Purpose: To assess the clinical features, hearing loss and neurodevelopmental outcomes of infants with congenital cytomegalovirus (CMV) infection and to discover significant brain magnetic resonance imaging (MRI) findings that predict poor neurodevelopmental outcomes. Methods: The medical records of 31 infants who were diagnosed with congenital CMV infection at the Neonatal Intensive Care Unit of Asan Medical Center between 2002 and 2012 after CMV was isolated from their urine within the second

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... week of their life were retrospectively reviewed. The long-term neurodevelopmental outcomes of the infants were monitored using the Bayley Scale of Infant Development II, the Korean Infants' Development Screening Test and the Wechsler Scale of Intelligence, as appropriate. Results: The infants' mean gestational age was 36.3±3.2 weeks and their mean birth weight was 2,395±715 g. Microcephaly were detected in three of them (9.7%), and petechiae and hepatosplenomegaly were detected in one (3.2%). Ten infants (32.2%) and nine infants (29.0%) showed preterm and intrauterine growth retardation, respectively. Of the 27 infants with whom long-term follow-up was possible, six (22.2%) showed developmental disabilities. The brain MRI findings, which included ventriculomegaly, periventricular calcification, polymicrogyria, microcephaly and cerebellar hypoplasia, were correlated with the poor neurodevelopmental outcomes, but no correlation was found between the presence of periventricular cysts and that of white matter disease. Of the infants who survived, six (20.0%) had sensorineural hearing loss at the median age of 30.6 months. Conclusion: Congenital cytomegalovirus infection is one of the most frequent causes of intrauterine viral fetal infection and need to be distinguished if congenital infection is suspected. On the follow up observation, the hearing loss was observed in 20% of the patients. A long term neurological observation is required for the patients who had unusual impression on MRI.