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The authors report the successful use of Descemet's stripping automated endothelial keratoplasty (DSAEK) to treat a 45-year-old woman with amantadine-associated corneal edema. Discontinuation of the medication and treatment with corticosteroids did not result in resolution of the edema. The patient underwent sequential phakic DSAEK in both eyes with significant anatomic, visual, and functional improvement. Histopathologic analysis of Descemet's membrane by light microscopy revealed a paucity of endothelial cells. This case highlights the importance of considering amantadine toxicity in the differential diagnosis of corneal edema without an identifiable ocular cause and suggests the utility of DSAEK in the treatment of this rare condition., (Copyright 2010, SLACK Incorporated.)

Purpose: To describe the clinical features of 3 patients with amantadine-associated corneal edema, including the histopathologic findings from 1 patient who underwent corneal transplantation for irreversible corneal edema., Design: Interventional case series., Participants: Three patients who sought treatment at the authors' institution with abrupt-onset, bilateral, diffuse corneal edema associated with systemic amantadine use., Methods: Retrospective chart review., Main Outcome Measures: Visual acuity, corneal thickness, slit-lamp observations, and histopathologic findings., Results: The duration of use of amantadine ranged from 2 months to 6 years before onset of corneal edema. Discontinuation of amantadine resulted in resolution of corneal edema in both eyes of 2 patients. A third patient underwent a full-thickness corneal transplantation, and subsequently, edema developed in the grafted cornea. Cessation of amantadine therapy in this patient resulted in resolution of corneal edema in both eyes, but the ungrafted corneal eventually decompensated and became edematous, requiring corneal transplantation. Histopathologic analysis of the cornea buttons showed significant loss of endothelial cells., Conclusions: Amantadine can cause corneal edema that begins a few months to several years after institution of therapy, and the edema can occur even in a corneal graft. Prolonged corneal edema in the setting of amantadine use can be irreversible. In cases of corneal edema without an obvious causative disease, the systemic medication list of the patient must be reviewed, and amantadine must be considered as a possible cause.