A 40-year-old woman was referred to intensive care unit after recurrent ventricular fibrillation. She was free of cardiac medical history or medications. The resting ECG displayed an extended early repolarization in the inferior leads and all the precordial leads. Incessant ventricular fibrillations justified a treatment by intravenous amiodarone associated with general anaesthesia and mechanical ventilation without success on ventricular fibrillation. Because of a low heart rate intravenous isoproterenol infusion was initiated. Isoproterenol infusion was associated with heart rate acceleration and a decrease in J point elevation and the disappearance of ventricular fibrillation episodes. No cardiac disease was documented and the patient was implanted of a single chamber ICD. Six months later the patient was free of syncope and ventricular fibrillation on ICD memory. This case report demonstrates the usefulness and efficiency of the isoproterenol infusion to eliminate recurrent ventricular fibrillation in patients with early repolarization.