| Summary: | Purpose: We report the case of an 11-year-old boy with posterior microphthalmos who exhibited normal and age appropriate development of visual acuity. Observations: At the initial diagnosis, when he was 3 years old, the best-corrected visual acuity (BCVA) was 20/125 in the right eye (OD) and 20/200 in the left eye (OS) with high hyperopia (cycloplegic refraction +15.75 D sphere OD and +16.25 D sphere OS). Eight years after he began wearing hyperopic glasses, BCVA was 20/16 OD and 20/20 OS. Optical coherence tomography did not reveal a foveal pit in either eye throughout the observation period. However, elongation of the outer segment and widening of the outer nuclear layers were observed. Conclusion and Importance: Many cases of posterior microphthalmos demonstrate subnormal BCVA due to an abnormal foveal structure (papillomacular retinal folds, absence of the foveal pit and avascular zone) and high hyperopia. However, if foveal maturity progresses, even if the foveal structure is abnormal, early aggressive amblyopia treatment can result in normal and age appropriate development of visual acuity. Keywords: Posterior microphthalmos, Fovea, Hypoplasia, Amblyopia, OCT, Hyperopia
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