The pathway of the vein of hypoglossal canal has not been mentioned in venous origin pulsatile tinnitus. We wished to clarify the possible complications related to this venous variant. We described 2 rare cases of troublesome pulsatile tinnitus associated with prominent vein of hypoglossal canal, which communicates with the jugular bulb and the marginal sinus. Both cases were successfully treated by positioning a stent across the vein of hypoglossal canal and jugular bulb. The 2 present clinical cases represent the first report of such a condition. The therapeutic decision-making is discussed in relation to the persistent pulsatile tinnitus and the etiopathologic hypothesis put forward.