We describe the occurrence of cerebral hyperperfusion syndrome (CHS) in a case of long-standing carotid-jugular fistula (CJF) treated by endovascular reconstruction of the carotid artery. A 43-year-old male with a high-flow CJF between the internal carotid artery (ICA) and internal jugular vein underwent endovascular reconstruction of the carotid artery using a stent graft. After treatment, the patient developed CHS. The patient succumbed to a large intracranial bleed in the left external capsule and parietal lobe on the fifth postoperative day. CHS following endovascular reconstruction of carotid artery is rare. We present the first reported case of CHS following endovascular reconstruction of ICA. A review of literature for patients treated by endovascular rerouting of blood flow to the cerebral parenchyma associated with hyperperfusion syndrome has been performed.