Neurosensory hearing loss is a well-known complication of antenatally acquired cytomegalovirus (CMV) infection. We here report an infant who developed auditory neuropathy after a postnatally acquired CMV infection. Infection probably occurred through ingestion of infected breast milk. Following a cochlear implant, there is normal language perception and a mildly delayed language expression at age 4. We speculate that the long-term effects of perinatal CMV infections are more dependent on the postconceptional age at which infection occurs than on whether the infection occurs antenatally or postnatally. An early acquired neonatal CMV infection in very preterm infants may therefore have long-term neurological sequelae, including auditory deficits.