Diffusion tensor imaging (DTI) is an MRI technique that can evaluate white matter integrity and neuronal connectivity. Normal brain maturation and myelination are associated with reduced water diffusion and increased diffusion anisotropy. The authors aimed to evaluate the anterior optic pathways with DTI in 10 children with unilateral amblyopia, five children with bilateral amblyopia and 10 control subjects. The mean age was 10.8±2.8 years for unilateral amblyopia, 11.5±1.5 years for bilateral amblyopia and 13.8±2.6 years for controls. In unilateral amblyopia eyes and fellow eyes, fractional anisotropy (FA) values were significantly reduced in prechiasmatic regions. Mean diffusivity (MD) values were greater than controls. In bilateral amblyopia, prechiasmatic FA values were lower but not significantly so. MD values were greater in all anatomic regions but were not significant. The findings may indicate a functional underdevelopment of the anterior visual pathways in patients with unilateral amblyopia affecting both eyes.