endothelin receptor type B enteric nervous system enteric neurons hirschsprung disease single-cell RNA sequencing

Journal

Frontiers in cell and developmental biology
ISSN: 2296-634X
Titre abrégé: Front Cell Dev Biol
Pays: Switzerland
ID NLM: 101630250

Informations de publication

Date de publication:
2022
Historique:
received: 10 04 2022
accepted: 06 07 2022
entrez: 12 8 2022
pubmed: 13 8 2022
medline: 13 8 2022
Statut: epublish

Résumé

Hirschsprung disease is most often characterized by aganglionosis limited to the distal colon and rectum, and mice lacking the Endothelin receptor type B (Ednrb) faithfully recapitulate this phenotype. However, despite the presence of enteric ganglia in the small intestine, both human patients and Ednrb-/- mice suffer from dysmotility and altered gastrointestinal function, thus raising the possibility of enteric nervous system (ENS) abnormalities proximal to the aganglionic region. We undertook the present study to determine whether abnormalities with the ENS in ganglionated regions may account for abnormal gastrointestinal function. We performed single-cell RNA sequencing on ENS cells from the small intestine of Ednrb-/- mice and compared the results to a published single-cell dataset. Our results identified a missing population of neurons marked by the enzyme Gad2, which catalyzes the production of

Identifiants

pubmed: 35959491
doi: 10.3389/fcell.2022.917243
pii: 917243
pmc: PMC9360620
doi:

Types de publication

Journal Article

Langues

eng

Pagination

917243

Subventions

Organisme : NIDDK NIH HHS
ID : R01 DK119210
Pays : United States

Informations de copyright

Copyright © 2022 Bhave, Guyer, Picard, Omer, Hotta and Goldstein.

Déclaration de conflit d'intérêts

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

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Auteurs

Sukhada Bhave (S)

Department of Pediatric Surgery, Massachusetts General Hospital, Harvard Medical School, Boston, MA, United States.

Richard A Guyer (RA)

Department of Pediatric Surgery, Massachusetts General Hospital, Harvard Medical School, Boston, MA, United States.

Nicole Picard (N)

Department of Pediatric Surgery, Massachusetts General Hospital, Harvard Medical School, Boston, MA, United States.

Meredith Omer (M)

Department of Pediatric Surgery, Massachusetts General Hospital, Harvard Medical School, Boston, MA, United States.

Ryo Hotta (R)

Department of Pediatric Surgery, Massachusetts General Hospital, Harvard Medical School, Boston, MA, United States.

Allan M Goldstein (AM)

Department of Pediatric Surgery, Massachusetts General Hospital, Harvard Medical School, Boston, MA, United States.

Classifications MeSH