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Here, we focussed on correcting metabolic defects in a catastrophic paediatric epilepsy, Dravet syndrome which is caused by mutations in sodium channel NaV1.1 gene, SCN1A. We utilized a translatable zebrafish model of Dravet syndrome (scn1lab) which exhibits key characteristics of patients with Dravet syndrome and shows metabolic deficits accompanied by down-regulation of gluconeogenesis genes, pck1 and pck2. Using a metabolism-based small library screen, we identified compounds that increased gluconeogenesis via up-regulation of pck1 gene expression in scn1lab larvae. Treatment with PK11195, a pck1 activator and a translocator protein ligand, normalized dys-regulated glucose levels, metabolic deficits, translocator protein expression and significantly decreased electrographic seizures in mutant larvae. Inhibition of pck1 in wild-type larvae mimicked metabolic and behaviour defects observed in scn1lab mutants. Together, this suggests that correcting dys-regulated metabolic pathways can be therapeutic in neurodevelopmental disorders such as Dravet syndrome arising from ion channel dysfunction.<\/jats:p>","DOI":"10.1093\/braincomms\/fcab004","type":"journal-article","created":{"date-parts":[[2021,1,26]],"date-time":"2021-01-26T02:02:44Z","timestamp":1611626564000},"source":"Crossref","is-referenced-by-count":31,"title":["Enhancing glucose metabolism via gluconeogenesis is therapeutic in a zebrafish model of Dravet syndrome"],"prefix":"10.1093","volume":"3","author":[{"given":"Rajeswari","family":"Banerji","sequence":"first","affiliation":[{"name":"Skaggs School of Pharmacy and Pharmaceutical Sciences, University of Colorado, Anschutz Medical Campus, Aurora, Colorado, CA 80045, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christopher","family":"Huynh","sequence":"additional","affiliation":[{"name":"Skaggs School of Pharmacy and Pharmaceutical Sciences, University of Colorado, Anschutz Medical Campus, Aurora, Colorado, CA 80045, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Francisco","family":"Figueroa","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Epilepsy Research Laboratory, University of California, San Francisco, CA 94143, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Matthew T","family":"Dinday","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Epilepsy Research Laboratory, University of California, San Francisco, CA 94143, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Scott C","family":"Baraban","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Epilepsy Research Laboratory, University of California, San Francisco, CA 94143, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Manisha","family":"Patel","sequence":"additional","affiliation":[{"name":"Skaggs School of Pharmacy and Pharmaceutical Sciences, University of Colorado, Anschutz Medical Campus, Aurora, Colorado, CA 80045, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2021,1,25]]},"reference":[{"key":"2021091118485188900_fcab004-B1","doi-asserted-by":"crossref","first-page":"576","DOI":"10.1016\/S0021-9258(17)36131-8","article-title":"The peripheral-type benzodiazepine receptor. 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