We report a case of dural arteriovenous malformation (dAVM) in the anterior cranial fossa presenting with intracranial hemorrhage. The patient was a 60-year-old man who became unconscious shortly after developing an acute headache. Computed tomography performed on admission demonstrated a left frontal intracranial hematoma together with subarachnoid hemorrhage and a subdural hematoma. Cerebral angiography revealed a dAVM in the left anterior cranial fossa supplied by the anterior ethmoidal arteries bilaterally from enlarged ophthalmic arteries and which drained into the superior sagittal sinus via a dilated cortical vein with aneurysmal dilatation. The lesion was successfully resected via a left frontal craniotomy. Although reports of dAVMs in the anterior fossa are rare, a number of new cases have been described recently. The clinical features and angiographic findings in 58 cases are analyzed. The average age at the time the patients presented was 55.8 years, and males predominated (85.0%). Intracranial hemorrhage was the presenting manifestation in most cases (74.1%). Ocular symptoms occurred especially in cases with a posterior venous drainage pattern. Angiographically, the dAVM was most frequently supplied by the anterior ethmoidal artery (94.8%) and drained into a pial vein with vascular sac in almost every case. Rupture of this varicose dilatation often caused intracranial hemorrhage. The main drainage sinus or vein was usually the superior sagittal sinus (86.0%). The nidus was usually in the dura mater, but recently a nidus in the brain parenchyma has also been reported. The pathogenesis of this entity remains unclear, but some impairment of normal embryonic vascular development is the most likely etiology. Surgical resection remains the treatment of choice.