INTRODUCTION: Severe visual deprivation during infancy can lead to long-term changes in ocular development, including significant differences in axial length (AL) between eyes. This case report presents three adult patients with monocular infantile visual deprivation who developed substantial AL interocular differences. The aim is to explore the impact of early visual deprivation on AL and its potential implications for myopia progression. CASE REPORT: Three male patients, aged 19, 42, and 50, were evaluated at the Ophthalmology Department of the University Hospital of Naples "Federico II." Each patient had a history of severe visual deprivation in one eye during infancy. The first patient experienced a dense corneal fibrovascular membrane from a chemical burn at age 1. The second suffered total corneal stromal scarring from a chemical burn at age 2. The third developed dense leukoma following infective keratitis at 0.3 years. Ophthalmological evaluations included refractive assessment, biomicroscopy, fundus examination (where feasible), electroretinogram (ERG), and A-Scan ultrasound. Affected eyes exhibited severe visual impairment (light perception in two patients, and 1.6 logMAR in the third), while the fundus could not be fully explored, and echographic assessment was used to exclude major chorioretinal changes. ERG findings were within normal limits, despite presumed early developmental impact on the retina due to lack of form vision. However, AL measurements revealed a 5.8-6.3 mm increase in the affected eyes compared to the healthy ones. CONCLUSION: Infantile severe visual deprivation can lead to significant interocular AL differences in adulthood. These findings underscore the importance of investigating the underlying mechanisms to better understand and manage myopia progression in similar cases.