PUBLICATION
Requirement of a novel gene, drish, in the zebrafish retinal ganglion cell and primary motor axon development
- Authors
- Gurung, S., Restrepo, N.K., Anand, S.K., Sittaramane, V., Sumanas, S.
- ID
- ZDB-PUB-240211-10
- Date
- 2024
- Source
- Developmental Dynamics : an official publication of the American Association of Anatomists 253(8): 750-770 (Journal)
- Registered Authors
- Restrepo, Nicole, Sumanas, Saulius
- Keywords
- axon, branching, differentiation, motor, retinal, visual, zebrafish
- MeSH Terms
-
- Animals
- Axons*/metabolism
- Axons*/physiology
- Embryo, Nonmammalian/metabolism
- Gene Expression Regulation, Developmental
- Motor Neurons/metabolism
- Motor Neurons/physiology
- Neurogenesis*/genetics
- Neurogenesis*/physiology
- Retina/embryology
- Retina/metabolism
- Retinal Ganglion Cells*/metabolism
- Retinal Ganglion Cells*/physiology
- Zebrafish*/genetics
- Zebrafish Proteins*/genetics
- Zebrafish Proteins*/metabolism
- PubMed
- 38340011 Full text @ Dev. Dyn.
Citation
Gurung, S., Restrepo, N.K., Anand, S.K., Sittaramane, V., Sumanas, S. (2024) Requirement of a novel gene, drish, in the zebrafish retinal ganglion cell and primary motor axon development. Developmental Dynamics : an official publication of the American Association of Anatomists. 253(8):750-770.
Abstract
Background During neurogenesis, growing axons must navigate through the complex extracellular environment and make correct synaptic connections for the proper functioning of neural circuits. The mechanisms underlying the formation of functional neural networks are still only partially understood.
Results Here we analyzed the role of a novel gene si:ch73-364h19.1/drish in the neural and vascular development of zebrafish embryos. We show that drish mRNA is expressed broadly and dynamically in multiple cell types including neural, glial, retinal progenitor and vascular endothelial cells throughout the early stages of embryonic development. To study Drish function during embryogenesis, we generated drish genetic mutant using CRISPR/Cas9 genome editing. drish loss-of-function mutant larvae displayed defects in early retinal ganglion cell, optic nerve and the retinal inner nuclear layer formation, as well as ectopic motor axon branching. In addition, drish mutant adults exhibited deficient retinal outer nuclear layer and showed defective light response and locomotory behavior. However, vascular patterning and blood circulation were not significantly affected.
Conclusions Together, these data demonstrate important roles of zebrafish drish in the retinal ganglion cell, optic nerve and interneuron development and in spinal motor axon branching.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping