A 33-year-old man presented with consciousness disturbance (Glasgow Coma Scale score 7) and right hemiplegia after suffering headache persisting for 10 days. Head computed tomography revealed an irregular intracerebral hematoma in the left temporoparietal region, associated with a tubular high density area compatible with a thrombosed transcerebral vein in the left temporal lobe. The patient was free of coagulopathy. Craniectomy was performed to remove the intracerebral hematoma and venous thrombosis was confirmed. Postoperative cerebral angiography demonstrated extensive venous malformation in the left parietal and occipital lobes. Multiple transcerebral draining veins converged in the vein of Galen associated with a varix. Segmental narrowing of the straight sinus was suggestive of congestion in the venous anomaly. The patient showed progressive recovery following surgery, and was discharged with moderate aphasia, mild right hemiparesis, and right homonymous hemianopsia 1 month later. Venous malformations are usually silent, but occasionally become symptomatic due to thrombosis of the draining vein. The presence of stenosis in the draining route may lead to venous congestion, thrombus formation, and catastrophic hemorrhagic venous infarct.