Centre d'Information et de documentation du CRA Rhône-Alpes
CRA
Informations pratiques
-
Adresse
Centre d'information et de documentation
du CRA Rhône-Alpes
Centre Hospitalier le Vinatier
bât 211
95, Bd Pinel
69678 Bron CedexHoraires
Lundi au Vendredi
9h00-12h00 13h30-16h00Contact
Tél: +33(0)4 37 91 54 65
Mail
Fax: +33(0)4 37 91 54 37
-
Résultat de la recherche
5 recherche sur le mot-clé 'zebrafish'
Affiner la recherche Générer le flux rss de la recherche
Partager le résultat de cette recherche Faire une suggestion
Zebrafish knockout of Down syndrome gene, DYRK1A, shows social impairments relevant to autism / O. H. KIM in Molecular Autism, 8 (2017)
[article]
Titre : Zebrafish knockout of Down syndrome gene, DYRK1A, shows social impairments relevant to autism Type de document : Texte imprimé et/ou numérique Auteurs : O. H. KIM, Auteur ; H. J. CHO, Auteur ; E. HAN, Auteur ; T. I. HONG, Auteur ; K. ARIYASIRI, Auteur ; J. H. CHOI, Auteur ; K. S. HWANG, Auteur ; Y. M. JEONG, Auteur ; S. Y. YANG, Auteur ; K. YU, Auteur ; D. S. PARK, Auteur ; H. W. OH, Auteur ; E. E. DAVIS, Auteur ; C. E. SCHWARTZ, Auteur ; J. S. LEE, Auteur ; H. G. KIM, Auteur ; C. H. KIM, Auteur Article en page(s) : 50p. Langues : Anglais (eng) Mots-clés : Autism Dyrk1a Down syndrome Group behavior Knockout Shoaling Social interaction Zebrafish Index. décimale : PER Périodiques Résumé : BACKGROUND: DYRK1A maps to the Down syndrome critical region at 21q22. Mutations in this kinase-encoding gene have been reported to cause microcephaly associated with either intellectual disability or autism in humans. Intellectual disability accompanied by microcephaly was recapitulated in a murine model by overexpressing Dyrk1a which mimicked Down syndrome phenotypes. However, given embryonic lethality in homozygous knockout (KO) mice, no murine model studies could present sufficient evidence to link Dyrk1a dysfunction with autism. To understand the molecular mechanisms underlying microcephaly and autism spectrum disorders (ASD), we established an in vivo dyrk1aa KO model using zebrafish. METHODS: We identified a patient with a mutation in the DYRK1A gene using microarray analysis. Circumventing the barrier of murine model studies, we generated a dyrk1aa KO zebrafish using transcription activator-like effector nuclease (TALEN)-mediated genome editing. For social behavioral tests, we have established a social interaction test, shoaling assay, and group behavior assay. For molecular analysis, we examined the neuronal activity in specific brain regions of dyrk1aa KO zebrafish through in situ hybridization with various probes including c-fos and crh which are the molecular markers for stress response. RESULTS: Microarray detected an intragenic microdeletion of DYRK1A in an individual with microcephaly and autism. From behavioral tests of social interaction and group behavior, dyrk1aa KO zebrafish exhibited social impairments that reproduce human phenotypes of autism in a vertebrate animal model. Social impairment in dyrk1aa KO zebrafish was further confirmed by molecular analysis of c-fos and crh expression. Transcriptional expression of c-fos and crh was lower than that of wild type fish in specific hypothalamic regions, suggesting that KO fish brains are less activated by social context. CONCLUSIONS: In this study, we established a zebrafish model to validate a candidate gene for autism in a vertebrate animal. These results illustrate the functional deficiency of DYRK1A as an underlying disease mechanism for autism. We also propose simple social behavioral assays as a tool for the broader study of autism candidate genes. En ligne : http://dx.doi.org/10.1186/s13229-017-0168-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=330
in Molecular Autism > 8 (2017) . - 50p.[article] Zebrafish knockout of Down syndrome gene, DYRK1A, shows social impairments relevant to autism [Texte imprimé et/ou numérique] / O. H. KIM, Auteur ; H. J. CHO, Auteur ; E. HAN, Auteur ; T. I. HONG, Auteur ; K. ARIYASIRI, Auteur ; J. H. CHOI, Auteur ; K. S. HWANG, Auteur ; Y. M. JEONG, Auteur ; S. Y. YANG, Auteur ; K. YU, Auteur ; D. S. PARK, Auteur ; H. W. OH, Auteur ; E. E. DAVIS, Auteur ; C. E. SCHWARTZ, Auteur ; J. S. LEE, Auteur ; H. G. KIM, Auteur ; C. H. KIM, Auteur . - 50p.
