Cerebral sinus venous thrombosis (CSVT) is a rare form of venous thromboembolism. Recurrent transient ischemic attacks were extremely rare clinical manifestation of CSVT. We reported a case of transient ischemic attack as an exceptionally rare form of CSVT. A 29-year-old woman presented with a headache and four episodes of a transient right limb weakness, numbness, and dysphasia lasting approximately 15 mins over the course of 2 weeks. Magnetic resonance venography revealed a superior sagittal sinus thrombosis with numerous prominent enlarged and dilated venous. Magnetic resonance imaging revealed venous infarction of the left frontal lobe. Transient ischemic attack-like episodes stopped with anticoagulation. We suggested that the patients with transient ischemic attack and symptoms of increased intracranial pressure should be carefully investigated in terms of CSVT.