Age-Dependent Effects of Loss of Contactin-Associated Protein-Like 2, an Autism-Associated Gene, on the Acquisition and Recall of Fear Memory / A. BERNS ; Myles JONES ; A. TOWNSEND ; A.K. EAGEN ; Sarah L. FERRI ; D.R. LANGBEHN ; H. JANOUSCHEK in Autism Research, 18-5 (May 2025)  

Public ISBD [article]  inAutism Research > 18-5 (May 2025) . - p.1011-1023 Titre : Age-Dependent Effects of Loss of Contactin-Associated Protein-Like 2, an Autism-Associated Gene, on the Acquisition and Recall of Fear Memory Type de document : texte imprimé Auteurs : A. BERNS, Auteur ; Myles JONES, Auteur ; A. TOWNSEND, Auteur ; A.K. EAGEN, Auteur ; Sarah L. FERRI, Auteur ; D.R. LANGBEHN, Auteur ; H. JANOUSCHEK, Auteur Article en page(s) : p.1011-1023 Langues : Anglais (eng) Mots-clés : anxiety  autism spectrum disorder  CASPR2  Cntnap2  development  fear  fear conditioning  memory Index. décimale : PER Périodiques Résumé : ABSTRACT The contactin-associated protein-like 2 (Cntnap2) gene is relevant to autism spectrum disorder (ASD), which is associated with age-specific structural alterations in limbic brain regions. The Cntnap2 gene encodes for the contactin-associated protein-like 2 (CASPR2) protein, and CASPR2 protein levels are high in the amygdala, a limbic region that is essential for the processing of fear and anxiety. In humans, reduced levels of this protein arising from CNTNAP2 mutations could potentially account for the autism-associated increase in fear and anxiety. Here, we report the extent to which loss of CASPR2 in mice contributes to the development of fear- and anxiety-related behaviors. Pavlovian fear conditioning experiments revealed that loss of CASPR2 has age-dependent effects on the acquisition of fear memory, recall of both cue-evoked and context-related fear memory, and stability of cue-evoked fear memory. Additionally, data from the elevated zero maze suggest that CASPR2 deficiency contributes to anxiety-related behaviors, especially in juvenile (29-day old) mice. These are the first reports of age-dependent effects of CASPR2 deficiency on fear and anxiety-related behaviors, and they set the stage for a better understanding of developmental alterations of fear and anxiety in ASD. En ligne : https://doi.org/10.1002/aur.70034 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=558 [article] Age-Dependent Effects of Loss of Contactin-Associated Protein-Like 2, an Autism-Associated Gene, on the Acquisition and Recall of Fear Memory [texte imprimé] / A. BERNS, Auteur ; Myles JONES, Auteur ; A. TOWNSEND, Auteur ; A.K. EAGEN, Auteur ; Sarah L. FERRI, Auteur ; D.R. LANGBEHN, Auteur ; H. JANOUSCHEK, Auteur . - p.1011-1023. Langues : Anglais (eng) in Autism Research > 18-5 (May 2025) . - p.1011-1023 Mots-clés : anxiety  autism spectrum disorder  CASPR2  Cntnap2  development  fear  fear conditioning  memory Index. décimale : PER Périodiques Résumé : ABSTRACT The contactin-associated protein-like 2 (Cntnap2) gene is relevant to autism spectrum disorder (ASD), which is associated with age-specific structural alterations in limbic brain regions. The Cntnap2 gene encodes for the contactin-associated protein-like 2 (CASPR2) protein, and CASPR2 protein levels are high in the amygdala, a limbic region that is essential for the processing of fear and anxiety. In humans, reduced levels of this protein arising from CNTNAP2 mutations could potentially account for the autism-associated increase in fear and anxiety. Here, we report the extent to which loss of CASPR2 in mice contributes to the development of fear- and anxiety-related behaviors. Pavlovian fear conditioning experiments revealed that loss of CASPR2 has age-dependent effects on the acquisition of fear memory, recall of both cue-evoked and context-related fear memory, and stability of cue-evoked fear memory. Additionally, data from the elevated zero maze suggest that CASPR2 deficiency contributes to anxiety-related behaviors, especially in juvenile (29-day old) mice. These are the first reports of age-dependent effects of CASPR2 deficiency on fear and anxiety-related behaviors, and they set the stage for a better understanding of developmental alterations of fear and anxiety in ASD. En ligne : https://doi.org/10.1002/aur.70034 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=558

