Effects of a postnatal Atrx conditional knockout in neurons on autism-like behaviours in male and female mice / Nicole MARTIN-KENNY in Journal of Neurodevelopmental Disorders, 12 (2020)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 12 (2020) Titre : Effects of a postnatal Atrx conditional knockout in neurons on autism-like behaviours in male and female mice Type de document : texte imprimé Auteurs : Nicole MARTIN-KENNY, Auteur ; Nathalie G. BÉRUBÉ, Auteur Langues : Anglais (eng) Mots-clés : Animals  Autistic Disorder/genetics  Chromatin Assembly and Disassembly  Female  Male  X-Linked Intellectual Disability/genetics  Mice  Mice, Knockout  Mutation  Neurons/metabolism  Postpartum Period  X-linked Nuclear Protein  alpha-Thalassemia/genetics  Atrx  Autism spectrum disorder  Cre/loxP system  Genetically engineered mice  Repetitive behaviours  Social behaviours  Startle response Index. décimale : PER Périodiques Résumé : BACKGROUND: Alpha-thalassemia/mental retardation, X-linked, or ATRX, is an autism susceptibility gene that encodes a chromatin remodeler. Mutations of ATRX result in the ATR-X intellectual disability syndrome and have been identified in autism spectrum disorder (ASD) patients. The mechanisms by which ATRX mutations lead to autism and autistic-like behaviours are not yet known. To address this question, we generated mice with postnatal Atrx inactivation in excitatory neurons of the forebrain and performed a battery of behavioural assays that assess autistic-like behaviours. METHODS: Male and female mice with a postnatal conditional ablation of ATRX were generated using the Cre/lox system under the control of the αCaMKII gene promoter. These mice were tested in a battery of behavioural tests that assess autistic-like features. We utilized paradigms that measure social behaviour, repetitive, and stereotyped behaviours, as well as sensory gating. Statistics were calculated by two-way repeated measures ANOVA with Sidak's multiple comparison test or unpaired Student's t tests as indicated. RESULTS: The behaviour tests revealed no significant differences between Atrx-cKO and control mice. We identified sexually dimorphic changes in odor habituation and discrimination; however, these changes did not correlate with social deficits. CONCLUSION: The postnatal knockout of Atrx in forebrain excitatory neurons does not lead to autism-related behaviours in male or female mice. En ligne : https://dx.doi.org/10.1186/s11689-020-09319-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 [article] Effects of a postnatal Atrx conditional knockout in neurons on autism-like behaviours in male and female mice [texte imprimé] / Nicole MARTIN-KENNY, Auteur ; Nathalie G. BÉRUBÉ, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 12 (2020) Mots-clés : Animals  Autistic Disorder/genetics  Chromatin Assembly and Disassembly  Female  Male  X-Linked Intellectual Disability/genetics  Mice  Mice, Knockout  Mutation  Neurons/metabolism  Postpartum Period  X-linked Nuclear Protein  alpha-Thalassemia/genetics  Atrx  Autism spectrum disorder  Cre/loxP system  Genetically engineered mice  Repetitive behaviours  Social behaviours  Startle response Index. décimale : PER Périodiques Résumé : BACKGROUND: Alpha-thalassemia/mental retardation, X-linked, or ATRX, is an autism susceptibility gene that encodes a chromatin remodeler. Mutations of ATRX result in the ATR-X intellectual disability syndrome and have been identified in autism spectrum disorder (ASD) patients. The mechanisms by which ATRX mutations lead to autism and autistic-like behaviours are not yet known. To address this question, we generated mice with postnatal Atrx inactivation in excitatory neurons of the forebrain and performed a battery of behavioural assays that assess autistic-like behaviours. METHODS: Male and female mice with a postnatal conditional ablation of ATRX were generated using the Cre/lox system under the control of the αCaMKII gene promoter. These mice were tested in a battery of behavioural tests that assess autistic-like features. We utilized paradigms that measure social behaviour, repetitive, and stereotyped behaviours, as well as sensory gating. Statistics were calculated by two-way repeated measures ANOVA with Sidak's multiple comparison test or unpaired Student's t tests as indicated. RESULTS: The behaviour tests revealed no significant differences between Atrx-cKO and control mice. We identified sexually dimorphic changes in odor habituation and discrimination; however, these changes did not correlate with social deficits. CONCLUSION: The postnatal knockout of Atrx in forebrain excitatory neurons does not lead to autism-related behaviours in male or female mice. En ligne : https://dx.doi.org/10.1186/s11689-020-09319-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573

