Cogan's syndrome is a rare idiopathic inflammatory disease with involvement of the eye and inner ear. This syndrome can be associated with aortic root vasculitis. We report the case of a young man with a history of uveitis and bilateral hearing loss who presented with proximal aortic vasculitis and dilatation after aortic valve replacement. Cogan's syndrome was diagnosed on the basis of large artery vasculitis with typical vestibuloauditory and ocular involvement. The patient underwent successful a Bentall operation after aortic valve replacement.
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