Background- Herpes Simplex Virus type-1 is commonly associated with oral mucosal infection but, in the last few years, the HSV-1 genital infection has been increasing in prevalence, even more than HSV-2 in several populations. The risk of a maternal-fetal transmission in pregnant women with genital infection is cause of big concerns for neonatologists. Genital herpes is frequently subclinical and could be easily transmitted to the fetus during pregnancy or at birth. Intrauterine HSV infection is very dangerous because it is usually followed by catastrophic consequences (classic triad: cutaneous, ophthalmologic and neurologic involvement). The early diagnosis and treatment are significant in modifying outcomes lifelong. Case presentation- We report a Caucasian female neonate, born by cesarean section, from a perinatally asymptomatic mother, who presented with skin macular rash on third day of life, followed by an intermittent fever up to 38,4°C. Because of an increased C Reactive Protein (CRP) up to 130 g/l associated with thrombocytopenia (37.000/mcl) and mild hyperexcitability at examination, a lumbar puncture was performed. The Polymerase Chain Reaction (PCR) revealed the presence of HSV type 1 DNA in CSF and Aciclovir iv, at 60 mg/kg per day, was started. Serological investigations revealed HSV1 IgG and IgM positivity in the mother’s blood and IgG positivity in the daughter’s sample. That means that the baby most likely received virus and maternal antibody through placenta in the last period of pregnancy. No history of maternal HSV infection. A clinical follow up at 2 years of age revealed no signs of motor, cognitive or language impairment. Conclusion- In conclusion, we surely could make several important considerations: neonatal HSV infections are not always associated with a positive maternal clinical history; cesarean section cannot always prevent maternal-fetal transmission; congenital herpes is not necessarily associated with neonatal vesicular lesions and can manifest without the classic triad at birth; finally, early diagnosis and early therapy are associated with an excellent outcome.