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The current article reviews the current literature related to congenital cytomegalovirus (CMV)-related hearing loss. The discussion will focus on the epidemiology, pathogenesis, and clinical presentation of human CMV infection as it pertains to hearing loss. Current methods of CMV diagnosis with a focus on the evolving trend toward broader neonatal screening protocols will also be explored. Discussion of medical, surgical, and audiologic management of the condition will also be addressed.Much of the current research on this topic is focused on improving detection of CMV through screening programs. Some advances in understanding cochlear pathogenesis have also been made.Congenital CMV infection remains an important cause of hearing loss in infants and children. Early detection of CMV infection can broaden treatment options and allow for improved hearing and language outcome for patients with CMV-associated sensorineural hearing loss.