Langues : Anglais (eng)
in Molecular Autism > 8 (2017) . - 50p.
Mots-clés : Autism Dyrk1a Down syndrome Group behavior Knockout Shoaling Social interaction Zebrafish Index. décimale : PER Périodiques Résumé : BACKGROUND: DYRK1A maps to the Down syndrome critical region at 21q22. Mutations in this kinase-encoding gene have been reported to cause microcephaly associated with either intellectual disability or autism in humans. Intellectual disability accompanied by microcephaly was recapitulated in a murine model by overexpressing Dyrk1a which mimicked Down syndrome phenotypes. However, given embryonic lethality in homozygous knockout (KO) mice, no murine model studies could present sufficient evidence to link Dyrk1a dysfunction with autism. To understand the molecular mechanisms underlying microcephaly and autism spectrum disorders (ASD), we established an in vivo dyrk1aa KO model using zebrafish. METHODS: We identified a patient with a mutation in the DYRK1A gene using microarray analysis. Circumventing the barrier of murine model studies, we generated a dyrk1aa KO zebrafish using transcription activator-like effector nuclease (TALEN)-mediated genome editing. For social behavioral tests, we have established a social interaction test, shoaling assay, and group behavior assay. For molecular analysis, we examined the neuronal activity in specific brain regions of dyrk1aa KO zebrafish through in situ hybridization with various probes including c-fos and crh which are the molecular markers for stress response. RESULTS: Microarray detected an intragenic microdeletion of DYRK1A in an individual with microcephaly and autism. From behavioral tests of social interaction and group behavior, dyrk1aa KO zebrafish exhibited social impairments that reproduce human phenotypes of autism in a vertebrate animal model. Social impairment in dyrk1aa KO zebrafish was further confirmed by molecular analysis of c-fos and crh expression. Transcriptional expression of c-fos and crh was lower than that of wild type fish in specific hypothalamic regions, suggesting that KO fish brains are less activated by social context. CONCLUSIONS: In this study, we established a zebrafish model to validate a candidate gene for autism in a vertebrate animal. These results illustrate the functional deficiency of DYRK1A as an underlying disease mechanism for autism. We also propose simple social behavioral assays as a tool for the broader study of autism candidate genes. En ligne : http://dx.doi.org/10.1186/s13229-017-0168-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=330 Zebrafish Social Behavior Testing in Developmental Brain Disorders / Christine BUSKE
Titre : Zebrafish Social Behavior Testing in Developmental Brain Disorders Type de document : Texte imprimé et/ou numérique Auteurs : Christine BUSKE, Auteur Année de publication : 2015 Importance : p.303-316 Langues : Anglais (eng) Mots-clés : Behavioral paradigms Zebrafish High-throughput screening Pervasive brain disorders Autism Index. décimale : AUT-B AUT-B - L'Autisme - Ouvrages généraux et scientifiques Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=265 Zebrafish Social Behavior Testing in Developmental Brain Disorders [Texte imprimé et/ou numérique] / Christine BUSKE, Auteur . - 2015 . - p.303-316.
