Expressive language development in adolescents with Down syndrome and fragile X syndrome: change over time and the role of family-related factors / Laura DEL HOYO SORIANO in Journal of Neurodevelopmental Disorders, 12 (2020)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 12 (2020) Titre : Expressive language development in adolescents with Down syndrome and fragile X syndrome: change over time and the role of family-related factors Type de document : texte imprimé Auteurs : Laura DEL HOYO SORIANO, Auteur ; Angela John THURMAN, Auteur ; Danielle HARVEY, Auteur ; Sara T. KOVER, Auteur ; Leonard ABBEDUTO, Auteur Langues : Anglais (eng) Mots-clés : Adolescent  Child  Cognition  Down Syndrome/psychology  Family/psychology  Fragile X Syndrome/psychology  Humans  Intellectual Disability/psychology  Language Development  Language Development Disorders/complications  Language Tests  Longitudinal Studies  Male  Mother-Child Relations/psychology  Mothers/psychology  Vocabulary  Adolescence  Conversation  Down syndrome  Expressive language development  Family-related factors  Fragile X syndrome  Longitudinal  Narration Index. décimale : PER Périodiques Résumé : BACKGROUND: It is well known that individuals with Down syndrome (DS) or fragile X syndrome (FXS) demonstrate expressive language difficulties beginning early in childhood. It is less clear, however, whether expressive language skills change during the adolescent period in these individuals, and if any of these changes are syndrome specific. Studying this, as well as the role of maternal and family-related factors in expressive language development, may provide the foundation for efficacious interventions for adolescents with DS or FXS. METHODS: In this study, we examined expressive language trajectories, assessed through conversation and narration, in 57 adolescent males with intellectual disability (ID) (20 DS and 37 FXS) in relation to the diagnostic group (DS vs. FXS) and family-related factors (maternal IQ, maternal psychological distress, closeness in the mother-child relationship, family income, and maternal and paternal education) after adjusting for chronological age (CA) and nonverbal cognition. RESULTS: Changes over repeated annual assessments for males with DS or FXS were observed only during conversation, such as an increase in talkativeness, but a decrease in syntax complexity and lexical diversity. We found a diagnosis-related effect in the change over time in conversational talkativeness favoring those with FXS. Finally, a closer mother-child relationship predicted less decrease over time in lexical diversity during conversation, and participants of mothers who graduated college showed a greater increase in conversational talkativeness over time compared to those of mothers with a high school education. CONCLUSIONS: Our results suggest that, during the adolescent period for males with DS or FXS, there is an increase in the amount of talk produced in conversational contexts, but also a decrease in the quality of the language produced. In addition, our results indicate syndrome-specificity for aspects of expressive language development and reinforce the protective role of family-related factors. En ligne : https://dx.doi.org/10.1186/s11689-020-09320-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 [article] Expressive language development in adolescents with Down syndrome and fragile X syndrome: change over time and the role of family-related factors [texte imprimé] / Laura DEL HOYO SORIANO, Auteur ; Angela John THURMAN, Auteur ; Danielle HARVEY, Auteur ; Sara T. KOVER, Auteur ; Leonard ABBEDUTO, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 12 (2020) Mots-clés : Adolescent  Child  Cognition  Down Syndrome/psychology  Family/psychology  Fragile X Syndrome/psychology  Humans  Intellectual Disability/psychology  Language Development  Language Development Disorders/complications  Language Tests  Longitudinal Studies  Male  Mother-Child Relations/psychology  Mothers/psychology  Vocabulary  Adolescence  Conversation  Down syndrome  Expressive language development  Family-related factors  Fragile X syndrome  Longitudinal  Narration Index. décimale : PER Périodiques Résumé : BACKGROUND: It is well known that individuals with Down syndrome (DS) or fragile X syndrome (FXS) demonstrate expressive language difficulties beginning early in childhood. It is less clear, however, whether expressive language skills change during the adolescent period in these individuals, and if any of these changes are syndrome specific. Studying this, as well as the role of maternal and family-related factors in expressive language development, may provide the foundation for efficacious interventions for adolescents with DS or FXS. METHODS: In this study, we examined expressive language trajectories, assessed through conversation and narration, in 57 adolescent males with intellectual disability (ID) (20 DS and 37 FXS) in relation to the diagnostic group (DS vs. FXS) and family-related factors (maternal IQ, maternal psychological distress, closeness in the mother-child relationship, family income, and maternal and paternal education) after adjusting for chronological age (CA) and nonverbal cognition. RESULTS: Changes over repeated annual assessments for males with DS or FXS were observed only during conversation, such as an increase in talkativeness, but a decrease in syntax complexity and lexical diversity. We found a diagnosis-related effect in the change over time in conversational talkativeness favoring those with FXS. Finally, a closer mother-child relationship predicted less decrease over time in lexical diversity during conversation, and participants of mothers who graduated college showed a greater increase in conversational talkativeness over time compared to those of mothers with a high school education. CONCLUSIONS: Our results suggest that, during the adolescent period for males with DS or FXS, there is an increase in the amount of talk produced in conversational contexts, but also a decrease in the quality of the language produced. In addition, our results indicate syndrome-specificity for aspects of expressive language development and reinforce the protective role of family-related factors. En ligne : https://dx.doi.org/10.1186/s11689-020-09320-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573

