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Introduction  Aortopulmonary collateral arteries are an uncommon variant of alternative blood supply in cases of complex congenital heart disease. Although surgery may still be the classic approach for this condition, mini-invasive endovascular occlusion has been recently attempted as an alternative less traumatic procedure. Children born to women with epilepsy are at increased risk of congenital malformations.    Case presentation  A cardiovascular malformation in a 6-year-old white boy with prenatal exposure to carbamazepine is described. At birth, he underwent atrial-ventricular septal defects repair. At 6 years of age, he was diagnosed to have an aberrant aortopulmonary artery from the descending aorta. He presented with recurrent respiratory infections and no cardiovascular signs, but there was associated right upper lobe hyperperfusion. Collateral percutaneous plug embolization was performed because of risk for cardiorespiratory infections, pulmonary hypertension and atrioventricular dilatation. The post-releasing control showed a complete occlusion of the aberrant artery. A chest radiogram and computed tomography showed normalization of vascular pattern of his right lung at 9-months follow-up. No complications and no respiratory infections in the first follow-up year were observed. A good growth gain was obtained.    Conclusions  Plug embolization in an aortopulmonary collateral artery is an interesting alternative to surgery and is suitable for children with minor congenital heart disease and without severe respiratory and/or cardiovascular symptoms. Management and long-term pediatric multidisciplinary follow-up is recommended. Prenatal exposure to carbamazepine could be considered in the pathogenesis and diagnosis of the malformation.

Aortopulmonary collateral artery in prenatal exposure to carbamazepine – endovascular therapy and technical considerations: a case report