A prospective follow-up study was conducted of a cohort of 65 at-risk neonates to assess the predictive value of neonatal cranial ultrasound abnormalities for cerebral visual impairment in infancy. Visual function was assessed using the acuity card procedure and behavioural visual responses were tested. Normal visual function was found in controls as well as in infants with small haemorrhages or mild leukomalacia. Infants with large haemorrhages performed poorly at 40 weeks postmenstrual age, but recovered to within the normal range in the first half year. In contrast, of the infants with extensive cystic leukomalacia, four of nine preterm infants and three of four term infants were severely visually impaired at 18 months. In the more mature infants, the cystic lesions extended deeper into the subcortical white matter, and this appeared to be associated with a worse visual outcome. Cystic leukomalacia proved to be highly predictive of cerebral visual impairment in infancy.