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To illustrate a rare cause of out-of-hospital cardiac arrest in children, its differential diagnoses, emergency and subsequent treatment at various steps in the rescue chain, and potential outcomes.A 4-year-old boy with unknown agenesis of the left coronary ostium sustained out-of-hospital cardiac arrest. Bystander cardio-pulmonary resuscitation was initiated and defibrillation was performed via an automated external defibrillator (AED) shortly after paramedics arrived at the scene, restoring sinus rhythm and spontaneous circulation. After admission to the intensive care unit the child was intubated for airway and seizure control. Further diagnostic work-up by angiography revealed agenesis of the left coronary artery. After initial seizures, the boy's neurological recovery was complete. He subsequently underwent successful internal mammary artery in-situ bypass surgery to the trunk of the left coronary artery. One year after cardiac arrest, the patient had completely recovered with no physical or intellectual sequelae. A catheter examination proved excellent growth of the bypass and good cardiac function.This case illustrates the long term outcome after agenesis of the LCA while reiterating that prompt access to pediatric defibrillation may be lifesaving—albeit in a minority of pediatric OHCA.