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A 1.4-kilogram, male infant was born at 27 weeks gestation at an outside hospital. After birth, the patient's trachea was intubated and surfactant administered. Initial echocardiogram showed a patent ductus arteriosus (PDA) and a ventricular septal defect (VSD), with a possible aberrant vessel arising from the carotid artery. The patient was transferred to our hospital for further management. Repeat echocardiogram showed a right aortic arch with the left common carotid/innominate artery arising from the pulmonary artery. The infant was subsequently brought to the operating room where following sternotomy, the aberrant left innominate artery was reimplanted into the aorta. The origin of the left carotid artery or innominate artery from the pulmonary artery is an extremely rare form of congenital heart disease with a limited number of reports in the literature. The differential pressure gradient between the systemic and pulmonary beds may lead to a steal phenomenon, with shunting of blood from the carotid system into the pulmonary vascular bed, resulting in decreased cerebral blood flow. The left-to-right shunting from the systemic to pulmonary bed may also lead to volume overload of the left side of the heart. In the perioperative care of such patients, control of physiologic factors that control the balance between the pulmonary and systemic vascular resistance is mandatory to ensure that cerebral blood flow is maintained.