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Mixed pial-dural arteriovenous malformation (MpdAVM) and dural arteriovenous fistula (dAVF) are rare entities in the anterior cranial fossa (ACF). As dural-pial vascular anastomosis can exist near the cribriform plate, MpdAVM with a small nidus, which cannot be apparently identified, can be logically indistinguishable from dAVF in ACF. A 71-year-old man was referred for evaluation of possible intracranial vascular malformation. Cerebral angiography revealed an arteriovenous shunt in the ACF. The lesion was fed by the bilateral ethmoidal arteries and right orbitofrontal artery, draining through the bilateral cortical veins to the superior sagittal sinus. As a nidus was not detected, dAVF was suspected. Venous interruption was planned with direct surgery. Intraoperatively, an arterial aggregation was observed in the right frontal lobe. The arterial aggregation seemed to be connected to the interrupted drainer in the right ACF. The arterial aggregation was removed and pathologically diagnosed as arteriovenous malformation. Postoperatively, intracerebral hemorrhage was confirmed, and postoperative cerebral angiography confirmed the resolved arteriovenous shunt. The intracranial hemorrhage was possibly due to the timing gap between drainer interruption and removal of the nidus. MpdAVM with a small nidus in the ACF can mimic dAVF. Clinicians must be aware that an unremoved nidus of MpdAVM may postoperatively result in fatal intracranial hemorrhage.

Mixed pial-dural arteriovenous malformation in the anterior cranial fossa mimicking dural arteriovenous fistula