Objective: The aim of this report is to describe a clinical case of an anomalous origin of the left pulmonary artery branch from the ascending aorta, and to present a current literature review of this rare disease.

Clinical Case: A 2 year-old infant was referred to our institution for surgical correction of sub aortic obstruction. The non invasive investigation disclosed a right aortic arch anatomy with left ventricle outflow tract obstruction due to sub aortic membrane associated but failed to show the anomalous origin of the left pulmonary artery branch from the ascending aorta. The diagnosis was made intraoperatively and the patient underwent a total surgical correction of the defects, including simultaneous anastomosis of the left pulmonary artery to the pulmonary trunk. Results of the surgical repair of this case were successful. CONCLUSIONS. Although anomalous origin of the left pulmonary artery from the ascending aorta is a rare disease which was not diagnosed appropriately, successful results can be obtained when opportune and fortunate total surgical corrections are performed.

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