A 58-year-old woman presented with acute onset of global aphasia. Imaging studies revealed a left frontotemporal enhancing tumor and ischemic stroke in the territory of the middle cerebral artery. The patient was operated on, and the diagnosis of glioblastoma multiforme was confirmed. At the time of surgery, several branches of the left middle cerebral artery were found embedded in the tumor. One branch, which was infiltrated by tumor and completely occluded, was resected to achieve complete resection. Postoperatively, the stroke area within the middle cerebral artery territory increased, together with worsening of the patient’s clinical status, thus requiring urgent decompressive craniectomy. Thereafter, the patient gradually improved, and received radiation therapy and chemotherapy with no recurrence after 24months of follow-up. To our knowledge, glioblastomas presenting with ischemic stroke are rare, and such patients should be considered to be at high surgical risk.