Colonic atresia and hirschsprung disease: a case report and review of the literature
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. Hirsch...
| Main Authors: | , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
BMC
2023-06-01
|
| Series: | Journal of Medical Case Reports |
| Subjects: | |
| Online Access: | https://doi.org/10.1186/s13256-023-03969-z |
| _version_ | 1797806748028895232 |
|---|---|
| author | Amirhossein Ladan Reza Mahdian Jouybari Mahnaz Zareh Akbari Pegah Moharrami Yeganeh |
| author_facet | Amirhossein Ladan Reza Mahdian Jouybari Mahnaz Zareh Akbari Pegah Moharrami Yeganeh |
| author_sort | Amirhossein Ladan |
| collection | DOAJ |
| description | 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. |
| first_indexed | 2024-03-13T06:12:00Z |
| format | Article |
| id | doaj.art-f3ad5a2396144618b7a2ee86d6d90ec1 |
| institution | Directory Open Access Journal |
| issn | 1752-1947 |
| language | English |
| last_indexed | 2024-03-13T06:12:00Z |
| publishDate | 2023-06-01 |
| publisher | BMC |
| record_format | Article |
| series | Journal of Medical Case Reports |
| spelling | doaj.art-f3ad5a2396144618b7a2ee86d6d90ec12023-06-11T11:15:12ZengBMCJournal of Medical Case Reports1752-19472023-06-011711510.1186/s13256-023-03969-zColonic atresia and hirschsprung disease: a case report and review of the literatureAmirhossein Ladan0Reza Mahdian Jouybari1Mahnaz Zareh Akbari2Pegah Moharrami Yeganeh3Department of Surgery, Ayatollah Mousavi Hospital, Zanjan University of Medical SciencesDepartment of Pediatrics, Ayatollah Mousavi Hospital, Zanjan University of Medical SciencesDepartment of Pediatrics, Ayatollah Mousavi Hospital, Zanjan University of Medical SciencesSchool of Medicine, Zanjan University of Medical SciencesAbstract 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.https://doi.org/10.1186/s13256-023-03969-zColon atresiaHirschsprung diseaseCase reportNewbornNeontology |
| spellingShingle | Amirhossein Ladan Reza Mahdian Jouybari Mahnaz Zareh Akbari Pegah Moharrami Yeganeh Colonic atresia and hirschsprung disease: a case report and review of the literature Journal of Medical Case Reports Colon atresia Hirschsprung disease Case report Newborn Neontology |
| title | Colonic atresia and hirschsprung disease: a case report and review of the literature |
| title_full | Colonic atresia and hirschsprung disease: a case report and review of the literature |
| title_fullStr | Colonic atresia and hirschsprung disease: a case report and review of the literature |
| title_full_unstemmed | Colonic atresia and hirschsprung disease: a case report and review of the literature |
| title_short | Colonic atresia and hirschsprung disease: a case report and review of the literature |
| title_sort | colonic atresia and hirschsprung disease a case report and review of the literature |
| topic | Colon atresia Hirschsprung disease Case report Newborn Neontology |
| url | https://doi.org/10.1186/s13256-023-03969-z |
| work_keys_str_mv | AT amirhosseinladan colonicatresiaandhirschsprungdiseaseacasereportandreviewoftheliterature AT rezamahdianjouybari colonicatresiaandhirschsprungdiseaseacasereportandreviewoftheliterature AT mahnazzarehakbari colonicatresiaandhirschsprungdiseaseacasereportandreviewoftheliterature AT pegahmoharramiyeganeh colonicatresiaandhirschsprungdiseaseacasereportandreviewoftheliterature |