Details

Autor(en) / Beteiligte

• Gurung, Suman
• Restrepo, Nicole K.
• Anand, Surendra Kumar
• Sittaramane, Vinoth
• Sumanas, Saulius

Titel

Requirement of a novel gene, drish, in the zebrafish retinal ganglion cell and primary motor axon development

Ist Teil von

• Developmental dynamics, 2024-08, Vol.253 (8), p.750-770

Ort / Verlag

Hoboken, USA: John Wiley & Sons, Inc

Erscheinungsjahr

2024

Quelle

Wiley-Blackwell Full Collection

Beschreibungen/Notizen

• 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. Key Findings si:ch73‐364h19.1 / drish gene encodes a putative transmembrane protein, which is expressed in neural, glial and vascular endothelial cells in zebrafish embryos. Drish zebrafish mutants show defects in retinal ganglion cell differentiation. Drish mutants exhibit defective branching of motor axons. Drish mutant larvae and adults display reduced swimming activity, a defective light response and deficient prey capture.