Thirty-five infants with cystic parenchymal lesions detected by ultrasonography during the neonatal period were evaluated via periodic neurologic and developmental assessments. Long-term neurologic status (abnormal, normal, and isolated motor deficit) was assigned at 2 years of age. Abnormal neurologic status was strongly associated with bilateral cystic lesions as well as hydrocephalus during the neonatal period. Microcephaly at 6-8 months of age also correlated with abnormal status at 2 years of age. The neurologic exam at discharge and at 6-8 months of age added further negative predictive value for the infants with isolated motor delays. A literature review was conducted to determine the incidence of motor and mental delays in a large series of infants with cystic intracranial lesions.