Delayed cerebral ischemia (DCI) remains a devastating complication in subarachnoid hemorrhage (SAH), however, there were no present reports that is associated with a ruptured spinal arteriovenous fistula (sAVF). We would like to present a rare case of DCI following embolization of a ruptured perimedullary sAVF. Initially, the patient clinical symptoms mimic a SAH caused by a ruptured intracranial aneurysm. Further evaluation revealed that the SAH was caused by a ruptured perimedullary sAVF and the patient’s condition improved following the embolization procedure. Three days later, the patient developed an acute left-sided facial and motor weakness, which persisted until the patient was discharged on the day-15 onset. A magnetic resonance imaging and angiography is performed 1.5 years after discharge and revealed no signs of cerebral infarction and hemorrhage. In this paper, we reported DCI after embolization in a ruptured sAVF with SAH, supported by evidence from the current literature. We would like to also stress the importance of complete spinal and cerebral vessel imaging to reveal the underlying abnormalities and determine the most appropriate intervention.