The persistent primitive hypoglossal artery (PPHA) is the second most common persistent carotid-vertebrobasilar anastomosis. We present a rare case of an 82-year-old woman who diagnosed as acute cerebral infarction three times in both the anterior and posterior circulation territories with a right-sided PPHA and ipsilateral carotid artery dissection (CAD). Microembolus monitoring results suggested that microembolic caused by CAD associated with PPHA should be recognized as a possible cause of multiple infarctions in both the anterior and posterior circulation territories. For these patients, appropriate treatment measures should be taken for prevention of stroke recurrence.