The current recommended treatment for cerebral venous sinus thrombosis (CVST) is anticoagulation, and the presence of intracranial hemorrhage (ICH) is not a contraindication. We present a case of ICH associated with CVST in which heparin treatment was associated with rebleeding, and we review current evidence of anticoagulation safety in patients with ICH associated with CVST. A 65-year-old man presented with right hemiparesis and loss of consciousness. Brain computed tomography showed a left frontoparietal hemorrhage. Angiographic studies with magnetic resonance imaging showed the presence of a partial superior saggital sinus thrombosis. With a diagnosis of CVST, intravenous heparin was administered. After 24 hours the patient had a symptomatic increase in ICH size, and 2 days later the patient developed a status epilepticus with new evidence of rebleeding. Anticoagulant treatment was stopped and the patient experienced neurological improvement, with no new episodes of rebleeding. Evidence for the safety of anticoagulants in CVST comes from 2 small trials involving a total of 79 patients, but only 18 had some degree of bleeding in baseline computed tomography. A meta-analysis suggested that in CVST patients who are treated with anticoagulants, the risk of ICH is low, but acknowledged that an impact of up to 9% of new ICH cannot be ruled out. As there is not enough evidence for the safety of anticoagulant therapy in patients with early ICH associated with CVST, the therapeutic decision must be individualized and the rebleeding risk should be weighed in those patients.