Langues : Anglais (eng)
Mots-clés : Behavioral paradigms Zebrafish High-throughput screening Pervasive brain disorders Autism Index. décimale : AUT-B AUT-B - L'Autisme - Ouvrages généraux et scientifiques Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=265 Exemplaires
Code-barres Cote Support Localisation Section Disponibilité aucun exemplaire Duloxetine ameliorates valproic acid-induced hyperactivity, anxiety-like behavior, and social interaction deficits in zebrafish / T. P. JOSEPH in Autism Research, 15-1 (January 2022)
[article]
Titre : Duloxetine ameliorates valproic acid-induced hyperactivity, anxiety-like behavior, and social interaction deficits in zebrafish Type de document : Texte imprimé et/ou numérique Auteurs : T. P. JOSEPH, Auteur ; F. ZHOU, Auteur ; L. Y. SAI, Auteur ; H. CHEN, Auteur ; S. L. LIN, Auteur ; M. SCHACHNER, Auteur Article en page(s) : p.27-41 Langues : Anglais (eng) Mots-clés : Animals Anxiety/chemically induced/drug therapy Autism Spectrum Disorder/drug therapy Behavior, Animal Disease Models, Animal Duloxetine Hydrochloride Prenatal Exposure Delayed Effects Social Behavior Social Interaction Valproic Acid Zebrafish L1cam autism spectrum disorder duloxetine social preference valproic acid zebrafish Index. décimale : PER Périodiques Résumé : Syndromic autism spectrum disorders (ASDs) are characterized by impaired social communication and repetitive/stereotyped behaviors. Currently available therapeutic agents against ASD have limited efficacy. Thus, searching for novel and effective drugs ameliorating core symptoms, in particular social deficits, is of utmost importance. Duloxetine (DLX), an antidepressant that has been identified as an agonist mimetic for the cell adhesion molecule L1, exhibits beneficial functions in vitro and in vivo. Therefore, in this study, we focused on the rapid and persistent neuroprotective function of DLX following valproic acid (VPA)-triggered hyperactivity, anxiety-like behavior and social deficits in zebrafish. Embryonic exposure to VPA reduced survival in a dose- and time-dependent manner, delayed hatching, and also resulted in a significant number of malformed larvae. After initial dose-response experiments in zebrafish larvae, 10 ?M VPA exposure between 0.33 and 4.5?days post fertilization (dpf) was identified as an effective concentration that led to an early and persistent ASD-like phenotype in zebrafish. ASD-like elevated acetylcholine esterase (AChE) activity and reduced Akt-mTOR signaling was observed in zebrafish whole brain. Acute administration of DLX (4.5-6 dpf) reduced the VPA-induced ASD-like phenotype in zebrafish larvae. Additionally, such early-life acute DLX treatment had long-term effects in ameliorating social impairments, hyperactivity, and anxiety-like behaviors through adulthood. This was accompanied by reduced AChE activity and by normalized Akt-mTOR signaling. Overall, DLX treatment showed a long-term therapeutic effect on autistic-like behaviors, and alteration of AChE activity and Akt-mTOR signaling were identified as crucial in the VPA-induced ASD zebrafish model. En ligne : http://dx.doi.org/10.1002/aur.2620 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=450
in Autism Research > 15-1 (January 2022) . - p.27-41[article] Duloxetine ameliorates valproic acid-induced hyperactivity, anxiety-like behavior, and social interaction deficits in zebrafish [Texte imprimé et/ou numérique] / T. P. JOSEPH, Auteur ; F. ZHOU, Auteur ; L. Y. SAI, Auteur ; H. CHEN, Auteur ; S. L. LIN, Auteur ; M. SCHACHNER, Auteur . - p.27-41.
