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Cytomegalovirus (CMV) is the most common cause of congenital infection, with a wide spectrum of clinical manifestations and different grade of severity. We report the case of a male baby born at term with an early prenatal diagnosis of severe intracranial hemorrhage (ICH), with no other evident risk factors. Urine and blood sample were tested for CMV-DNA, and diagnosis of congenital CMV infection was established. This case describes intracranial hemorrhage as uncommon although possible sign of early fetal CMV infection. Considering that pathogenic factors cannot be defined in 25 % of term neonates with ICH, this case report highlights the importance of CMV screening in pregnant women and in term infants with prenatal ICH of unknown origin., Competing Interests: The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest., (© 2021 The Authors.)