Phenotypic impact of individual conserved neuronal microexons and their master regulators in zebrafish.

ABSTRACT: Microexons exhibit striking evolutionary conservation and are subject to precise, switch-like regulation in neurons, orchestrated by the splicing factors Srrm3 and Srrm4. Disruption of these regulators in mice leads to severe neurological phenotypes, and their misregulation is linked to human disease. However, the specific microexons involved in these phenotypes and the effects of individual microexon deletions on neurodevelopment, physiology, and behavior remain poorly understood. To explore this, we generated zebrafish lines with deletions of 18 individual microexons, alongside srrm3 and srrm4 mutant lines, and conducted comprehensive phenotypic analyses. We discovered that while loss of srrm3, alone or together with srrm4, resulted in significan

SUBMITTER: Lopez-Blanch L 

PROVIDER: S-EPMC12626424 | biostudies-literature | 2025 Nov

REPOSITORIES: biostudies-literature