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Faire une suggestionCognitive and Electrophysiological Correlates of Working Memory Impairments in Neurofibromatosis Type 1 / Gorana POBRIC in Journal of Autism and Developmental Disorders, 52-4 (April 2022)
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Titre : Cognitive and Electrophysiological Correlates of Working Memory Impairments in Neurofibromatosis Type 1 Type de document : texte imprimé Auteurs : Gorana POBRIC, Auteur ; Jason R. TAYLOR, Auteur ; Hemavathy M. RAMALINGAM, Auteur ; Emily PYE, Auteur ; Louise ROBINSON, Auteur ; Grace VASSALLO, Auteur ; JeYoung JUNG, Auteur ; Misty BHANDARY, Auteur ; Karolina SZUMANSKA-RYT, Auteur ; Louise THEODOSIOU, Auteur ; D. Gareth EVANS, Auteur ; Judith EELLOO, Auteur ; Emma BURKITT-WRIGHT, Auteur ; Johan HULLEMAN, Auteur ; Jonathan GREEN, Auteur ; Shruti GARG, Auteur Article en page(s) : p.1478-1494 Langues : Anglais (eng) Mots-clés : Adolescent Autism Spectrum Disorder Cognition Evoked Potentials/physiology Humans Memory, Short-Term/physiology Neurofibromatosis 1/complications Eeg N-back task Neurofibromatosis 1 P300 Working memory Index. décimale : PER Périodiques Résumé : Neurofibromatosis 1 (NF1) is a single gene disorder associated with working Memory (WM) impairments. The aim of this study was to investigate P300 event-related potential (ERP) associated with WM in NF1. Sixteen adolescents with NF1 were compared with controls on measures of WM and EEG was recorded during a WM nback task. The NF1 group showed poorer performance on measures of WM as compared to the control group. No group differences were observed in P300 amplitude at Pz, but P300 latency was shorter in the NF1 group. Topographic analyses of P300 amplitude showed group differences indicating neural processing differences in the NF1 group relative to controls, which possibly contribute to the cognitive deficits seen in this population. En ligne : http://dx.doi.org/10.1007/s10803-021-05043-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=475
in Journal of Autism and Developmental Disorders > 52-4 (April 2022) . - p.1478-1494[article] Cognitive and Electrophysiological Correlates of Working Memory Impairments in Neurofibromatosis Type 1 [texte imprimé] / Gorana POBRIC, Auteur ; Jason R. TAYLOR, Auteur ; Hemavathy M. RAMALINGAM, Auteur ; Emily PYE, Auteur ; Louise ROBINSON, Auteur ; Grace VASSALLO, Auteur ; JeYoung JUNG, Auteur ; Misty BHANDARY, Auteur ; Karolina SZUMANSKA-RYT, Auteur ; Louise THEODOSIOU, Auteur ; D. Gareth EVANS, Auteur ; Judith EELLOO, Auteur ; Emma BURKITT-WRIGHT, Auteur ; Johan HULLEMAN, Auteur ; Jonathan GREEN, Auteur ; Shruti GARG, Auteur . - p.1478-1494.
