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

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 Hirsc...

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Detalles Bibliográficos
Autores principales: Sreedher, Gayathri, Garrison, Aaron, Novak, Robert, Keisling, Matthew, Ganapathy, Shankar Srinivas
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6222261/
https://www.ncbi.nlm.nih.gov/pubmed/30416639
http://dx.doi.org/10.1016/j.radcr.2018.10.007
Descripción
Sumario: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.