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Tetralogy of Fallot (TOF) accounts for approximately 5% of all congenital heart disease. However, only 1% of patients with TOF survive to the age of 40 years without undergoing surgery. Additionally, the relationship between intracerebral hemorrhage and unrepaired TOF remains unknown. We report a rare case of unrepaired TOF in a patient who presented with intracerebral hemorrhage, and we also present a literature review.A 40-year-old man presented with headache and right-sided limb weakness.He was diagnosed with TOF approximately a year prior to presentation and did not undergo any definitive treatment or any symptomatic management. Head computed tomography revealed an intracerebral hematoma in the left basal ganglia. The patient was drowsy, and his blood oxygen saturation was 77%.Owing to his poor cardiopulmonary status, the patient did not undergo surgery and was treated with only symptomatic supportive therapy.After 2 days of therapy, his disturbance of consciousness and motor ability showed improvement.Literature reviews reveal that intracerebral hemorrhage is rarely observed in patients with TOF, and to date, only 3 cases have been reported. Furthermore, this patient was 40 years old and did not undergo cardiac surgery. Severe hypoxia, as well as low levels of platelets and coagulation factors in the blood could have led to intracerebral hemorrhage.