[article]
| Titre : |
Developmental delays in cortical auditory temporal processing in a mouse model of Fragile X syndrome |
| Type de document : |
texte imprimé |
| Auteurs : |
Katilynne CROOM, Auteur ; Jeffrey A. RUMSCHLAG, Auteur ; Michael A. ERICKSON, Auteur ; Devin K. BINDER, Auteur ; Khaleel A. RAZAK, Auteur |
| Langues : |
Anglais (eng) |
| Mots-clés : |
Humans Adult Animals Mice Fragile X Syndrome/complications Cross-Sectional Studies Time Perception Disease Models, Animal Mice, Knockout Fragile X Mental Retardation Protein/genetics Autism spectrum disorders Cerebral cortex Fragile X syndrome Language Neurodevelopment Sensory hypersensitivity Speech processing Temporal processing |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: Autism spectrum disorders (ASD) encompass a wide array of debilitating symptoms, including sensory dysfunction and delayed language development. Auditory temporal processing is crucial for speech perception and language development. Abnormal development of temporal processing may account for the language impairments associated with ASD. Very little is known about the development of temporal processing in any animal model of ASD. METHODS: In the current study, we quantify auditory temporal processing throughout development in the Fmr1 knock-out (KO) mouse model of Fragile X Syndrome (FXS), a leading genetic cause of intellectual disability and ASD-associated behaviors. Using epidural electrodes in awake and freely moving wildtype (WT) and KO mice, we recorded auditory event related potentials (ERP) and auditory temporal processing with a gap-in-noise auditory steady state response (gap-ASSR) paradigm. Mice were recorded at three different ages in a cross sectional design: postnatal (p)21, p30 and p60. Recordings were obtained from both auditory and frontal cortices. The gap-ASSR requires underlying neural generators to synchronize responses to gaps of different widths embedded in noise, providing an objective measure of temporal processing across genotypes and age groups. RESULTS: We present evidence that the frontal, but not auditory, cortex shows significant temporal processing deficits at p21 and p30, with poor ability to phase lock to rapid gaps in noise. Temporal processing was similar in both genotypes in adult mice. ERP amplitudes were larger in Fmr1 KO mice in both auditory and frontal cortex, consistent with ERP data in humans with FXS. CONCLUSIONS: These data indicate cortical region-specific delays in temporal processing development in Fmr1 KO mice. Developmental delays in the ability of frontal cortex to follow rapid changes in sounds may shape language delays in FXS, and more broadly in ASD. |
| En ligne : |
https://dx.doi.org/10.1186/s11689-023-09496-8 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=575 |
in Journal of Neurodevelopmental Disorders > 15 (2023)
[article] Developmental delays in cortical auditory temporal processing in a mouse model of Fragile X syndrome [texte imprimé] / Katilynne CROOM, Auteur ; Jeffrey A. RUMSCHLAG, Auteur ; Michael A. ERICKSON, Auteur ; Devin K. BINDER, Auteur ; Khaleel A. RAZAK, Auteur. Langues : Anglais ( eng) in Journal of Neurodevelopmental Disorders > 15 (2023)
| Mots-clés : |
Humans Adult Animals Mice Fragile X Syndrome/complications Cross-Sectional Studies Time Perception Disease Models, Animal Mice, Knockout Fragile X Mental Retardation Protein/genetics Autism spectrum disorders Cerebral cortex Fragile X syndrome Language Neurodevelopment Sensory hypersensitivity Speech processing Temporal processing |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: Autism spectrum disorders (ASD) encompass a wide array of debilitating symptoms, including sensory dysfunction and delayed language development. Auditory temporal processing is crucial for speech perception and language development. Abnormal development of temporal processing may account for the language impairments associated with ASD. Very little is known about the development of temporal processing in any animal model of ASD. METHODS: In the current study, we quantify auditory temporal processing throughout development in the Fmr1 knock-out (KO) mouse model of Fragile X Syndrome (FXS), a leading genetic cause of intellectual disability and ASD-associated behaviors. Using epidural electrodes in awake and freely moving wildtype (WT) and KO mice, we recorded auditory event related potentials (ERP) and auditory temporal processing with a gap-in-noise auditory steady state response (gap-ASSR) paradigm. Mice were recorded at three different ages in a cross sectional design: postnatal (p)21, p30 and p60. Recordings were obtained from both auditory and frontal cortices. The gap-ASSR requires underlying neural generators to synchronize responses to gaps of different widths embedded in noise, providing an objective measure of temporal processing across genotypes and age groups. RESULTS: We present evidence that the frontal, but not auditory, cortex shows significant temporal processing deficits at p21 and p30, with poor ability to phase lock to rapid gaps in noise. Temporal processing was similar in both genotypes in adult mice. ERP amplitudes were larger in Fmr1 KO mice in both auditory and frontal cortex, consistent with ERP data in humans with FXS. CONCLUSIONS: These data indicate cortical region-specific delays in temporal processing development in Fmr1 KO mice. Developmental delays in the ability of frontal cortex to follow rapid changes in sounds may shape language delays in FXS, and more broadly in ASD. |
| En ligne : |
https://dx.doi.org/10.1186/s11689-023-09496-8 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=575 |
|  |
Developmental delays in cortical auditory temporal processing in a mouse model of Fragile X syndrome in Journal of Neurodevelopmental Disorders, 15 (2023)
