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Aortopulmonary window is a rare congenital cardiac anomaly characterized by communication between the aorta and the pulmonary artery above the semilunar valves. Prenatal diagnosis is rare. We report four fetuses with aortopulmonary window and review the relevant literature. Approximately half of the reported cases had additional cardiac defects. None had chromosomal abnormalities. In cases with normal cardiac connections, the diagnosis can be made prenatally on the standard three-vessel view, as seen in two of our cases. In one fetus with complete transposition of the great arteries, the diagnosis was made retrospectively on sagittal views. In the remaining case, the window was seen postnatally but could not be identified retrospectively due to the abnormal superoinferior relationship of the ventricles and vessels. Copyright © 2016 ISUOG. Published by John Wiley & Sons Ltd.

SEAs were underestimated in children due to the rarity and spectrum of differential diagnoses. Timely diagnosis, immediate antibiotics, spinal magnetic resonance imaging and prompt neurosurgical consultations were essential for favourable outcomes.