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Muneer Fazea,1,2 Mansour Alhameli,2,3 Faisal Ahmed,4 Mohammad Reza Askarpour,5 Wafa Murshed,2 Azizh Jarwsh,2 Amal Alkbous2 1Department of Radiology, School of Medicine, Ibb University of Medical Sciences, Ibb, Yemen; 2Department of Radiology, Al-Ma’amon Diagnostic Center, Sana’a, Yemen; 3Department of Radiology, Faculty of Medicine, Sana’a University of Medical Sciences, Sana’a, Yemen; 4Urology Research Center, Al-Thora General Hospital, Department of Urology, School of Medicine, Ibb University of Medical Sciences, Ibb, Yemen; 5Department of Urology, School of Medicine, Shiraz University of Medical Sciences, Shiraz, IranCorrespondence: Faisal Ahmed, Al-Thora General Hospital, Alodine Street, Ibb, Yemen, Email fmaaa2006@yahoo.comAbstract: Pentalogy of Cantrell is a congenital anomaly of the median mesodermal constructions with a poor prognosis. It is characterized by defects of the anterior diaphragm, the lower sternum, the abdominal wall, pericardium, and various congenital heart malformations. We present a case of ectopia cordis and Pentalogy of Cantrell in a newborn of a healthy 35-year-old woman with no history of embryotoxic exposure or smoking. The infant was first diagnosed with the anomaly in the second trimester of pregnancy and was delivered at 35 weeks of gestational age via a caesarian section. Shortly after birth, he was transferred to the neonatal intensive care unit (NICU) due to progressive respiratory failure, which ultimately, along with septicemia, led to infant death on the second day. In conclusion, the Pentalogy of Cantrell should be appropriately assessed for effective prenatal counseling and postnatal management with a multidisciplinary team; since infant survival assessment and early diagnosis give the parents the option of terminating the pregnancy.Keywords: case report, Pentalogy of Cantrell, ectopia cordis