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Epilepsy has been found to have numerous etiologies and whilst about two thirds of epilepsies were classically considered idiopathic, a majority of those is now believed to be of genetic origin. Mutations in genes involved in GABA-mediated inhibitory neurotransmission have been associated with a broad range of epilepsy syndromes. Mutations in the GABA-A receptor gamma 2 subunit gene (GABRG2), for example, have been associated with absence epilepsy and febrile seizures in humans. Several rodent models of GABRG2 loss-of-function depict clinical features of the disease, however, alternative genetic models more amenable for the study of ictogenesis and for high-throughput screening purposes are still needed. In this context, we generated a gabrg2 knock-out zebrafish model (R23X) that displayed light\/dark-induced reflex seizures. Through high-resolution in vivo calcium imaging of the brain, we showed that this phenotype is associated with widespread increases in neuronal activity that can be effectively alleviated by the anti-epileptic drug valproic acid. Moreover these seizures only occur at the larval stages but disappear after one week of age. Interestingly, our whole transcriptome analysis showed that gabrg2-KO does not alter the expression of genes in the larval brain. As a result, gabrg2-\/- zebrafish is a novel in vivo genetic model of early epilepsies that opens new doors to investigate ictogenesis and for further drug-screening assays.<\/jats:p>","DOI":"10.1242\/dmm.040782","type":"journal-article","created":{"date-parts":[[2019,10,3]],"date-time":"2019-10-03T12:55:13Z","timestamp":1570107313000},"source":"Crossref","is-referenced-by-count":37,"title":["Targeted knockout of GABA receptor gamma 2 subunit provokes transient light-induced reflex seizures in zebrafish larvae"],"prefix":"10.1242","author":[{"given":"Meijiang","family":"Liao","sequence":"first","affiliation":[{"name":"Research Center of the University of Montreal Hospital Center (CRCHUM), Department of Neurosciences, Universit\u00e9 de Montr\u00e9al, Montr\u00e9al, H2X 0A9 QC, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4791-4646","authenticated-orcid":false,"given":"Uday","family":"Kundap","sequence":"additional","affiliation":[{"name":"Research Center of the University of Montreal Hospital Center (CRCHUM), Department of Neurosciences, Universit\u00e9 de Montr\u00e9al, Montr\u00e9al, H2X 0A9 QC, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0316-5818","authenticated-orcid":false,"given":"Richard E.","family":"Rosch","sequence":"additional","affiliation":[{"name":"MRC Centre for Neurodevelopmental Disorders, Institute of Psychiatry, Psychology and Neuroscience, King's College London, London, UK"},{"name":"Department of Bioengineering, University of Pennsylvania, PA, USA"},{"name":"Department of Paediatric Neurology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dominic R. 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