Delineating the autistic phenotype in children with neurofibromatosis type 1 / Anita K. CHISHOLM in Molecular Autism, 13 (2022)  

Public ISBD [article]  inMolecular Autism > 13 (2022) . - 3p. Titre : Delineating the autistic phenotype in children with neurofibromatosis type 1 Type de document : texte imprimé Auteurs : Anita K. CHISHOLM, Auteur ; Kristina M. HAEBICH, Auteur ; Natalie A. PRIDE, Auteur ; Karin S. WALSH, Auteur ; Francesca LAMI, Auteur ; Alexandra URE, Auteur ; Tiba MALOOF, Auteur ; Amanda BRIGNELL, Auteur ; Melissa ROUEL, Auteur ; Yael GRANADER, Auteur ; Alice MAIER, Auteur ; Belinda BARTON, Auteur ; Hayley DARKE, Auteur ; Gabriel DABSCHECK, Auteur ; Vicki ANDERSON, Auteur ; Katrina WILLIAMS, Auteur ; Kathryn N. NORTH, Auteur ; Jonathan M. PAYNE, Auteur Article en page(s) : 3p. Langues : Anglais (eng) Mots-clés : Autism  Autism Diagnostic Interview-Revised (ADI-R)  Autism Diagnostic Observation Schedule-Second Edition (ADOS-2)  Autistic behaviours  Neurofibromatosis type 1 Index. décimale : PER Périodiques Résumé : BACKGROUND: Existing research has demonstrated elevated autistic behaviours in children with neurofibromatosis type 1 (NF1), but the autistic phenotype and its relationship to other neurodevelopmental manifestations of NF1 remains unclear. To address this gap, we performed detailed characterisation of autistic behaviours in children with NF1 and investigated their association with other common NF1 child characteristics. METHODS: Participants were drawn from a larger cross-sectional study examining autism in children with NF1. The population analysed in this study scored above threshold on the Social Responsiveness Scale-Second Edition (T-score ≥ 60; 51% larger cohort) and completed the Autism Diagnostic Interview-Revised (ADI-R) and/or the Autism Diagnostic Observation Schedule-Second Edition (ADOS-2). All participants underwent evaluation of their intellectual function, and behavioural data were collected via parent questionnaires. RESULTS: The study cohort comprised 68 children (3-15 years). Sixty-three per cent met the ADOS-2 'autism spectrum' cut-off, and 34% exceeded the more stringent threshold for 'autistic disorder' on the ADI-R. Social communication symptoms were common and wide-ranging, while restricted and repetitive behaviours (RRBs) were most commonly characterised by 'insistence on sameness' (IS) behaviours such as circumscribed interests and difficulties with minor changes. Autistic behaviours were weakly correlated with hyperactive/impulsive attention deficit hyperactivity disorder (ADHD) symptoms but not with inattentive ADHD or other behavioural characteristics. Language and verbal IQ were weakly related to social communication behaviours but not to RRBs. LIMITATIONS: Lack of genetic validation of NF1, no clinical diagnosis of autism, and a retrospective assessment of autistic behaviours in early childhood. CONCLUSIONS: Findings provide strong support for elevated autistic behaviours in children with NF1. While these behaviours were relatively independent of other NF1 comorbidities, the importance of taking broader child characteristics into consideration when interpreting data from autism-specific measures in this population is highlighted. Social communication deficits appear similar to those observed in idiopathic autism and are coupled with a unique RRB profile comprising prominent IS behaviours. This autistic phenotype and its relationship to common NF1 comorbidities such as anxiety and executive dysfunction will be important to examine in future research. Current findings have important implications for the early identification of autism in NF1 and clinical management. En ligne : http://dx.doi.org/10.1186/s13229-021-00481-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=459 [article] Delineating the autistic phenotype in children with neurofibromatosis type 1 [texte imprimé] / Anita K. CHISHOLM, Auteur ; Kristina M. HAEBICH, Auteur ; Natalie A. PRIDE, Auteur ; Karin S. WALSH, Auteur ; Francesca LAMI, Auteur ; Alexandra URE, Auteur ; Tiba MALOOF, Auteur ; Amanda BRIGNELL, Auteur ; Melissa ROUEL, Auteur ; Yael GRANADER, Auteur ; Alice MAIER, Auteur ; Belinda BARTON, Auteur ; Hayley DARKE, Auteur ; Gabriel DABSCHECK, Auteur ; Vicki ANDERSON, Auteur ; Katrina WILLIAMS, Auteur ; Kathryn N. NORTH, Auteur ; Jonathan M. PAYNE, Auteur . - 3p. Langues : Anglais (eng) in Molecular Autism > 13 (2022) . - 3p. Mots-clés : Autism  Autism Diagnostic Interview-Revised (ADI-R)  Autism Diagnostic Observation Schedule-Second Edition (ADOS-2)  Autistic behaviours  Neurofibromatosis type 1 Index. décimale : PER Périodiques Résumé : BACKGROUND: Existing research has demonstrated elevated autistic behaviours in children with