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Faire une suggestionLanguage Abilities of Russian Primary-School-Aged Children with Autism Spectrum Disorder: Evidence from Comprehensive Assessment / Vardan ARUTIUNIAN in Journal of Autism and Developmental Disorders, 52-2 (February 2022)
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Titre : Language Abilities of Russian Primary-School-Aged Children with Autism Spectrum Disorder: Evidence from Comprehensive Assessment Type de document : texte imprimé Auteurs : Vardan ARUTIUNIAN, Auteur ; Anastasiya LOPUKHINA, Auteur ; Alina MINNIGULOVA, Auteur ; Anastasia SHLYAKHOVA, Auteur ; Elizaveta DAVYDOVA, Auteur ; Darya PEREVERZEVA, Auteur ; Alexander SOROKIN, Auteur ; Svetlana TYUSHKEVICH, Auteur ; Uliana MAMOKHINA, Auteur ; Kamilla DANILINA, Auteur ; Olga DRAGOY, Auteur Article en page(s) : p.584-599 Langues : Anglais (eng) Mots-clés : Autism Spectrum Disorder/diagnosis Child Humans Language Language Development Disorders/diagnosis Language Tests Schools Autism Spectrum Disorder Language abilities Language comprehension Language production Russian Index. décimale : PER Périodiques Résumé : The purpose of the present research was to comprehensively assess the language abilities of Russian primary-school-aged children with Autism Spectrum Disorder (ASD), varying in non-verbal IQ, at all linguistic levels (phonology, lexicon, morphosyntax, and discourse) in production and comprehension. Yet, the influence of such non-language factors as children's age, the severity of autistic traits, and non-verbal IQ on language functioning was studied. Our results indicate a high variability of language skills in children with ASD (from normal to impaired) which is in line with the previous studies. Interestingly, the number of children with normal language abilities was related to the linguistic levels: according to more complex morphosyntax and discourse tests, fewer children with ASD were within the normal range unlike the results in simpler phonological and lexical tests. Importantly, we found that language abilities were best predicted by non-verbal IQ but were independent from age and the severity of autistic traits. The findings support the claim that formal language assessment of children with ASD needs to include all linguistic levels, from phonology to discourse, for helping speech-language therapists to choose an appropriate therapy target. En ligne : http://dx.doi.org/10.1007/s10803-021-04967-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=455
in Journal of Autism and Developmental Disorders > 52-2 (February 2022) . - p.584-599[article] Language Abilities of Russian Primary-School-Aged Children with Autism Spectrum Disorder: Evidence from Comprehensive Assessment [texte imprimé] / Vardan ARUTIUNIAN, Auteur ; Anastasiya LOPUKHINA, Auteur ; Alina MINNIGULOVA, Auteur ; Anastasia SHLYAKHOVA, Auteur ; Elizaveta DAVYDOVA, Auteur ; Darya PEREVERZEVA, Auteur ; Alexander SOROKIN, Auteur ; Svetlana TYUSHKEVICH, Auteur ; Uliana MAMOKHINA, Auteur ; Kamilla DANILINA, Auteur ; Olga DRAGOY, Auteur . - p.584-599.
Langues : Anglais (eng)
in Journal of Autism and Developmental Disorders > 52-2 (February 2022) . - p.584-599
Mots-clés : Autism Spectrum Disorder/diagnosis Child Humans Language Language Development Disorders/diagnosis Language Tests Schools Autism Spectrum Disorder Language abilities Language comprehension Language production Russian Index. décimale : PER Périodiques Résumé : The purpose of the present research was to comprehensively assess the language abilities of Russian primary-school-aged children with Autism Spectrum Disorder (ASD), varying in non-verbal IQ, at all linguistic levels (phonology, lexicon, morphosyntax, and discourse) in production and comprehension. Yet, the influence of such non-language factors as children's age, the severity of autistic traits, and non-verbal IQ on language functioning was studied. Our results indicate a high variability of language skills in children with ASD (from normal to impaired) which is in line with the previous studies. Interestingly, the number of children with normal language abilities was related to the linguistic levels: according to more complex morphosyntax and discourse tests, fewer children with ASD were within the normal range unlike the results in simpler phonological and lexical tests. Importantly, we found that language abilities were best predicted by non-verbal IQ but were independent from age and the severity of autistic traits. The findings support the claim that formal language assessment of children with ASD needs to include all linguistic levels, from phonology to discourse, for helping speech-language therapists to choose an appropriate therapy target. En ligne : http://dx.doi.org/10.1007/s10803-021-04967-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=455 Family history of FXTAS is associated with age-related cognitive-linguistic decline among mothers with the FMR1 premutation / Jessica KLUSEK in Journal of Neurodevelopmental Disorders, 14 (2022)
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[article]
