Epilepsy affects approximately 70 million people globally, with 30–40% of patients experiencing drug-resistant seizures despite treatment with multiple antiseizure medications (ASMs). To address this issue, in a study in Disease Models & Mechanisms, the team developed a novel zebrafish model targeting slc25a22a, the ortholog of human SLC25A22—a gene associated with a range of epileptic conditions. Unlike existing zebrafish models, which often rely on gene knockdowns and show weak seizure phenotypes, the new CRISPR-Cas9–generated slc25a22a knockout exhibits robust epilepsy traits, including spontaneous seizures, behavioral hyperactivity and electrophysiological hyperexcitability. Importantly, treatment with valproic acid effectively suppressed these phenotypes, highlighting the model’s relevance for both mechanistic studies and drug screening. These findings indicate that slc25a22a can be a promising therapeutic target for refractory epilepsy and establish this zebrafish line as a valuable platform for preclinical research into novel ASMs.
Original reference: Lee, S.-H. et al. Dis. Model & Mech. 18, dmm052275 (2025)
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