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Genetic and maternal predictors of cognitive and behavioral trajectories in females with fragile X syndrome / L. DEL HOYO SORIANO in Journal of Neurodevelopmental Disorders, 10-1 (December 2018)
[article]
Titre : Genetic and maternal predictors of cognitive and behavioral trajectories in females with fragile X syndrome Type de document : Texte imprimé et/ou numérique Auteurs : L. DEL HOYO SORIANO, Auteur ; A. J. THURMAN, Auteur ; D. 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
in Journal of Neurodevelopmental Disorders > 10-1 (December 2018) . - 22 p.[article] Genetic and maternal predictors of cognitive and behavioral trajectories in females with fragile X syndrome [Texte imprimé et/ou numérique] / L. DEL HOYO SORIANO, Auteur ; A. J. THURMAN, Auteur ; D. 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 Development and Psychometric Evaluation of the Hansen Research Services Matrix Adaptive Test: A Measure of Nonverbal IQ / J. A. HANSEN in Journal of Autism and Developmental Disorders, 49-7 (July 2019)
[article]
Titre : Development and Psychometric Evaluation of the Hansen Research Services Matrix Adaptive Test: A Measure of Nonverbal IQ Type de document : Texte imprimé et/ou numérique Auteurs : J. A. HANSEN, Auteur Article en page(s) : p.2721-2732 Langues : Anglais (eng) Mots-clés : Computer adaptive test Fluid intelligence Iq Instrument development Item response theory Nonverbal IQ Online phenotyping Psychometrics Raven's Standard Progressive Matrices Index. décimale : PER Périodiques Résumé : Assessment of individuals on the autism spectrum often includes a measure of nonverbal IQ. One such measure is the Raven's Standard Progressive Matrices (RSPM). For large research studies with participants distributed nationally it is desirable for assessments to be available online. Because time is a premium, it is ideal that the measure produces accurate scores quickly. The Hansen Research Services Matrix Adaptive Test (HRS-MAT) addresses these needs and with similar psychometric properties of the RSPM. Scores based on the HRS-MAT correlated at r = .81 with those of the RSPM. In adult-child pairs, HRS-MAT scores correlated at approximately r = .50. Details from respondents in a national sample and psychometric properties including reliability and validity are discussed. En ligne : http://dx.doi.org/10.1007/s10803-016-2932-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=401
in Journal of Autism and Developmental Disorders > 49-7 (July 2019) . - p.2721-2732[article] Development and Psychometric Evaluation of the Hansen Research Services Matrix Adaptive Test: A Measure of Nonverbal IQ [Texte imprimé et/ou numérique] / J. A. HANSEN, Auteur . - p.2721-2732.
Langues : Anglais (eng)
in Journal of Autism and Developmental Disorders > 49-7 (July 2019) . - p.2721-2732
Mots-clés : Computer adaptive test Fluid intelligence Iq Instrument development Item response theory Nonverbal IQ Online phenotyping Psychometrics Raven's Standard Progressive Matrices Index. décimale : PER Périodiques Résumé : Assessment of individuals on the autism spectrum often includes a measure of nonverbal IQ. One such measure is the Raven's Standard Progressive Matrices (RSPM). For large research studies with participants distributed nationally it is desirable for assessments to be available online. Because time is a premium, it is ideal that the measure produces accurate scores quickly. The Hansen Research Services Matrix Adaptive Test (HRS-MAT) addresses these needs and with similar psychometric properties of the RSPM. Scores based on the HRS-MAT correlated at r = .81 with those of the RSPM. In adult-child pairs, HRS-MAT scores correlated at approximately r = .50. Details from respondents in a national sample and psychometric properties including reliability and validity are discussed. En ligne : http://dx.doi.org/10.1007/s10803-016-2932-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=401