Infants with congenital cytomegalovirus (CMV) infection were identified through urine cultures of 15,212 consecutive neonates and studied prospectively to determine whether their neurodevelopmental and audiologic status was different from that of matched uninfected control subjects. Of 64 children with congenital CMV infection, three died, 11 could not be located for follow-up, one had quadriplegic cerebral palsy, and seven had varying degrees of sensorineural hearing loss. All matched control subjects were normal neurologically, and none of them had sensorineural hearing impairment. The Stanford-Binet test revealed scores within the normal range, at 3 and 5 years of age, for both children with CMV infection and matched control subjects, as did the preschool assessment (Wide Range Achievement Test) in children older than 5 years. However, in children with CMV infection, the home environment was less stimulating, discipline and punishment were more readily implemented, and behavioral problems were significantly greater than in the matched control subjects.