Horseshoe Lung Associated With Holt-Oram Syndrome

Horseshoe lung is an uncommon congenital malformation in which the bases of the right and the left lungs are fused to each other by a narrow isthmus posterior to the cardiac apex (1). There have been 52 case reports in the English literature. Holt-Oram syndrome is characterized by cardiac septation defects and pre-axial radial ray abnormalities (2). To our knowledge, it is the first report of horseshoe lung combined with Holt-Oram syndrome. An 18-month-old boy was admitted to our hospital because of discovered heart murmur at local hospital. He was born to a G1P1, 25-year-old mother at 39 weeks’ gestation at a local hospital. The pregnancy and prenatal history were uneventful. He was naturally delivered with a birth weight of 3400 g, and at admission he had a weight of 10 kg and height of 83 cm. He has mild respiratory distress since he was born but without cyanosis. There was no family history of congenital anomalies. Physical examination revealed a sloping left shoulder, compressed left thorax, malformed left upper-extremity and asymmetric face when crying. Tachypnea, intercostal retractions, decreased breathing sounds over the left side of the chest, and a systolic murmur grade 3/6 over lower left sternal border were noted. Malformations of the left upper arm included inward twisting of left wrist and absence of left thumb.