Langues : Anglais (eng)
in Autism Research > 15-1 (January 2022) . - p.27-41
Mots-clés : Animals Anxiety/chemically induced/drug therapy Autism Spectrum Disorder/drug therapy Behavior, Animal Disease Models, Animal Duloxetine Hydrochloride Prenatal Exposure Delayed Effects Social Behavior Social Interaction Valproic Acid Zebrafish L1cam autism spectrum disorder duloxetine social preference valproic acid zebrafish Index. décimale : PER Périodiques Résumé : Syndromic autism spectrum disorders (ASDs) are characterized by impaired social communication and repetitive/stereotyped behaviors. Currently available therapeutic agents against ASD have limited efficacy. Thus, searching for novel and effective drugs ameliorating core symptoms, in particular social deficits, is of utmost importance. Duloxetine (DLX), an antidepressant that has been identified as an agonist mimetic for the cell adhesion molecule L1, exhibits beneficial functions in vitro and in vivo. Therefore, in this study, we focused on the rapid and persistent neuroprotective function of DLX following valproic acid (VPA)-triggered hyperactivity, anxiety-like behavior and social deficits in zebrafish. Embryonic exposure to VPA reduced survival in a dose- and time-dependent manner, delayed hatching, and also resulted in a significant number of malformed larvae. After initial dose-response experiments in zebrafish larvae, 10 ?M VPA exposure between 0.33 and 4.5?days post fertilization (dpf) was identified as an effective concentration that led to an early and persistent ASD-like phenotype in zebrafish. ASD-like elevated acetylcholine esterase (AChE) activity and reduced Akt-mTOR signaling was observed in zebrafish whole brain. Acute administration of DLX (4.5-6 dpf) reduced the VPA-induced ASD-like phenotype in zebrafish larvae. Additionally, such early-life acute DLX treatment had long-term effects in ameliorating social impairments, hyperactivity, and anxiety-like behaviors through adulthood. This was accompanied by reduced AChE activity and by normalized Akt-mTOR signaling. Overall, DLX treatment showed a long-term therapeutic effect on autistic-like behaviors, and alteration of AChE activity and Akt-mTOR signaling were identified as crucial in the VPA-induced ASD zebrafish model. En ligne : http://dx.doi.org/10.1002/aur.2620 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=450 Use of the Zebrafish Model to Understand Behavioral Disorders Associated with Altered Oxytocin System Development: Implications for Autism and Prader–Willi Syndrome / Nicole JOHNSTON
Titre : Use of the Zebrafish Model to Understand Behavioral Disorders Associated with Altered Oxytocin System Development: Implications for Autism and Prader–Willi Syndrome Type de document : Texte imprimé et/ou numérique Auteurs : Nicole JOHNSTON, Auteur ; Eric GLASGOW, Auteur Année de publication : 2015 Importance : p.451-470 Langues : Anglais (eng) Mots-clés : Zebrafish Autism spectrum disorder Prader–Willi syndrome Oxytocin Shoaling assay Anxiety assay Zebrafish behavior Animal model Index. décimale : AUT-B AUT-B - L'Autisme - Ouvrages généraux et scientifiques Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=265 Use of the Zebrafish Model to Understand Behavioral Disorders Associated with Altered Oxytocin System Development: Implications for Autism and Prader–Willi Syndrome [Texte imprimé et/ou numérique] / Nicole JOHNSTON, Auteur ; Eric GLASGOW, Auteur . - 2015 . - p.451-470.
Langues : Anglais (eng)
Mots-clés : Zebrafish Autism spectrum disorder Prader–Willi syndrome Oxytocin Shoaling assay Anxiety assay Zebrafish behavior Animal model Index. décimale : AUT-B AUT-B - L'Autisme - Ouvrages généraux et scientifiques Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=265 Exemplaires
Code-barres Cote Support Localisation Section Disponibilité aucun exemplaire CRISPR/Cas9-induced shank3b mutant zebrafish display autism-like behaviors / C. X. LIU in Molecular Autism, 9 (2018)
[article]
