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Dawit Seyoum Gebremariam,1 Asmamaw Miruts,2 Kibrom Gebreselassie Desta3 1Department of Pediatrics and Child Health, College of Health Sciences, Mekelle University, Mekelle, Tigray, Ethiopia; 2Department Surgery, Pediatric Surgery Unit, College of Health Sciences, Mekelle University, Mekelle, Tigray, Ethiopia; 3Department of Surgery, Cardiothoracic Unit, College of Health Sciences, Mekelle University, Mekelle, Tigray, EthiopiaCorrespondence: Dawit Seyoum Gebremariam, Department of Pediatrics and Child Health, College of Health Sciences, Mekelle University, P.O. Box: 1871, Mekelle, Tigray, Ethiopia, Tel +251911731454, Email dgseyoum@yahoo.comAbstract: Sternal cleft is a rare chest wall anomaly resulting from a failure of the lateral mesodermal folds to migrate to the midline, causing a cleft in the early stage of embryological development. This can be a complete or partial defect. It can also occur as an isolated anomaly or in association with other syndromes. Fetal sonographic diagnosis of this defect is possible, but less practiced. After birth, this defect can be easily diagnosed clinically because of the presence of paradoxical chest wall movement. The flexibility of the thorax is maximal and compression of the underlying structures is minimal during the neonatal and early infancy period, and this period is the preferred time for surgical repair. We report a 39-day-old infant who presented with an isolated V-shaped inferior sternal cleft, its surgical primary closure, and postoperative course.Keywords: sternal cleft, surgery, congenital anomaly, complication