A mouse model of ATRX deficiency with cognitive deficits and autistic traits / Katherine M. QUESNEL in Journal of Neurodevelopmental Disorders, 15 (2023)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 15 (2023) Titre : A mouse model of ATRX deficiency with cognitive deficits and autistic traits Type de document : texte imprimé Auteurs : Katherine M. QUESNEL, Auteur ; Nicole MARTIN-KENNY, Auteur ; Nathalie G. BÉRUBÉ, Auteur Langues : Anglais (eng) Mots-clés : Male  Cognition  X-Linked Intellectual Disability  Neurons/physiology  Memory Disorders/etiology  Female  alpha-Thalassemia  Autistic Disorder/complications/genetics  Mice  Animals  Atrx  Autism  Intellectual disability  Sex differences Index. décimale : PER Périodiques Résumé : BACKGROUND: ATRX is an ATP-dependent chromatin remodeling protein with essential roles in safeguarding genome integrity and modulating gene expression. Deficiencies in this protein cause ATR-X syndrome, a condition characterized by intellectual disability and an array of developmental abnormalities, including features of autism. Previous studies demonstrated that deleting ATRX in mouse forebrain excitatory neurons postnatally resulted in male-specific memory deficits, but no apparent autistic-like behaviours. METHODS: We generated mice with an earlier embryonic deletion of ATRX in forebrain excitatory neurons and characterized their behaviour using a series of memory and autistic-related paradigms. RESULTS: We found that mutant mice displayed a broader spectrum of impairments, including fear memory, decreased anxiety-like behaviour, hyperactivity, as well as self-injurious and repetitive grooming. Sex-specific alterations were also observed, including male-specific aggression, sensory gating impairments, and decreased social memory. CONCLUSIONS: Collectively, the findings indicate that early developmental abnormalities arising from ATRX deficiency in forebrain excitatory neurons contribute to the presentation of fear memory deficits as well as autistic-like behaviours. En ligne : https://dx.doi.org/10.1186/s11689-023-09508-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=575 [article] A mouse model of ATRX deficiency with cognitive deficits and autistic traits [texte imprimé] / Katherine M. QUESNEL, Auteur ; Nicole MARTIN-KENNY, Auteur ; Nathalie G. BÉRUBÉ, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 15 (2023) Mots-clés : Male  Cognition  X-Linked Intellectual Disability  Neurons/physiology  Memory Disorders/etiology  Female  alpha-Thalassemia  Autistic Disorder/complications/genetics  Mice  Animals  Atrx  Autism  Intellectual disability  Sex differences Index. décimale : PER Périodiques Résumé : BACKGROUND: ATRX is an ATP-dependent chromatin remodeling protein with essential roles in safeguarding genome integrity and modulating gene expression. Deficiencies in this protein cause ATR-X syndrome, a condition characterized by intellectual disability and an array of developmental abnormalities, including features of autism. Previous studies demonstrated that deleting ATRX in mouse forebrain excitatory neurons postnatally resulted in male-specific memory deficits, but no apparent autistic-like behaviours. METHODS: We generated mice with an earlier embryonic deletion of ATRX in forebrain excitatory neurons and characterized their behaviour using a series of memory and autistic-related paradigms. RESULTS: We found that mutant mice displayed a broader spectrum of impairments, including fear memory, decreased anxiety-like behaviour, hyperactivity, as well as self-injurious and repetitive grooming. Sex-specific alterations were also observed, including male-specific aggression, sensory gating impairments, and decreased social memory. CONCLUSIONS: Collectively, the findings indicate that early developmental abnormalities arising from ATRX deficiency in forebrain excitatory neurons contribute to the presentation of fear memory deficits as well as autistic-like behaviours. En ligne : https://dx.doi.org/10.1186/s11689-023-09508-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=575

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