| Summary: | Introduction: The combination of a patent omphalomesenteric duct in the setting of an omphalocele is rare and has an unusual physical appearance on presentation. Case presentation: We share the case of a neonate who presented with omphalocele and a concurrent patent omphalomesenteric duct with intestinal prolapse. We report the early diagnosis and operative management of this presentation with intestinal resection and anastomosis, and primary closure of the omphalocele. The patient had no other abnormalities and after uncomplicated recovery, was able to discharge uneventfully tolerating oral feeds. Conclusion: Because the initial appearance of a patent omphalomesenteric duct in an omphalocele is unusual with visible bowel mucosa and an intact omphalocele membrane, this variation of an abdominal wall defect is at risk of being misdiagnosed as a ruptured omphalocele, gastroschisis, or another complex abdominal wall defect. Expeditious surgical management is indicated.
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