Introduction: Most of infected infants presenting with Cytomegalovirus (CMV) remain asymptomatic1. Symptoms include growth restriction, microcephaly, seizures, cerebral ventriculomegaly, chorioretin is, hepatitis syndrome, thrombocytopenia, anemia, and pneumonitis2. Approximately 5 to 10% of these infants manifest signs of serious neurological defects at birth, including deafness, mental retardation, blindness, microcephaly, hydrocephalus, and cerebral calcification3,4. In addition, 10 to 15% of congenitally infected infants who are asymptomatic at birth subsequently develop brain disorders such as sensorineural hearing loss.5 One case of congenital CMV disease with developmental delay and leukoencephalopathy has been reported here.