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To determine the frequency of congenital cytomegalovirus (CMV) infection in infants born to human immunodeficiency virus (HIV)-infected mothers and assess risk factors that may facilitate intrauterine transmission of CMV, including the role of perinatal HIV infection.Retrospective cohort study of infants who were born to HIV-infected mothers at Parkland Memorial Hospital and screened for congenital CMV infection according to a standard nursery protocol between February 1, 1997 and May 31, 2005.During the 8-year study period that included 125,781 live births, there were 367 infants (0.3%) born to 303 HIV-infected mothers. Of 333 HIV-exposed infants who were screened for CMV, 10 (3%) had congenital CMV infection and 6 (60%) of these were identified only because of the CMV screening protocol. Four (1%) infants were infected with HIV, and none of these was CMV-infected. Compared with CMV-uninfected infants, CMV-infected, HIV-exposed newborns had lower mean birth weight (2508 versus 3148 g, P < 0.01), lower gestational age (37 vs. 39 weeks, P < 0.01), and higher median maternal HIV viral load at the start of prenatal care (15,411 vs. 2209 copies/mL, P = 0.02). CMV-infected infants were more likely to be born to mothers who were diagnosed with HIV during the pregnancy or at delivery (P = 0.03).The prevalence of congenital CMV infection among HIV-exposed newborns was 3%. Screening of these infants for CMV would allow identification of infants who are at risk for delayed onset of hearing loss and other neurodevelopmental impairment.