Lymphocytic choriomeningitis virus is a rodent-borne virus, whch is an uncommon cause of congenital infection. Affected infants present with brain and eye lesions, including periventricular calcifications, neuronal migration defects, and chorioretinitis. Lymphocytic choriomeningitis virus mimics the presentation of congenital cytomegalovirus, and it should be considered in symptomatic infants whose cytomegalovirus testing is negative.