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A male infant was referred to us at 11 days of life because of cardiac murmur and tachypnoea. Echocardiography at presentation and cardiac catheterisation at 20 days of life revealed a right sided double lumen aortic arch with hypoplastic fourth arch and stenotic fifth arch (left panel), aberrant retro-oesophageal innominate artery (middle panel) with a remnant of a left arterial duct (white arrowhead), and ventricular septal defect. The diameter of the fourth arch was 5.0 mm and the origin of the fifth arch was 6.4 mm, but the diameter of stenosis of the fifth arch was only 1.2 mm with a string-like blood flow through it. The infant showed coarctation physiology with a pressure gradient of 28 mm Hg between the ascending and descending aorta. Because the infant continued to show signs of congestive heart failure, he underwent successful balloon dilation of the stenotic fifth aortic arch at 32 days old. Retrogradely we dilated the stenosis repeatedly using two different sized balloons, 3 mm and 5 mm in diameter, respectively (Cordis, Miami Lakes, Florida, USA). After these procedures, the diameter of stenosis increased to 4.1 mm and the pressure gradient disappeared (right panel). The patient was discharged three days after these procedures and has been doing well without any sign of decompensation. This case shows that balloon dilation of the fifth aortic arch is a safe and effective alternative to surgery as a treatment for double lumen aortic arch with hypoplastic fourth aortic arch and stenotic fifth aortic arch in infants.