Pulsatile tinnitus associated with normal otoscopic findings may present as the only sign of a potentially life-threatening serious disease such as a dural arteriovenous fistula (dAVF) of the transverse or sigmoid sinus. If left untreated, dAVFs may lead to focal neurologic symptoms, intracranial hypertension, or hemorrhage, and finally to death. We report two cases of dAVF presenting with pulsatile tinnitus. Following diagnosis, both patients underwent transarterial embolization, after which their symptoms resolved.