Genetic and maternal predictors of cognitive and behavioral trajectories in females with fragile X syndrome / Laura DEL HOYO SORIANO in Journal of Neurodevelopmental Disorders, 10-1 (December 2018)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 10-1 (December 2018) . - 22 p. Titre : Genetic and maternal predictors of cognitive and behavioral trajectories in females with fragile X syndrome Type de document : texte imprimé Auteurs : Laura DEL HOYO SORIANO, Auteur ; Angela J. THURMAN, Auteur ; Danielle J. HARVEY, Auteur ; W. Ted BROWN, Auteur ; Leonard ABBEDUTO, Auteur Année de publication : 2018 Article en page(s) : 22 p. Langues : Anglais (eng) Mots-clés : Anxiety  Closeness in the mother-child relationship  Crystallized intelligence  Fmrp  Females with FXS  Fluid intelligence  Longitudinal  Maternal psychological distress  Ratio of affected to total chromosomes  Withdrawal Index. décimale : PER Périodiques Résumé : BACKGROUND: Fragile X syndrome (FXS) is caused by a mutation in the FMR1 gene on the X chromosome, leading to decreased levels of FMR1 protein (FMRP), which causes the array of neuropsychological impairments that define FXS. Because FXS is an X-linked condition, fewer females display FXS and females with FXS are more mildly affected than males, on average. However, there is a considerable variability in terms of severity of affectedness among females with FXS. The current study was designed to investigate potential genetic (FMRP level and ratio of affected to total chromosomes) and environmental factors (maternal psychological distress and closeness in the mother-child relationship) influencing the cognitive (fluid and crystallized intelligence) and behavioral (anxiety and withdrawal) phenotype of females with FXS. METHODS: We conducted a prospective 3-year longitudinal study of 16 females with FXS (with up to four assessments, each separated by a year) using an accelerated longitudinal design so that we had coverage of the age range of 10-15 years at study start and 13-18 at study end. We focused on both the level of functioning related to chronological age expectations (standard scores) and absolute change in skill (raw scores) over the 3-year period. RESULTS: At a cross-sectional level, fluid intelligence and crystallized intelligence were both predicted by a closer mother-child relationship and lower maternal psychological distress. However, only fluid intelligence was predicted by a lower ratio of affected to total chromosomes. Anxiety and withdrawal were predicted by a higher ratio of affected to total chromosomes. Withdrawal was also predicted by lower closeness in the mother-child relationship and higher maternal distress. In terms of longitudinal change, gains were observed in fluid and crystallized intelligence, whereas anxious and withdrawn behaviors remained stable over visits. Gains in fluid intelligence were solely predicted by FXS biomarkers (higher FMRP level and lower ratio of affected to total chromosomes), while gains in crystallized intelligence were not predicted by any of the biological and environmental variables. CONCLUSIONS: Our results show that FXS biomarkers and maternal variables contribute differentially to the cognitive and behavioral features of the adolescent female with FXS. These findings can help in the design of treatment studies aimed at enhancing cognitive and behavioral abilities in the FXS population. En ligne : http://dx.doi.org/10.1186/s11689-018-9240-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=386 [article] Genetic and maternal predictors of cognitive and behavioral trajectories in females with fragile X syndrome [texte imprimé] / Laura DEL HOYO SORIANO, Auteur ; Angela J. THURMAN, Auteur ; Danielle J. HARVEY, Auteur ; W. Ted BROWN, Auteur ; Leonard ABBEDUTO, Auteur . - 2018 . - 22 p. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 10-1 (December 2018) . - 22 p. Mots-clés : Anxiety  Closeness in the mother-child relationship  Crystallized intelligence  Fmrp  Females with FXS  Fluid intelligence  Longitudinal  Maternal psychological distress  Ratio of affected to total chromosomes  Withdrawal Index. décimale : PER Périodiques Résumé : BACKGROUND: Fragile X syndrome (FXS) is caused by a mutation in the FMR1 gene on the X chromosome, leading to decreased levels of FMR1 protein (FMRP), which causes the array of neuropsychological impairments that define FXS. Because FXS is an X-linked condition, fewer females display FXS and females with FXS are more mildly affected than males, on average. However, there is a considerable variability in terms of severity of affectedness among females with FXS. The current study was designed to investigate potential genetic (FMRP level and ratio of affected to total chromosomes) and environmental factors (maternal psychological distress and closeness in the mother-child relationship) influencing the cognitive (fluid and crystallized intelligence) and behavioral (anxiety and withdrawal) phenotype of females with FXS. METHODS: We conducted a prospective 3-year longitudinal study of 16 females with FXS (with up to four assessments, each separated by a year) using an accelerated longitudinal design so that we had coverage of the age range of 10-15 years at study start and 13-18 at study end. We focused on both the level of functioning related to chronological age expectations (standard scores) and absolute change in skill (raw scores) over the 3-year period. RESULTS: At a cross-sectional level, fluid intelligence and crystallized intelligence were both predicted by a closer mother-child relationship and lower maternal psychological distress. However, only fluid intelligence was predicted by a lower ratio of affected to total chromosomes. Anxiety and withdrawal were predicted by a higher ratio of affected to total chromosomes. Withdrawal was also predicted by lower closeness in the mother-child relationship and higher maternal distress. In terms of longitudinal change, gains were observed in fluid and crystallized intelligence, whereas anxious and withdrawn behaviors remained stable over visits. Gains in fluid intelligence were solely predicted by FXS biomarkers (higher FMRP level and lower ratio of affected to total chromosomes), while gains in crystallized intelligence were not predicted by any of the biological and environmental variables. CONCLUSIONS: Our results show that FXS biomarkers and maternal variables contribute differentially to the cognitive and behavioral features of the adolescent female with FXS. These findings can help in the design of treatment studies aimed at enhancing cognitive and behavioral abilities in the FXS population. En ligne : http://dx.doi.org/10.1186/s11689-018-9240-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=386

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