Titre : Family history of FXTAS is associated with age-related cognitive-linguistic decline among mothers with the FMR1 premutation Type de document : texte imprimé Auteurs : Jessica KLUSEK, Auteur ; Amanda FAIRCHILD, Auteur ; Carly MOSER, Auteur ; Marsha R. MAILICK, Auteur ; Angela John THURMAN, Auteur ; Leonard ABBEDUTO, Auteur Langues : Anglais (eng) Mots-clés : Adult Alleles Ataxia/genetics Child Cognitive Dysfunction/complications/genetics Female Fragile X Mental Retardation Protein/genetics Fragile X Syndrome/complications/genetics Humans Language Disorders Middle Aged Mothers Neurodegenerative Diseases/complications/genetics Tremor/genetics Aging Fragile X premutation Grammatical complexity Language production trials from F. Hoffman-LaRoche, Ltd., Roche TCRC, Inc., Neuren Pharmaceuticals Limited, Inc, and the LuMind IDSC Foundation. AJT has received funding to develop and implement outcome measures from Fulcrum Therapeutic. MM serves as the chair of the Scientific Advisory Board of the John Merck Fund Developmental Disabilities Program. The authors have no other relevant conflicts of interest to disclose. Index. décimale : PER Périodiques Résumé : BACKGROUND: Women who carry a premutation allele of the FMR1 gene are at increased vulnerability to an array of age-related symptoms and disorders, including age-related decline in select cognitive skills. However, the risk factors for age-related decline are poorly understood, including the potential role of family history and genetic factors. In other forms of pathological aging, early decline in syntactic complexity is observed and predicts the later onset of neurodegenerative disease. To shed light on the earliest signs of degeneration, the present study characterized longitudinal changes in the syntactic complexity of women with the FMR1 premutation across midlife, and associations with family history of fragile X-associated tremor/ataxia syndrome (FXTAS) and CGG repeat length. METHODS: Forty-five women with the FMR1 premutation aged 35-64 years at study entry participated in 1-5 longitudinal assessments spaced approximately a year apart (130 observations total). All participants were mothers of children with confirmed fragile X syndrome. Language samples were analyzed for syntactic complexity and participants provided information on family history of FXTAS. CGG repeat length was determined via molecular genetic testing. RESULTS: Hierarchical linear models indicated that women who reported a family history of FXTAS exhibited faster age-related decline in syntactic complexity than those without a family history, with that difference emerging as the women reached their mid-50 s. CGG repeat length was not a significant predictor of age-related change. CONCLUSIONS: Results suggest that women with the FMR1 premutation who have a family history of FXTAS may be at increased risk for neurodegenerative disease, as indicated by age-related loss of syntactic complexity. Thus, family history of FXTAS may represent a personalized risk factor for age-related disease. Follow-up study is needed to determine whether syntactic decline is an early indicator of FXTAS specifically, as opposed to being a more general age-related cognitive decline associated with the FMR1 premutation. En ligne : https://dx.doi.org/10.1186/s11689-022-09415-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574
in Journal of Neurodevelopmental Disorders > 14 (2022)[article] Family history of FXTAS is associated with age-related cognitive-linguistic decline among mothers with the FMR1 premutation [texte imprimé] / Jessica KLUSEK, Auteur ; Amanda FAIRCHILD, Auteur ; Carly MOSER, Auteur ; Marsha R. MAILICK, Auteur ; Angela John THURMAN, Auteur ; Leonard ABBEDUTO, Auteur.
Langues : Anglais (eng)
in Journal of Neurodevelopmental Disorders > 14 (2022)
Mots-clés : Adult Alleles Ataxia/genetics Child Cognitive Dysfunction/complications/genetics Female Fragile X Mental Retardation Protein/genetics Fragile X Syndrome/complications/genetics Humans Language Disorders Middle Aged Mothers Neurodegenerative Diseases/complications/genetics Tremor/genetics Aging Fragile X premutation Grammatical complexity Language production trials from F. Hoffman-LaRoche, Ltd., Roche TCRC, Inc., Neuren Pharmaceuticals Limited, Inc, and the LuMind IDSC Foundation. AJT has received funding to develop and implement outcome measures from Fulcrum Therapeutic. MM serves as the chair of the Scientific Advisory Board of the John Merck Fund Developmental Disabilities Program. The authors have no other relevant conflicts of interest to disclose. Index. décimale : PER Périodiques Résumé : BACKGROUND: Women who carry a premutation allele of the FMR1 gene are at increased vulnerability to an array of age-related symptoms and disorders, including age-related decline in select cognitive skills. However, the risk factors for age-related decline are poorly understood, including the potential role of family history and genetic factors. In other forms of pathological aging, early decline in syntactic complexity is observed and predicts the later onset of neurodegenerative disease. To shed light on the earliest signs of degeneration, the present study characterized longitudinal changes in the syntactic complexity of women with the FMR1 premutation across midlife, and associations with family history of fragile X-associated tremor/ataxia syndrome (FXTAS) and CGG repeat length. METHODS: Forty-five women with the FMR1 premutation aged 35-64 years at study entry participated in 1-5 longitudinal assessments spaced approximately a year apart (130 observations total). All participants were mothers of children with confirmed fragile X syndrome. Language samples were analyzed for syntactic complexity and participants provided information on family history of FXTAS. CGG repeat length was determined via molecular genetic testing. RESULTS: Hierarchical linear models indicated that women who reported a family history of FXTAS exhibited faster age-related decline in syntactic complexity than those without a family history, with that difference emerging as the women reached their mid-50 s. CGG repeat length was not a significant predictor of age-related change. CONCLUSIONS: Results suggest that women with the FMR1 premutation who have a family history of FXTAS may be at increased risk for neurodegenerative disease, as indicated by age-related loss of syntactic complexity. Thus, family history of FXTAS may represent a personalized risk factor for age-related disease. Follow-up study is needed to determine whether syntactic decline is an early indicator of FXTAS specifically, as opposed to being a more general age-related cognitive decline associated with the FMR1 premutation. En ligne : https://dx.doi.org/10.1186/s11689-022-09415-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574

