Cerebral venous and dural sinus thrombosis (CVDST) is a rare but not uncommon life-threatening disease accounting for less than 1% of stroke, affecting people of any age group, predominantly the extremes of age. CVDST carries a variable prognosis: although the majority of cases are associated with complete recovery, outcome may be fatal in less than 10% of patients. Thrombolysis has been widely used in CVDST in patients with rapidly deteriorating symptoms who fail to improve despite adequate anticoagulation, but the exact therapeutic time window remains unclear. We report a case of CVDST with rapid clinical deterioration despite therapeutic anticoagulation treated with direct endovascular thrombolysis with intravenous recombinant tissue plasminogen activator 1 week after presentation with complete recovery, and we present a review of the literature on the efficacy and safety of thrombolysis in CVDST.