AbstractThe full range of neurological manifestations of novel coronavirus (severe acute respiratory syndrome coronavirus 2 [SARS-CoV-2]) infection in children remains incompletely characterized. Here, we discussed a 7-year-old male child patient with sickle cell disease (SCD), who presented with acute encephalopathy and nonconvulsive (electrographic only) seizures immediately prior to the onset of severe symptomatic SARS-CoV-2 infection, manifesting as respiratory failure, systemic inflammatory response, and hypertension. Brain imaging confirmed hyperintense lesions consistent with posterior reversible encephalopathy syndrome (PRES). Following aggressive symptomatic management including antiseizure medication, immunomodulatory treatment of SARS-CoV-2 infection, and intensive blood pressure control, he made a full neurological recovery. PRES has been observed in adults with SARS-CoV-2 infection, but there are few published reports of this neurological manifestation in children. Our case demonstrates that PRES should be a consideration in children with SARS-CoV-2 infection presenting with acute neurological decompensation, especially in the setting of preexisting risk factors for cerebrovascular dysregulation such as SCD.