Atypical neural variability in carriers of 16p11.2 copy number variants / Reem AL-JAWAHIRI in Autism Research, 12-9 (September 2019)  

Public ISBD [article]  inAutism Research > 12-9 (September 2019) . - p.1322-1333 Titre : Atypical neural variability in carriers of 16p11.2 copy number variants Type de document : texte imprimé Auteurs : Reem AL-JAWAHIRI, Auteur ; Myles JONES, Auteur ; Elizabeth MILNE, Auteur Article en page(s) : p.1322-1333 Langues : Anglais (eng) Mots-clés : alpha rhythm  cognitive neuroscience  copy number variation/copy number variants  electroencephalography  event-related potentials  gene-dosage effect  genetic/genomic syndromes Index. décimale : PER Périodiques Résumé : Copy number variations (CNVs) at the 16p11.2 chromosomal region are associated with myriad clinical features including intellectual disability and autism spectrum disorder. The aim of this study is to determine whether 16p11.2 deletion (DEL) and duplication (DUP) carriers demonstrate a distinct and reciprocal pattern of electroencephalography (EEG) activity as represented by neural variability measures. EEG data were previously collected as part of the Simons Variation in Individuals Project. Variability measures, as estimated by single-trial ERP and spectral power analyses in the alpha and beta frequency bands, in addition to signal-to-noise ratios (SNRs), were analyzed in DEL (n = 20), DUP (n = 8), and typical (n = 11) groups. We also analyzed mean visual evoked potentials and spectral power (alpha and beta power) to facilitate comparisons with other studies of associated disorders and CNVs. From measures of single-trial variability, we found higher intraparticipant variability in P1 amplitude and timecourse amplitude in DEL compared to controls. Compared to DUP, DEL showed higher variability in absolute alpha and absolute beta power but lower variability in P1 latency. SNRs did not differ between the groups. From measures of amplitude, latency, and spectral power, DUP showed lower relative alpha power compared to controls. Although it is yet unclear whether 16p11.2 CNV dosage impacts neural activity in an opposing manner, findings suggest that 16p11.2 DEL impacts the level of variability of neural responses. Higher neural variability may play a role in a range of cognitive processes in 16p11.2 CNV carriers. Autism Res 2019, 12: 1322-1333. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: The study analyzed the consistency of patterns of brain waves and rhythms in those affected with a loss or gain of DNA material in the 16p11.2 region. Compared with typical individuals, 16p11.2 deletion carriers showed greater inconsistency in the way the brain responds to the same visual event. This high inconsistency in brain activity may play a role in some core symptoms in 16p11.2 copy number variation carriers. En ligne : http://dx.doi.org/10.1002/aur.2166 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=406 [article] Atypical neural variability in carriers of 16p11.2 copy number variants [texte imprimé] / Reem AL-JAWAHIRI, Auteur ; Myles JONES, Auteur ; Elizabeth MILNE, Auteur . - p.1322-1333. Langues : Anglais (eng) in Autism Research > 12-9 (September 2019) . - p.1322-1333 Mots-clés : alpha rhythm  cognitive neuroscience  copy number variation/copy number variants  electroencephalography  event-related potentials  gene-dosage effect  genetic/genomic syndromes Index. décimale : PER Périodiques Résumé : Copy number variations (CNVs) at the 16p11.2 chromosomal region are associated with myriad clinical features including intellectual disability and autism spectrum disorder. The aim of this study is to determine whether 16p11.2 deletion (DEL) and duplication (DUP) carriers demonstrate a distinct and reciprocal pattern of electroencephalography (EEG) activity as represented by neural variability measures. EEG data were previously collected as part of the Simons Variation in Individuals Project. Variability measures, as estimated by single-trial ERP and spectral power analyses in the alpha and beta frequency bands, in addition to signal-to-noise ratios (SNRs), were analyzed in DEL (n = 20), DUP (n = 8), and typical (n = 11) groups. We also analyzed mean visual evoked potentials and spectral power (alpha and beta power) to facilitate comparisons with other studies of associated disorders and CNVs. From measures of single-trial variability, we found higher intraparticipant variability in P1 amplitude and timecourse amplitude in DEL compared to controls. Compared to DUP, DEL showed higher variability in absolute alpha and absolute beta power but lower variability in P1 latency. SNRs did not differ between the groups. From measures of amplitude, latency, and spectral power, DUP showed lower relative alpha power compared to controls. Although it is yet unclear whether 16p11.2 CNV dosage impacts neural activity in an opposing manner, findings suggest that 16p11.2 DEL impacts the level of variability of neural responses. Higher neural variability may play a role in a range of cognitive processes in 16p11.2 CNV carriers. Autism Res 2019, 12: 1322-1333. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: The study analyzed the consistency of patterns of brain waves and rhythms in those affected with a loss or gain of DNA material in the 16p11.2 region. Compared with typical individuals, 16p11.2 deletion carriers showed greater inconsistency in the way the brain responds to the same visual event. This high inconsistency in brain activity may play a role in some core symptoms in 16p11.2 copy number variation carriers. En ligne : http://dx.doi.org/10.1002/aur.2166 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=406

