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Background: Anterolateral defect of diaphragm is unusual and has been reported less than 5 times in literature.Case Presentation: We are reporting a case with typical initial presentation of a congenital diaphragmatic hernia. A chest x-ray raised suspicion of a cystic lesion or diaphragmatic eventration which was excluded by chest ultrasound. At operation he was found to have a localised and large anterolateral defect with a very thick sac which was plicated to bridge the diaphragmatic defect.Conclusion: A rare variant and should be differentiated from other variants using imaging.

A newborn with anterolateral diaphragmatic defect: An unusual anomaly?