PUBLICATION
Posterior axis formation requires Dlx5/Dlx6 expression at the neural plate border
- Authors
- Narboux-Neme, N., Ekker, M., Levi, G., Heude, E.
- ID
- ZDB-PUB-190320-12
- Date
- 2019
- Source
- PLoS One 14: e0214063 (Journal)
- Registered Authors
- Ekker, Marc
- Keywords
- none
- MeSH Terms
-
- Animals
- Body Patterning/genetics
- Body Patterning/physiology
- Female
- Gene Expression Regulation, Developmental
- Gene Knockdown Techniques
- Homeodomain Proteins/genetics
- Homeodomain Proteins/metabolism*
- Humans
- Male
- Mice
- Mice, Inbred C57BL
- Mice, Inbred DBA
- Mice, Knockout
- Multigene Family
- Neural Plate/embryology*
- Neural Plate/metabolism*
- Neural Tube Defects/embryology
- Neural Tube Defects/genetics
- Neural Tube Defects/metabolism
- Neurulation/genetics
- Neurulation/physiology
- Pregnancy
- Transcription Factors/deficiency
- Transcription Factors/genetics
- Transcription Factors/metabolism*
- Zebrafish/embryology
- Zebrafish/genetics
- Zebrafish/metabolism
- Zebrafish Proteins/deficiency
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism
- PubMed
- 30889190 Full text @ PLoS One
Citation
Narboux-Neme, N., Ekker, M., Levi, G., Heude, E. (2019) Posterior axis formation requires Dlx5/Dlx6 expression at the neural plate border. PLoS One. 14:e0214063.
Abstract
Neural tube defects (NTDs), one of the most common birth defects in human, present a multifactorial etiology with a poorly defined genetic component. The Dlx5 and Dlx6 bigenic cluster encodes two evolutionary conserved homeodomain transcription factors, which are necessary for proper vertebrate development. It has been shown that Dlx5/6 genes are essential for anterior neural tube closure, however their role in the formation of the posterior structures has never been described. Here, we show that Dlx5/6 expression is required during vertebrate posterior axis formation. Dlx5 presents a similar expression pattern in neural plate border cells during posterior neurulation of zebrafish and mouse. Dlx5/6-inactivation in the mouse results in a phenotype reminiscent of NTDs characterized by open thoracic and lumbar vertebral arches and failure of epaxial muscle formation at the dorsal midline. The dlx5a/6a zebrafish morphants present posterior NTDs associated with abnormal delamination of neural crest cells showing altered expression of cell adhesion molecules and defects of motoneuronal development. Our findings provide new molecular leads to decipher the mechanisms of vertebrate posterior neurulation and might help to gather a better understanding of human congenital NTDs etiology.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping