The authors present two cases with irreversible visual loss and vitelliform macular lesions after prolonged deferoxamine treatment. Iron overload from repeated blood transfusions can lead to cardiomyopathy, hepatic cirrhosis, diabetes, and/or hypoparathyroidism.(1) Deferoxamine (Desferal; Novartis Pharmaceuticals Corp., East Hanover, NJ) methylselate, an iron chelating agent, can reduce these complications, but ocular side effects, including cataracts, optic neuropathy, and retinopathy, can occur.(1-6) Retinal findings include pigment mottling in the macula and equatorial regions, bulls-eye maculopathy, and cystoid macular edema.(1-6) Central loss of transparency and hypopigmentation of the outer retina/retinal pigment epithelium (RPE) have also been reported in cases of acute toxicity.(1) Visual deficits usually recover after cessation of the medication, but irreversible visual loss has been reported.(3,5) Mehta et al(2) described a patient on chronic deferoxamine therapy with color vision disturbance and macular pigmentary mottling. Subsequent to their published report, this patient developed bilateral vitelliform lesions, a finding that has not previously been reported with deferoxamine retinal toxicity. We report these new findings and report a second patient with deferoxamine retinopathy. Both patients had permanent visual loss and bilateral vitelliform macular lesions.