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Background: Congenital segmental intestinal dilatation (CSID) is a rare cause of neonatal bowel obstruction. Patients can present in the early newborn period with feeding intolerance and abdominal distension. Diagnosis is challenging due to its rarity and non-specific clinical and radiological findings in a well neonate. Surgical excision remains the mainstay treatment for this disease. Case presentation: We report a case of a newborn male with congenital segmental ileal dilatation, who presented with abdominal distension and feeding intolerance. He was born preterm at 32 weeks and was treated for presumed sepsis due to a premature rupture of the membrane. The patient developed persistent abdominal distension upon initiation of feedings with large amount of gastric aspirates. Otherwise, there was no delay in the passage of meconium. Serial abdominal radiographs showed persistent focal bowel dilatation on the right side. He was initially diagnosed with small bowel atresia. Ultrasound abdomen and lower gastrointestinal contrasted study showed focal ileal dilatation with no evidence of pneumoperitoneum. Intraoperative findings revealed a segmental ileal dilatation with no evidence of mechanical obstruction. The abnormal dilated bowel was excised and sent for histopathological examination. He recovered well after surgery. Conclusion: Diagnosis of CSID can be challenging due to its clinical polymorphism and non-specific radiological findings. CSID should be suspected in neonates with feeding intolerance and focal intestinal dilatation without clinical features of sepsis, peritonitis, or mechanical obstruction. Surgical excision is curative with a good outcome. [ABSTRACT FROM AUTHOR]