Langues : Anglais (eng)
in Journal of Autism and Developmental Disorders > 52-4 (April 2022) . - p.1478-1494
Mots-clés : Adolescent Autism Spectrum Disorder Cognition Evoked Potentials/physiology Humans Memory, Short-Term/physiology Neurofibromatosis 1/complications Eeg N-back task Neurofibromatosis 1 P300 Working memory Index. décimale : PER Périodiques Résumé : Neurofibromatosis 1 (NF1) is a single gene disorder associated with working Memory (WM) impairments. The aim of this study was to investigate P300 event-related potential (ERP) associated with WM in NF1. Sixteen adolescents with NF1 were compared with controls on measures of WM and EEG was recorded during a WM nback task. The NF1 group showed poorer performance on measures of WM as compared to the control group. No group differences were observed in P300 amplitude at Pz, but P300 latency was shorter in the NF1 group. Topographic analyses of P300 amplitude showed group differences indicating neural processing differences in the NF1 group relative to controls, which possibly contribute to the cognitive deficits seen in this population. En ligne : http://dx.doi.org/10.1007/s10803-021-05043-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=475 Developmental trajectories in infants and pre-school children with Neurofibromatosis 1 / Hannah SLEVIN in Molecular Autism, 15 (2024)
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Titre : Developmental trajectories in infants and pre-school children with Neurofibromatosis 1 Type de document : texte imprimé Auteurs : Hannah SLEVIN, Auteur ; Fiona KEHINDE, Auteur ; Jannath BEGUM-ALI, Auteur ; Ceri ELLIS, Auteur ; Emma BURKITT-WRIGHT, Auteur ; Jonathan GREEN, Auteur ; Mark H. JOHNSON, Auteur ; Greg PASCO, Auteur ; Tony CHARMAN, Auteur ; Emily J.H. JONES, Auteur ; Shruti GARG, Auteur ; EDEN-STAARS TEAM, Auteur Article en page(s) : 45p. Langues : Anglais (eng) Mots-clés : Humans Neurofibromatosis 1 Infant Female Male Child, Preschool Cognition Child Development Attention Deficit Disorder with Hyperactivity/diagnosis Prospective Studies Autistic Disorder/diagnosis Adhd Autism Children Cohort Nf1 Neurofibromatosis Trajectories has received royalties from Sage Publications and Guilford Publications. The other authors have no conflicts of interest to disclose. Index. décimale : PER Périodiques Résumé : BACKGROUND: Children with Neurofibromatosis 1 (NF1) show cognitive, behavioural and social differences compared to their peers. However, the age and sequence at which these differences begin to emerge is not fully understood. This prospective cohort study examines the cognitive, behavioural, ADHD trait and autism symptom development in infant and pre-school children with NF1 compared with typically developing (TD) children without a family history of neurodevelopmental conditions. METHODS: Data from standardised tests was gathered at 5, 10, 14, 24 and 36 months of age (NF1 n = 35, TD n = 29). Developmental trajectories of cognitive (Mullen Scales of Early Learning, MSEL) and adaptive behavioural (Vineland Adaptive Behavior Scales, VABS) development from 5 to 36 months were analysed using linear mixed modelling. Measures of ADHD (Child Behavior Checklist) and autism traits (ADOS-2, BOSA-MV and ADI-R) were assessed at 24 and 36 months. RESULTS: The developmental trajectory of cognitive skills (all domains of the MSEL) and behavioural skills (four domains of the VABS) differed significantly between NF1 and TD groups. Post-hoc tests demonstrated that the NF1 participants scored significantly lower than TD participants at 24 months on all MSEL and VABS domains. The NF1 cohort demonstrated higher mean autism and ADHD traits at 24 months and 14% of the NF1 cohort met a research diagnostic classification for autism at 36 months. LIMITATIONS: The study has a relatively small sample size due to variable retention and rolling recruitment. Due to limitations imposed by the COVID-19 pandemic, we utilised the Brief Observation of Symptoms of Autism for Minimally Verbal children (BOSA-MV) for some participants, which was administered online and may not gather as accurate a picture of traits as ADOS-2. The BOSA-MV was utilised for 41% of participants with NF1 at 36 months compared to 11% at 24 months. This may explain the reduction in the percentage of children with NF1 that met autism criteria at 36 months. CONCLUSIONS: By 24 months of age, the NF1 cohort show lower cognitive skills and adaptive behaviour and higher levels of autism and ADHD traits as compared to TD children. This has implications for developmental monitoring and referral for early interventions. TRIAL REGISTRATION: Not applicable. En ligne : https://dx.doi.org/10.1186/s13229-024-00621-5 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=538
in Molecular Autism > 15 (2024) . - 45p.[article] Developmental trajectories in infants and pre-school children with Neurofibromatosis 1 [texte imprimé] / Hannah SLEVIN, Auteur ; Fiona KEHINDE, Auteur ; Jannath BEGUM-ALI, Auteur ; Ceri ELLIS, Auteur ; Emma BURKITT-WRIGHT, Auteur ; Jonathan GREEN, Auteur ; Mark H. JOHNSON, Auteur ; Greg PASCO, Auteur ; Tony CHARMAN, Auteur ; Emily J.H. JONES, Auteur ; Shruti GARG, Auteur ; EDEN-STAARS TEAM, Auteur . - 45p.
