Profound hypotony maculopathy in a first episode of bilateral idiopathic acute anterior uveitis

BACKGROUND We report a case of a HLA-B27 negative patient presenting with severe, bilateral, idiopathic acute anterior uveitis with acute hypotony and hypotony maculopathy as their first uveitic episode. CASE PRESENTATION Within a week of onset of her first episode of acute anterior uveitis, a 45 year-old Caucasian lady developed profound ocular hypotony with unrecordable intraocular pressures, reduced vision and choroidal folds. All investigations were negative. Uveitic hypotony responded slowly to corticosteroids--intravenous, oral and topical--with normalization of intraocular pressure and resolution of choroidal folds after two months. Anterior uveitis and hypotony have not returned with six months of follow-up. CONCLUSION Bilateral, profound hypotony maculopathy may present acutely in idiopathic acute anterior uveitis, may be slow to respond to treatment and should be considered as a cause of vision loss in patients with this condition.