Modeling epilepsy by loss-of-function of the CUG-binding protein Elav-like family member 2 in zebrafish with multi-omics analysis.

ABSTRACT:

Background

The CUG-binding protein Elav-like family member 2 ( CELF2 ) gene has been linked to the pathogenesis of epilepsy, but its precise role remains unclear. This study aimed to investigate the pathogenic mechanisms of CELF2  mutation in epilepsy, utilizing zebrafish models to explore its molecular pathways and biological impact.

Methods

Whole-exome sequencing was performed to identify CELF2  mutations associated with epilepsy. CELF2 zebrafish model was generated using clustered regularly interspaced short palindromic repeats (CRISPR)/CRISPR-related protein 9 technology and morpholinos, followed by behavioral and electroencephalographic analyses to confirm epileptic phenotypes. Proteomic and metabolomic analyses were conducted to examine the impact of CELF2  deficiency

SUBMITTER: Wang X 

PROVIDER: S-EPMC12875712 | biostudies-literature | 2026 Feb

REPOSITORIES: biostudies-literature