Cytomegalovirus infection remains the main congenital infectious cause of abnormal development, notably neurological or auditory. In case of early maternal infection, vertical transmission is lower than later in pregnancy, but fetal/neonatal sequelae are more frequent and severe. Until recently, there was no available treatment to prevent transmission and complications and only preventive measures were recommended. Based on a recent literature review, we will discuss the possible indication for CMV screening before conception and/or in the first trimester of pregnancy, in order to improve patient's information, prevention and treatment.