Cerebral sinus thrombosis is a rare cause of spontaneous subarachnoid hemorrhage. The development of cerebral sinus thrombosis as a complication of heparin-induced thrombocytopenia is even rarer. In this paper, we present a 59-year-old patient admitted to our service with cerebral sinus thrombosis secondary to type II heparin-induced thrombocytopenia. We also review the literature in regard to the incidence, pathophysiology and management of this rare clinicopathological entity.