Oblique Orientation Discrimination Thresholds Are Superior in Those with a High Level of Autistic Traits / Abigail DICKINSON in Journal of Autism and Developmental Disorders, 44-11 (November 2014)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 44-11 (November 2014) . - p.2844-2850 Titre : Oblique Orientation Discrimination Thresholds Are Superior in Those with a High Level of Autistic Traits Type de document : texte imprimé Auteurs : Abigail DICKINSON, Auteur ; Myles JONES, Auteur ; Elizabeth MILNE, Auteur Article en page(s) : p.2844-2850 Langues : Anglais (eng) Mots-clés : Autistic traits  Orientation discrimination  Visual perception Index. décimale : PER Périodiques Résumé : Enhanced low-level perception, although present in individuals with autism, is not seen in individuals with high, but non-clinical, levels of autistic traits (Brock et al.in Percept Lond 40(6):739. doi:10.1068/p6953, 2011). This is surprising, as many of the higher-level visual differences found in autism have been shown to correlate with autistic traits in non-clinical samples. Here we measure vertical–oblique and, more difficult, oblique–oblique orientation discrimination thresholds in a non-clinical sample. As predicted, oblique–oblique thresholds provided a more sensitive test of orientation discrimination, and were negatively related to autistic traits (N = 94, r = −.356, p .0001). We conclude that individual differences in orientation discrimination and autistic traits are related, and suggest that both of these factors could be mediated by increased levels of the inhibitory neurotransmitter GABA. En ligne : http://dx.doi.org/10.1007/s10803-014-2147-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=241 [article] Oblique Orientation Discrimination Thresholds Are Superior in Those with a High Level of Autistic Traits [texte imprimé] / Abigail DICKINSON, Auteur ; Myles JONES, Auteur ; Elizabeth MILNE, Auteur . - p.2844-2850. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 44-11 (November 2014) . - p.2844-2850 Mots-clés : Autistic traits  Orientation discrimination  Visual perception Index. décimale : PER Périodiques Résumé : Enhanced low-level perception, although present in individuals with autism, is not seen in individuals with high, but non-clinical, levels of autistic traits (Brock et al.in Percept Lond 40(6):739. doi:10.1068/p6953, 2011). This is surprising, as many of the higher-level visual differences found in autism have been shown to correlate with autistic traits in non-clinical samples. Here we measure vertical–oblique and, more difficult, oblique–oblique orientation discrimination thresholds in a non-clinical sample. As predicted, oblique–oblique thresholds provided a more sensitive test of orientation discrimination, and were negatively related to autistic traits (N = 94, r = −.356, p .0001). We conclude that individual differences in orientation discrimination and autistic traits are related, and suggest that both of these factors could be mediated by increased levels of the inhibitory neurotransmitter GABA. En ligne : http://dx.doi.org/10.1007/s10803-014-2147-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=241

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