Thirteen males with Leber's optic neuropathy had magnetic resonance imaging (MRI) of the brain, and in eight the optic nerves were imaged using STIR (Short Time Inversion Recovery) sequences. All optic nerve scans were abnormal. In seven with bilateral visual loss four showed bilateral increased optic nerve signal and three unilateral increase. The involvement was of the mid and posterior intra-orbital sections over three 5 mm slices or more with sparing of the anterior portion. One patient with unilateral visual loss had increased signal only on the affected side. Brain MRI was normal, in marked contrast to the findings in clinically isolated optic neuritis in which multiple white matter lesions are seen in the majority.