Journal of Medical Case Reports (Jun 2023)

Colonic atresia and hirschsprung disease: a case report and review of the literature

  • Amirhossein Ladan,
  • Reza Mahdian Jouybari,
  • Mahnaz Zareh Akbari,
  • Pegah Moharrami Yeganeh

DOI
https://doi.org/10.1186/s13256-023-03969-z
Journal volume & issue
Vol. 17, no. 1
pp. 1 – 5

Abstract

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Abstract Background Colon atresia is one of the rarest congenital anomalies of the gastrointestinal tract, with an incident range of between 1 in 10,000 and 66,000 live births. Type I colonic atresia affects only the mucosal layer of the intestine and spares the intestinal wall and mesentery. Hirschsprung Disease is a rare association of Colon atresia and is usually diagnosed as a complication of atresia treatment. Case presentation This study reports a 14-h term white middle-eastern female infant with type I transverse colonic atresia complicated by the association of Hirschsprung disease and provides a brief literature review of the topic. She presented with poor feeding, weakness, and failure to pass meconium, and her abdominal X-ray showed complete distal bowel obstruction. The presence of Hirschsprung disease was realized after complications of atresia surgery. The infant underwent a total of three surgeries involving an end-to-end anastomosis of the atresia, colostomy formation following anastomosis leakage, and Hirschsprung surgery. The patient ultimately expired. Conclusions The association between colonic atresia and Hirschsprung disease poses a diagnostic and therapeutic challenge. Considering Hirschsprung disease as a possible association in colon atresia patients can facilitate proper decision-making in the course of treating colon atresia cases and achieving better outcomes.

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