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Developmental delays in cortical auditory temporal processing in a mouse model of Fragile X syndrome

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Publicado en:Journal of Neurodevelopmental Disorders vol. 15 (2023), p. 1
Autor principal: Croom, Katilynne
Otros Autores: Rumschlag, Jeffrey A, Erickson, Michael A, Binder, Devin K, Razak, Khaleel A
Publicado:
Springer Nature B.V.
Materias:
Child development
Language
Speech perception
Noise
Event-related potentials
Autism
Intellectual disabilities
Fragile X syndrome
Autistic children
Cortex (auditory)
Cortex (frontal)
Language disorders
Genotypes
Animal models
Phonology
Amplitude (Acoustics)
Delayed language acquisition
Surgery
Hypotheses
Ketamine
Epidural
Temporal processing
Hearing
Information processing
Speech
FMR1 protein
Sensory stimulation
Age groups
Delayed
Developmental delays
Somatosensory cortex
Data processing
Language acquisition
Cortex
Animals
Age differences
Genetics
Disorders
Language shift
Time
Temporal lobe
Postpartum period
Acceso en línea:Citation/Abstract
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Resumen:BackgroundAutism 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.MethodsIn 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.ResultsWe 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.ConclusionsThese 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.
ISSN:1866-1947
1866-1955
DOI:10.1186/s11689-023-09496-8
Fuente:Health & Medical Collection