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Although several of the typical anomalies are diagnosable by ultrasound <jats:italic>in utero<\/jats:italic> (hypertelorism, median cleft lip, anterior cephalocele), very few cases have been reported prenatally, the present being only the third. In the present case, hemimegalencephaly is first reported among the anomalies possibly associated with frontonasal dysplasia. The diagnosis was made at 22\u2009weeks' gestation and was confirmed by necropsy following termination of pregnancy. 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