This report describes a rare case of anomalous origin of the right coronary artery from the pulmonary artery associated with a large aortopulmonary window in a 2-month-old boy. The right coronary artery was exposed to systemic pressure and carried fairly well-oxygenated blood to the myocardium. Closure of the aortopulmonary window alone could have caused acute myocardial ischemia. The purpose of this case report is to describe successful diagnosis and management of anomalous origin of the right coronary artery associated with an aortopulmonary window. The pathological findings and the physiological effects, clinical importance, and method of correction used are discussed.
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