Titre : CRISPR/Cas9-induced shank3b mutant zebrafish display autism-like behaviors Type de document : Texte imprimé et/ou numérique Auteurs : C. X. LIU, Auteur ; C. Y. LI, Auteur ; C. C. HU, Auteur ; Y. WANG, Auteur ; J. LIN, Auteur ; Y. H. JIANG, Auteur ; Q. LI, Auteur ; X. XU, Auteur Article en page(s) : 23p. Langues : Anglais (eng) Mots-clés : Asd Animal model CRISPR/Cas9 Social behavior Zebrafish shank3 Index. décimale : PER Périodiques Résumé : Background: Human genetic and genomic studies have supported a strong causal role of SHANK3 deficiency in autism spectrum disorder (ASD). However, the molecular mechanism underlying SHANK3 deficiency resulting in ASD is not fully understood. Recently, the zebrafish has become an attractive organism to model ASD because of its high efficiency of genetic manipulation and robust behavioral phenotypes. The orthologous gene to human SHANK3 is duplicated in the zebrafish genome and has two homologs, shank3a and shank3b. Previous studies have reported shank3 morphants in zebrafish using the morpholino method. Here, we report the generation and characterization of shank3b mutant zebrafish in larval and adult stages using the CRISPR/Cas9 genome editing technique. Methods: CRISPR/Cas9 was applied to generate a shank3b loss-of-function mutation (shank3b(-/-) ) in zebrafish. A series of morphological measurements, behavioral tests, and molecular analyses were performed to systematically characterize the behavioral and molecular changes in shank3b mutant zebrafish. Results: shank3b(-/-) zebrafish exhibited abnormal morphology in early development. They showed reduced locomotor activity both as larvae and adults, reduced social interaction and time spent near conspecifics, and significant repetitive swimming behaviors. Additionally, the levels of both postsynaptic homer1 and presynaptic synaptophysin were significantly reduced in the adult brain of shank3b-deficient zebrafish. Conclusions: We generated the first inheritable shank3b mutant zebrafish model using CRISPR/Cas9 gene editing approach. shank3b(-/-) zebrafish displayed robust autism-like behaviors and altered levels of the synaptic proteins homer1 and synaptophysin. The versatility of zebrafish as a model for studying neurodevelopment and conducting drug screening will likely have a significant contribution to future studies of human SHANK3 function and ASD. En ligne : http://dx.doi.org/10.1186/s13229-018-0204-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=354
in Molecular Autism > 9 (2018) . - 23p.[article] CRISPR/Cas9-induced shank3b mutant zebrafish display autism-like behaviors [Texte imprimé et/ou numérique] / C. X. LIU, Auteur ; C. Y. LI, Auteur ; C. C. HU, Auteur ; Y. WANG, Auteur ; J. LIN, Auteur ; Y. H. JIANG, Auteur ; Q. LI, Auteur ; X. XU, Auteur . - 23p.
Langues : Anglais (eng)
in Molecular Autism > 9 (2018) . - 23p.
Mots-clés : Asd Animal model CRISPR/Cas9 Social behavior Zebrafish shank3 Index. décimale : PER Périodiques Résumé : Background: Human genetic and genomic studies have supported a strong causal role of SHANK3 deficiency in autism spectrum disorder (ASD). However, the molecular mechanism underlying SHANK3 deficiency resulting in ASD is not fully understood. Recently, the zebrafish has become an attractive organism to model ASD because of its high efficiency of genetic manipulation and robust behavioral phenotypes. The orthologous gene to human SHANK3 is duplicated in the zebrafish genome and has two homologs, shank3a and shank3b. Previous studies have reported shank3 morphants in zebrafish using the morpholino method. Here, we report the generation and characterization of shank3b mutant zebrafish in larval and adult stages using the CRISPR/Cas9 genome editing technique. Methods: CRISPR/Cas9 was applied to generate a shank3b loss-of-function mutation (shank3b(-/-) ) in zebrafish. A series of morphological measurements, behavioral tests, and molecular analyses were performed to systematically characterize the behavioral and molecular changes in shank3b mutant zebrafish. Results: shank3b(-/-) zebrafish exhibited abnormal morphology in early development. They showed reduced locomotor activity both as larvae and adults, reduced social interaction and time spent near conspecifics, and significant repetitive swimming behaviors. Additionally, the levels of both postsynaptic homer1 and presynaptic synaptophysin were significantly reduced in the adult brain of shank3b-deficient zebrafish. Conclusions: We generated the first inheritable shank3b mutant zebrafish model using CRISPR/Cas9 gene editing approach. shank3b(-/-) zebrafish displayed robust autism-like behaviors and altered levels of the synaptic proteins homer1 and synaptophysin. The versatility of zebrafish as a model for studying neurodevelopment and conducting drug screening will likely have a significant contribution to future studies of human SHANK3 function and ASD. En ligne : http://dx.doi.org/10.1186/s13229-018-0204-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=354