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Periventricular leukomalacia (PVL) is coagulation necrosis of white matter seen in preterm, low birth weight (LBW) infants and associated with a range of ocular manifestations. We report a case of 2½ months old (47 weeks gestational age) female infant with PVL associated with bilateral high myopia (−16 DS). On examination, child had difficulty in tracking objects and inferior field defect with preference for superior gaze. She was prescribed spectacles and visual stimulation exercises to preserve existing vision and prevent amblyopia. Periventricular leukomalacia can affect full-term infants although it is more common in preterm and LBW infants. All preterm, LBW infants, and those with positive neuroradiological findings must be assessed by an ophthalmologist as early as possible to detect the associated ocular manifestations.