Herpes simplex virus 1 is a prevalent neurotropic pathogen that infects and establishes latency in peripheral sensory neurons. It can migrate into the central nervous system and cause encephalitis. The association between herpes simplex virus encephalitis and cerebral venous thrombosis is rare, with a very limited number of case reports described in the literature, despite the recognized thrombogenic effects of the virus. A 44-year-old man was brought to the emergency department with generalized tonic-clonic seizures requiring sedation and ventilation to control it. Initial brain computed tomography revealed cortical and subcortical edema on the left frontal lobe, and a subsequent contrast-enhanced exam showed absence of venous flow over the anterior half of the superior sagittal sinus. Cerebrospinal fluid polymerase chain reaction was positive for herpes simplex virus type 1, and the patient was started on acyclovir and anticoagulation, with clinical improvement. Acyclovir administration was maintained for 14 days and oral anticoagulation for one year, with no recurrence of thrombotic events or other complications. A well-timed treatment has a validated prognostic impact on herpes simplex encephalitis, making early recognition of its clinical aspects of main importance.