neurofibromatosis type 1 (NF1), but the autistic phenotype and its relationship to other neurodevelopmental manifestations of NF1 remains unclear. To address this gap, we performed detailed characterisation of autistic behaviours in children with NF1 and investigated their association with other common NF1 child characteristics. METHODS: Participants were drawn from a larger cross-sectional study examining autism in children with NF1. The population analysed in this study scored above threshold on the Social Responsiveness Scale-Second Edition (T-score ≥ 60; 51% larger cohort) and completed the Autism Diagnostic Interview-Revised (ADI-R) and/or the Autism Diagnostic Observation Schedule-Second Edition (ADOS-2). All participants underwent evaluation of their intellectual function, and behavioural data were collected via parent questionnaires. RESULTS: The study cohort comprised 68 children (3-15 years). Sixty-three per cent met the ADOS-2 'autism spectrum' cut-off, and 34% exceeded the more stringent threshold for 'autistic disorder' on the ADI-R. Social communication symptoms were common and wide-ranging, while restricted and repetitive behaviours (RRBs) were most commonly characterised by 'insistence on sameness' (IS) behaviours such as circumscribed interests and difficulties with minor changes. Autistic behaviours were weakly correlated with hyperactive/impulsive attention deficit hyperactivity disorder (ADHD) symptoms but not with inattentive ADHD or other behavioural characteristics. Language and verbal IQ were weakly related to social communication behaviours but not to RRBs. LIMITATIONS: Lack of genetic validation of NF1, no clinical diagnosis of autism, and a retrospective assessment of autistic behaviours in early childhood. CONCLUSIONS: Findings provide strong support for elevated autistic behaviours in children with NF1. While these behaviours were relatively independent of other NF1 comorbidities, the importance of taking broader child characteristics into consideration when interpreting data from autism-specific measures in this population is highlighted. Social communication deficits appear similar to those observed in idiopathic autism and are coupled with a unique RRB profile comprising prominent IS behaviours. This autistic phenotype and its relationship to common NF1 comorbidities such as anxiety and executive dysfunction will be important to examine in future research. Current findings have important implications for the early identification of autism in NF1 and clinical management. En ligne : http://dx.doi.org/10.1186/s13229-021-00481-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=459

Delineating Visual Habituation Profiles in Preschoolers with Neurofibromatosis Type 1 and Autism Spectrum Disorder: A Cross-Syndrome Study / Darren R. HOCKING in Journal of Autism and Developmental Disorders, 54-5 (May 2024)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 54-5 (May 2024) . - p.1998-2011 Titre : Delineating Visual Habituation Profiles in Preschoolers with Neurofibromatosis Type 1 and Autism Spectrum Disorder: A Cross-Syndrome Study Type de document : texte imprimé Auteurs : Darren R. HOCKING, Auteur ; Xiaoyun SUN, Auteur ; Kristina M. HAEBICH, Auteur ; Hayley DARKE, Auteur ; Kathryn N. NORTH, Auteur ; Giacomo VIVANTI, Auteur ; Jonathan M. PAYNE, Auteur Article en page(s) : p.1998-2011 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Atypical habituation to repetitive information has been commonly reported in Autism Spectrum Disorder (ASD) but it is not yet clear whether similar abnormalities are present in Neurofibromatosis Type 1 (NF1). We employed a cross-syndrome design using a novel eye tracking paradigm to measure habituation in preschoolers with NF1, children with idiopathic ASD and typically developing (TD) children. Eye movements were recorded to examine fixation duration to simultaneously presented repeating and novel stimuli. Children with NF1 showed a bias for longer look durations to repeating stimuli at the expense of novel stimuli, and slower habituation in NF1 was associated with elevated ASD traits. These findings could indicate aberrant modulation of bottom-up attentional networks that interact with the emergence of ASD phenotypes. En ligne : https://doi.org/10.1007/s10803-023-05913-y Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=530 [article] Delineating Visual Habituation Profiles in Preschoolers with Neurofibromatosis Type 1 and Autism Spectrum Disorder: A Cross-Syndrome Study [texte imprimé] / Darren R. HOCKING, Auteur ; Xiaoyun SUN, Auteur ; Kristina M. HAEBICH, Auteur ; Hayley DARKE, Auteur ; Kathryn N. NORTH, Auteur ; Giacomo VIVANTI, Auteur ; Jonathan M. PAYNE, Auteur . - p.1998-2011. