AbstractNeonatal encephalitis by either enteroviruses (EVs) or parechoviruses (PeVs) is often complicated by hemispheric periventricular white matter lesions. Although showing many similarities, the two types of encephalitis differ in some clinical and laboratory aspects, mostly because PeV encephalitis does not show any change of protein and white cell content in the cerebrospinal fluid, and clinically, the onset of PeV encephalitis is often marked by early seizures accompanying a fever of sepsis-like disease. Instead, no difference exists relative to the white matter lesions, which are constantly described as periventricular, even in rare detailed neuropathological studies. Herein, taking a cue from a neonate with EV encephalitis who showed occipital white matter lesions involving the overlying cortex, but completely sparing the periventricular area, we demonstrate that the brain lesions in EV encephalitis in newborns can be more extended than known. To our knowledge, the not-periventricular involvement of the white matter with EV encephalitis has never been described so far, as well as the potential of EV to injure the cortex. We confirm the pathogenetic role of a vascular mechanism for the tissue injury, but other proposed mechanisms are also discussed. It is noteworthy that the neurological outcome of this newborn remained favorable, and no epileptic seizures occurred in the first few days nor afterward.