Phenotypic analysis of catastrophic childhood epilepsy genes.
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ABSTRACT:
SUBMITTER: Griffin A
PROVIDER: S-EPMC8175701 | biostudies-literature | 2021 Jun
REPOSITORIES: biostudies-literature
Publications
Phenotypic analysis of catastrophic childhood epilepsy genes.
Griffin Aliesha A Carpenter Colleen C Liu Jing J Paterno Rosalia R Grone Brian B Hamling Kyla K Moog Maia M Dinday Matthew T MT Figueroa Francisco F Anvar Mana M Ononuju Chinwendu C Qu Tony T Baraban Scott C SC
Communications biology 20210603 1
Genetic engineering techniques have contributed to the now widespread use of zebrafish to investigate gene function, but zebrafish-based human disease studies, and particularly for neurological disorders, are limited. Here we used CRISPR-Cas9 to generate 40 single-gene mutant zebrafish lines representing catastrophic childhood epilepsies. We evaluated larval phenotypes using electrophysiological, behavioral, neuro-anatomical, survival and pharmacological assays. Local field potential recordings ...[more]
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