Langues : Anglais (eng)
in Molecular Autism > 15 (2024) . - 45p.
Mots-clés : Humans Neurofibromatosis 1 Infant Female Male Child, Preschool Cognition Child Development Attention Deficit Disorder with Hyperactivity/diagnosis Prospective Studies Autistic Disorder/diagnosis Adhd Autism Children Cohort Nf1 Neurofibromatosis Trajectories has received royalties from Sage Publications and Guilford Publications. The other authors have no conflicts of interest to disclose. Index. décimale : PER Périodiques Résumé : BACKGROUND: Children with Neurofibromatosis 1 (NF1) show cognitive, behavioural and social differences compared to their peers. However, the age and sequence at which these differences begin to emerge is not fully understood. This prospective cohort study examines the cognitive, behavioural, ADHD trait and autism symptom development in infant and pre-school children with NF1 compared with typically developing (TD) children without a family history of neurodevelopmental conditions. METHODS: Data from standardised tests was gathered at 5, 10, 14, 24 and 36 months of age (NF1 n = 35, TD n = 29). Developmental trajectories of cognitive (Mullen Scales of Early Learning, MSEL) and adaptive behavioural (Vineland Adaptive Behavior Scales, VABS) development from 5 to 36 months were analysed using linear mixed modelling. Measures of ADHD (Child Behavior Checklist) and autism traits (ADOS-2, BOSA-MV and ADI-R) were assessed at 24 and 36 months. RESULTS: The developmental trajectory of cognitive skills (all domains of the MSEL) and behavioural skills (four domains of the VABS) differed significantly between NF1 and TD groups. Post-hoc tests demonstrated that the NF1 participants scored significantly lower than TD participants at 24 months on all MSEL and VABS domains. The NF1 cohort demonstrated higher mean autism and ADHD traits at 24 months and 14% of the NF1 cohort met a research diagnostic classification for autism at 36 months. LIMITATIONS: The study has a relatively small sample size due to variable retention and rolling recruitment. Due to limitations imposed by the COVID-19 pandemic, we utilised the Brief Observation of Symptoms of Autism for Minimally Verbal children (BOSA-MV) for some participants, which was administered online and may not gather as accurate a picture of traits as ADOS-2. The BOSA-MV was utilised for 41% of participants with NF1 at 36 months compared to 11% at 24 months. This may explain the reduction in the percentage of children with NF1 that met autism criteria at 36 months. CONCLUSIONS: By 24 months of age, the NF1 cohort show lower cognitive skills and adaptive behaviour and higher levels of autism and ADHD traits as compared to TD children. This has implications for developmental monitoring and referral for early interventions. TRIAL REGISTRATION: Not applicable. En ligne : https://dx.doi.org/10.1186/s13229-024-00621-5 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=538 Early differences in auditory processing relate to Autism Spectrum Disorder traits in infants with Neurofibromatosis Type I / Jannath BEGUM-ALI in Journal of Neurodevelopmental Disorders, 13 (2021)
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Titre : Early differences in auditory processing relate to Autism Spectrum Disorder traits in infants with Neurofibromatosis Type I Type de document : texte imprimé Auteurs : Jannath BEGUM-ALI, Auteur ; Anna KOLESNIK-TAYLOR, Auteur ; Isabel QUIROZ, Auteur ; Luke MASON, Auteur ; Shruti GARG, Auteur ; Jonathan GREEN, Auteur ; Mark H. JOHNSON, Auteur ; Emily J.H. JONES, Auteur ; STAARS AND EDEN TEAMS, Auteur Langues : Anglais (eng) Mots-clés : Auditory Perception Autism Spectrum Disorder Humans Infant Longitudinal Studies Neurofibromatosis 1 