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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.

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