Cerebral hyperperfusion syndrome (CHPS) is a rare disease with complications of carotid artery revascularization. Acute retinal hemorrhage is a very rare entity that has been previously described as a manifestation of CHPS followed by the carotid artery stenting (CAS), but to the best of our knowledge, not yet described as a complication of carotid surgery. An Ascending Aorta to bilateral Carotid bypass was performed in a 35 years old woman with active Takayasu arteritis and 95% symptomatic stenosis of both common carotid arteries. Severe retinal hemorrhage appeared on the second post operative day combined with high blood pressure, brain edema on CT scan and grand mal seizures. It seems that fundoscopic examination followed by the carotid revascularization of tightness can be helpful in identifying those patients who develop symptoms suggesting of CHPS.