Prospective Studies Auditory processing Autism spectrum disorder Change detection Eeg Habituation Neurofibromatosis type 1 Index. décimale : PER Périodiques Résumé : BACKGROUND: Sensory modulation difficulties are common in children with conditions such as Autism Spectrum Disorder (ASD) and could contribute to other social and non-social symptoms. Positing a causal role for sensory processing differences requires observing atypical sensory reactivity prior to the emergence of other symptoms, which can be achieved through prospective studies. METHODS: In this longitudinal study, we examined auditory repetition suppression and change detection at 5 and 10 months in infants with and without Neurofibromatosis Type 1 (NF1), a condition associated with higher likelihood of developing ASD. RESULTS: In typically developing infants, suppression to vowel repetition and enhanced responses to vowel/pitch change decreased with age over posterior regions, becoming more frontally specific; age-related change was diminished in the NF1 group. Whilst both groups detected changes in vowel and pitch, the NF1 group were largely slower to show a differentiated neural response. Auditory responses did not relate to later language, but were related to later ASD traits. CONCLUSIONS: These findings represent the first demonstration of atypical brain responses to sounds in infants with NF1 and suggest they may relate to the likelihood of later ASD. En ligne : https://dx.doi.org/10.1186/s11689-021-09364-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574
in Journal of Neurodevelopmental Disorders > 13 (2021)[article] Early differences in auditory processing relate to Autism Spectrum Disorder traits in infants with Neurofibromatosis Type I [texte imprimé] / Jannath BEGUM-ALI, Auteur ; Anna KOLESNIK-TAYLOR, Auteur ; Isabel QUIROZ, Auteur ; Luke MASON, Auteur ; Shruti GARG, Auteur ; Jonathan GREEN, Auteur ; Mark H. JOHNSON, Auteur ; Emily J.H. JONES, Auteur ; STAARS AND EDEN TEAMS, Auteur.
Langues : Anglais (eng)
in Journal of Neurodevelopmental Disorders > 13 (2021)
Mots-clés : Auditory Perception Autism Spectrum Disorder Humans Infant Longitudinal Studies Neurofibromatosis 1 Prospective Studies Auditory processing Autism spectrum disorder Change detection Eeg Habituation Neurofibromatosis type 1 Index. décimale : PER Périodiques Résumé : BACKGROUND: Sensory modulation difficulties are common in children with conditions such as Autism Spectrum Disorder (ASD) and could contribute to other social and non-social symptoms. Positing a causal role for sensory processing differences requires observing atypical sensory reactivity prior to the emergence of other symptoms, which can be achieved through prospective studies. METHODS: In this longitudinal study, we examined auditory repetition suppression and change detection at 5 and 10 months in infants with and without Neurofibromatosis Type 1 (NF1), a condition associated with higher likelihood of developing ASD. RESULTS: In typically developing infants, suppression to vowel repetition and enhanced responses to vowel/pitch change decreased with age over posterior regions, becoming more frontally specific; age-related change was diminished in the NF1 group. Whilst both groups detected changes in vowel and pitch, the NF1 group were largely slower to show a differentiated neural response. Auditory responses did not relate to later language, but were related to later ASD traits. CONCLUSIONS: These findings represent the first demonstration of atypical brain responses to sounds in infants with NF1 and suggest they may relate to the likelihood of later ASD. En ligne : https://dx.doi.org/10.1186/s11689-021-09364-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574