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 54-5 (May 2024) . - p.1998-2011 Index. décimale : PER Périodiques Résumé : Atypical habituation to repetitive information has been commonly reported in Autism Spectrum Disorder (ASD) but it is not yet clear whether similar abnormalities are present in Neurofibromatosis Type 1 (NF1). We employed a cross-syndrome design using a novel eye tracking paradigm to measure habituation in preschoolers with NF1, children with idiopathic ASD and typically developing (TD) children. Eye movements were recorded to examine fixation duration to simultaneously presented repeating and novel stimuli. Children with NF1 showed a bias for longer look durations to repeating stimuli at the expense of novel stimuli, and slower habituation in NF1 was associated with elevated ASD traits. These findings could indicate aberrant modulation of bottom-up attentional networks that interact with the emergence of ASD phenotypes. En ligne : https://doi.org/10.1007/s10803-023-05913-y Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=530

Sex- and age-related differences in autistic behaviours in children with neurofibromatosis type 1 / Anita K. CHISHOLM in Journal of Autism and Developmental Disorders, 53-7 (July 2023)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 53-7 (July 2023) . - p.2835-2850 Titre : Sex- and age-related differences in autistic behaviours in children with neurofibromatosis type 1 Type de document : texte imprimé Auteurs : Anita K. CHISHOLM, Auteur ; Francesca LAMI, Auteur ; Kristina M. HAEBICH, Auteur ; Alex URE, Auteur ; Amanda BRIGNELL, Auteur ; Tiba MALOOF, Auteur ; Natalie A. PRIDE, Auteur ; Karin S. WALSH, Auteur ; Alice MAIER, Auteur ; Melissa ROUEL, Auteur ; Yael GRANADER, Auteur ; Belinda BARTON, Auteur ; Hayley DARKE, Auteur ; Ian FUELSCHER, Auteur ; Gabriel DABSCHECK, Auteur ; Vicki ANDERSON, Auteur ; Katrina WILLIAMS, Auteur ; Kathryn N. NORTH, Auteur ; Jonathan M. PAYNE, Auteur Article en page(s) : p.2835-2850 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : This study investigated sex and age differences in autistic behaviours in children with neurofibromatosis type 1 (NF1) who scored within the clinical range on the Social Responsiveness Scale - Second Edition (T score ? 60). Thirty-four males and 28 females (3-16 years) were assessed with the Autism Diagnostic Observation Schedule - Second Edition and Autism Diagnostic Interview - Revised. Across both measures, males exhibited greater social communication deficits relative to females. Age-related abatement of social communication difficulties was observed for males but not females. Conversely, no sex differences were found for restricted/repetitive behaviours, which were stable over time for both males and females. The findings are discussed within the context of broader neurodevelopmental considerations that are common in NF1. En ligne : https://doi.org/10.1007/s10803-022-05571-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=508 [article] Sex- and age-related differences in autistic behaviours in children with neurofibromatosis type 1 [texte imprimé] / Anita K. CHISHOLM, Auteur ; Francesca LAMI, Auteur ; Kristina M. HAEBICH, Auteur ; Alex URE, Auteur ; Amanda BRIGNELL, Auteur ; Tiba MALOOF, Auteur ; Natalie A. PRIDE, Auteur ; Karin S. WALSH, Auteur ; Alice MAIER, Auteur ; Melissa ROUEL, Auteur ; Yael GRANADER, Auteur ; Belinda BARTON, Auteur ; Hayley DARKE, Auteur ; Ian FUELSCHER, Auteur ; Gabriel DABSCHECK, Auteur ; Vicki ANDERSON, Auteur ; Katrina WILLIAMS, Auteur ; Kathryn N. NORTH, Auteur ; Jonathan M. PAYNE, Auteur . - p.2835-2850. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 53-7 (July 2023) . - p.2835-2850 Index. décimale : PER Périodiques Résumé : This study investigated sex and age differences in autistic behaviours in children with neurofibromatosis type 1 (NF1) who scored within the clinical range on the Social Responsiveness Scale - Second Edition (T score ? 60). Thirty-four males and 28 females (3-16 years) were assessed with the Autism Diagnostic Observation Schedule - Second Edition and Autism Diagnostic Interview - Revised. Across both measures, males exhibited greater social communication deficits relative to females. Age-related abatement of social communication difficulties was observed for males but not females. Conversely, no sex differences were found for restricted/repetitive behaviours, which were stable over time for both males and females. The findings are discussed within the context of broader neurodevelopmental considerations that are common in NF1. En ligne : https://doi.org/10.1007/s10803-022-05571-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=508

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