Objectives: This study aimed to evaluate the macular pigment optical density (MPOD) scores in eyes with hyperopic anisometropic amblyopia and compare those with their fellow and control eyes using one-wavelength fundus reflectometry. Methods: This cross-sectional study enrolled 33 patients diagnosed with hyperopic anisometropic amblyopia aged 12–40 years. The control group consisted of 36 hyperopic and 42 emmetropic children, age-matched to the patients. Central macular thickness (CMT), MPOD, axial length (AL), best-corrected visual acuity, and refraction errors were measured between the study group and the control group. Results: Eyes with the diagnosis of hyperopic anisometropic amblyopia had significantly higher mean and maximum (max) MPOD scores compared with their fellow eyes as well as hyperopic and emmetropic eyes (p<0.001 for all). The mean AL in eyes with hyperopic anisometropic amblyopia was statistically shorter than that in hyperopic and emmetropic controls (p=0.027, p<0.001, respectively). The mean CMT was found to be thicker in eyes with hyperopic anisometropic amblyopia when compared to their fellow eyes, as well as hyperopic and emmetropic controls, eventhough there was no significant difference was found among the four groups (p=0.052). The mean MPOD levels were significantly correlated with the difference in CMT (r=−0.21, p=0.032), and logMAR visual acuity scores (r=−0.44, p<0.001) in the hyperopic anisometropic amblyopia group. Conclusion: The present study indicates that the MPOD is reduced in eyes with hyperopic anisometropic amblyopia. This reduction may be due to less visual stimulus-induced deterioration of foveal development and microarchitecture in anisometropic amblyopic eyes.