PUBLICATION
Neuregulin 1 is involved in enteric nervous system development in zebrafish
- Authors
- Pu, J., Tang, S., Tong, Q., Wang, G., Jia, H., Jia, Q., Li, K., Li, D., Yang, D., Yang, J., Li, H., Li, S., Mei, H.
- ID
- ZDB-PUB-170214-13
- Date
- 2017
- Source
- Journal of Pediatric Surgery 52(7): 1182-1187 (Journal)
- Registered Authors
- Jia, Haibo
- Keywords
- Development, Enteric nervous system, Neuregulin 1, Zebrafish
- MeSH Terms
-
- Animals
- Enteric Nervous System/pathology
- Gastrointestinal Motility/genetics
- Hirschsprung Disease/genetics*
- Humans
- Intestines/pathology
- Neuregulin-1/genetics*
- Phenotype
- Polymorphism, Single Nucleotide
- Zebrafish/genetics
- PubMed
- 28190554 Full text @ J. Pediatr. Surg.
Citation
Pu, J., Tang, S., Tong, Q., Wang, G., Jia, H., Jia, Q., Li, K., Li, D., Yang, D., Yang, J., Li, H., Li, S., Mei, H. (2017) Neuregulin 1 is involved in enteric nervous system development in zebrafish. Journal of Pediatric Surgery. 52(7):1182-1187.
Abstract
Background Hirschsprung's disease (HD, also known as congenital colon aganglionosis) is a congenital disorder characterized by the absence of intramural ganglion cells in the distal gastrointestinal tract, which results in tonic contraction of the aganglionic gut segment and functional intestinal obstruction. Recent studies have indicated neuregulin 1 (NRG1) as a new candidate gene involved in the development of the enteric nervous system (ENS) in humans.
Methods In our study, we investigated the role of NRG1 in zebrafish ENS development by assessing NRG1 expression patterns during ENS development. Knockdown, overexpression and rescue zebrafish models of NRG1 were created to evaluate differences in phenotype, numbers of enteric neurons, ENS-related factors and nerve fiber arrangements.
Results NRG1 was expressed in zebrafish intestine at both the larval and adult stage. We also found that decreased expression of NRG1 resulted in reductions in enteric neuron number and decreased expression of ENS development markers. Moreover, NRG1-knockdown zebrafish exhibited a disordered arrangement of nerve fibers.
Conclusions Collectively, these results demonstrated that NRG1 expression might play a role in zebrafish ENS development. In addition, by modulating NRG1 expression, we created a model that may be useful for investigating the mechanism underlying HD pathogenesis.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping