Introduction: Combination of congenital diaphragmatic hernia (CDH) and anorectal malformation (ARM) is rare. In this report, we describe a combination of imperforate anus and CDH in one of twins who conceived by Assisted Reproductive Technology (ART).

Case Report: A female preterm newborn at 27 weeks of gestation was referred to our neonatal intensive care unit due to respiratory distress. She was conceived by ART and had combination of imperforate anus and CDH. She expired 18-hours after birth as a result of severe lung hemorrhage and there was no possibility of surgical repair due to poor clinical condition.

Conclusions: In spite of the low incidence of birth defects in the ART-conceived babies, continuing surveillance of them is necessary and the report of their birth defects is helpful.