Knockout of tanc2 causes autism-like behavior and sleep disturbance in zebrafish / Fei LONG in Autism Research, 16-3 (March 2023)  

Public ISBD [article]  inAutism Research > 16-3 (March 2023) . - p.524-534 Titre : Knockout of tanc2 causes autism-like behavior and sleep disturbance in zebrafish Type de document : Texte imprimé et/ou numérique Auteurs : Fei LONG, Auteur ; Jing ZHENG, Auteur ; Jiayi ZHOU, Auteur ; Ping HU, Auteur ; Bo XIONG, Auteur Article en page(s) : p.524-534 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Abstract Tanc2 is a large multi-domain postsynaptic scaffold protein mainly expressed in the brain. In humans, tanc2 mutations have been associated with autism spectrum disorder (ASD) and other related neurodevelopmental disorders. However, the role of tanc2 in neurodevelopment and in controlling behaviors are not fully understood. Here, we generated and characterized a tanc2 knockout allele in zebrafish. Loss of tanc2 increases the larval brain size and body length by promoting proliferation and inhibiting apoptosis. We observed that the glutamatergic neuron population is significantly increased in tanc2 mutants while the GABAergic and the glycinergic neurons are not affected, suggesting that an excitatory/inhibitory (E/I) imbalance. Indeed, the tanc2 knockout larvae exhibited increase sleep. In adult zebrafish, the mutants display anxiolytic-behavior, reduced aggression, and impaired social preference. The alterations in these behaviors are phenotypically similar to the ASD patients carrying tanc2 mutations. Therefore, the tanc2 knockout allele could serve as a valuable model to further study the role of tanc2 in the nervous system. En ligne : https://doi.org/10.1002/aur.2880 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=498 [article] Knockout of tanc2 causes autism-like behavior and sleep disturbance in zebrafish [Texte imprimé et/ou numérique] / Fei LONG, Auteur ; Jing ZHENG, Auteur ; Jiayi ZHOU, Auteur ; Ping HU, Auteur ; Bo XIONG, Auteur . - p.524-534. Langues : Anglais (eng) in Autism Research > 16-3 (March 2023) . - p.524-534 Index. décimale : PER Périodiques Résumé : Abstract Tanc2 is a large multi-domain postsynaptic scaffold protein mainly expressed in the brain. In humans, tanc2 mutations have been associated with autism spectrum disorder (ASD) and other related neurodevelopmental disorders. However, the role of tanc2 in neurodevelopment and in controlling behaviors are not fully understood. Here, we generated and characterized a tanc2 knockout allele in zebrafish. Loss of tanc2 increases the larval brain size and body length by promoting proliferation and inhibiting apoptosis. We observed that the glutamatergic neuron population is significantly increased in tanc2 mutants while the GABAergic and the glycinergic neurons are not affected, suggesting that an excitatory/inhibitory (E/I) imbalance. Indeed, the tanc2 knockout larvae exhibited increase sleep. In adult zebrafish, the mutants display anxiolytic-behavior, reduced aggression, and impaired social preference. The alterations in these behaviors are phenotypically similar to the ASD patients carrying tanc2 mutations. Therefore, the tanc2 knockout allele could serve as a valuable model to further study the role of tanc2 in the nervous system. En ligne : https://doi.org/10.1002/aur.2880 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=498

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