Congenital intestinal hypoganglionosis: A radiologic mimic of Hirschsprung's disease.

Enteric neuropathy Hirschsprung's disease Intestinal hypoganglionosis

Journal

Radiology case reports
ISSN: 1930-0433
Titre abrégé: Radiol Case Rep
Pays: Netherlands
ID NLM: 101467888

Informations de publication

Date de publication:
Feb 2019
Historique:
received: 04 09 2018
revised: 10 10 2018
accepted: 13 10 2018
entrez: 13 11 2018
pubmed: 13 11 2018
medline: 13 11 2018
Statut: epublish

Résumé

Intestinal hypoganglionosis or isolated hypoganglionosis is a rare entity with a clinical and radiologic presentation that can mimic Hirschsprung's disease in the neonatal period. The diagnosis of this entity can be challenging with suction rectal biopsies that are standard for diagnosing Hirschsprung's disease. We present this case of congenital intestinal hypoganglionosis detailing the neonatal course, due to its rarity and the conundrums faced before an eventual diagnosis could be rendered. This case also illustrates the role of full thickness rectal biopsy in selected cases such as ours where the radiologic features are typical of Hirschsprung's, despite negative suction biopsies.

Identifiants

pubmed: 30416639
doi: 10.1016/j.radcr.2018.10.007
pii: S1930-0433(18)30421-7
pmc: PMC6222261
doi:

Types de publication

Case Reports

Langues

eng

Pagination

171-174

Références

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pubmed: 23943250
Semin Pediatr Surg. 1998 Aug;7(3):187-91
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Pediatr Surg Int. 2010 Nov;26(11):1111-5
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Auteurs

Gayathri Sreedher (G)

Department of Radiology, Akron Childrens Hospital, Akron, OH, USA.

Aaron Garrison (A)

Department of Surgery, University of Cincinnati, Cincinnati Children's hospital, OH, USA.

Robert Novak (R)

Department of Pathology, Akron Childrens Hospital, Akron, OH, USA.

Matthew Keisling (M)

Department of Pathology, Akron Childrens Hospital, Akron, OH, USA.

Shankar Srinivas Ganapathy (SS)

Department of Radiology, Akron Childrens Hospital, Akron, OH, USA.

Classifications MeSH