A Case of Atypical Cogan Syndrome with Late-Onset Corneal Involvement.

Kim, Sang Jin; Han, Young Keun

Case Report

Journal of the Korean Ophthalmological Society 2004;45(2):310-314.
Published online February 1, 2004.

A Case of Atypical Cogan Syndrome with Late-Onset Corneal Involvement.

¹Department of ophthalmology, Seoul National University College of Medicine, Korea.
²Seoul Artificial Eye Center, Clinical Research Institute, Seoul National University Hospital, Korea.
³Department of ophthalmology, Seoul Municipal Boramae Hospital, Korea. eye129@netian.com

말기에 각막을 침범한 비전형적 Cogan 증후군 1예

김상진 ( Kim Sang Jin ) , 한영근 ( Han Yeong Geun )

Abstract

PURPOSE
To report a case of atypical Cogan syndrome with late-onset corneal involvement. METHODS: Bilateral conjunctival injection with severe headache developed in a 38-years old woman who had suffered from tinnitus, vertigo and hearing difficulty. Inflammation in anterior chamber and vitreous cavity, macular edema, disc edema and retinal hemorrhages were found on ocular examination. RESULTS: After initiation of systemic steroid therapy, ocular inflammatory reaction and visual acuity improved, but hearing difficulty did not respond. After 7 years of lost follow-up, corneal stromal opacity occurred and she was diagnosed as atypical Cogan syndrome. Elevated ESR, multiple stenosis in renal and lower extremity arteries in angiography were found. CONCLUSIONS: Atypical Cogan syndrome with late-onset corneal involvement is rare and difficult to diagnose, which may delay in initiation of adequate treatment.

Key Words: Atypical Cogan syndrome;Late-onset corneal involvement