some patients remain refractory to intense immunosuppression and delineation of adverse prognostic factors which may direct treatment, perhaps including the use of PET-CT, will contribute in the future to improving patient outcomes.
    Item Type
    Article
    Language
    en
    ISSN
    1437-160X
    ae974a485f413a2113503eed53cd6c53
    10.1007/s00296-009-0945-0
    Scopus Count
    Collections
    Cork University Hospital

    entitlement

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