Cerebral venous sinus thrombosis (CVST) is a rare cause of stroke. We report the case of a patient with acute CVST, showing perfusion abnormalities on computed tomography perfusion with perfusion defect in the relevant hemisphere. The defect was found in a region adjacent to the occluded sinus and was not corresponding to an arterial territory. To the best of our knowledge this is the first ever report on CT perfusion abnormalities few hours after acute symptom onset in CVST.