Risk of cerebrovascular disease is increased in patients with HIV infection; however, cerebral venous sinus thrombosis is not commonly reported in this condition. We report a case of young man with intracranial hemorrhage whose radiologic investigations revealed thrombosis of the left transverse and sigmoid sinuses extending into the left internal jugular vein. On further investigations, he was found to be positive for HIV-1, and his procoagulant workup showed protein S deficiency. He was started on anticoagulant therapy, after which he showed gradual recovery. This case highlights a rarely encountered presentation of HIV infection as cerebral venous sinus thrombosis.