Dissociation of Cross-Sectional Trajectories for Verbal and Visuo Spatial Working Memory Development in Rubinstein Taybi syndrome.

Jane Waite, Sarah Beck, Mary Heald, Laurie Powis, Chris Oliver

Research output: Contribution to journalArticle

Abstract

Background. Impairments in working memory (WM) might amplify behavioural difference in genetic syndromes associated with intellectual disability (ID) and account for variability in behavioural phenotypes. Murine models of the genetic disorder Rubinstein-Taybi syndrome (RTS) evidence memory impairments but there is limited research on memory in this syndrome. We examine the cross-sectional trajectory of domains of WM development in RTS.
Methods. Individuals with RTS (N = 32) and typically developing (TD) children (N = 89) completed a battery of WM tasks. Participants with RTS also completed an IQ assessment and parent/carers completed the Vineland Adaptive Behavior Scales (VABS). A crosssectional trajectory analysis was conducted.
Results. The RTS group showed significant WM deficits relative to mental age on measures of WM span in both verbal and visuo-spatial domains. However, whilst better performance on verbal WM span tasks was positively correlated with higher mental age in RTS, this association was not observed on the visuo-spatial span task despite being evident in the TD comparison group.
Conclusions. Individuals with RTS are likely to have difficulties with tasks that rely on WM, above and beyond difficulties predicted by overall ability. In addition, there is a dissociation between the cross-sectional trajectories for verbal and visuo-spatial skills in RTS. Interventions and education strategies for individuals with RTS may need to be tailored to reduce or accommodate these difficulties.
LanguageEnglish
Pages2064-2071
JournalJournal of Autism and Developmental Disorders
Volume46
Issue number6
Early online date24 Mar 2016
DOIs
Publication statusPublished - 1 Jun 2016

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Rubinstein-Taybi Syndrome
Short-Term Memory
Spatial Memory
Inborn Genetic Diseases
Aptitude
Psychological Adaptation
Memory Disorders
Intellectual Disability
Caregivers

Bibliographical note

Copyright: The Author(s) 2016.
Open Access - This article is distributed under the terms of the
Creative Commons Attribution 4.0 International License (http://crea
tivecommons.org/licenses/by/4.0/), which permits unrestricted use,
distribution, and reproduction in any medium, provided you give
appropriate credit to the original author(s) and the source, provide a
link to the Creative Commons license, and indicate if changes were
made.

Funding - Cerebra and the Rubinstein–Taybi Syndrome Support Group.

Keywords

  • Working memory Short-term memory Rubinstein–Taybi syndrome Typically developing children Dissociation

Cite this

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title = "Dissociation of Cross-Sectional Trajectories for Verbal and Visuo Spatial Working Memory Development in Rubinstein Taybi syndrome.",
abstract = "Background. Impairments in working memory (WM) might amplify behavioural difference in genetic syndromes associated with intellectual disability (ID) and account for variability in behavioural phenotypes. Murine models of the genetic disorder Rubinstein-Taybi syndrome (RTS) evidence memory impairments but there is limited research on memory in this syndrome. We examine the cross-sectional trajectory of domains of WM development in RTS.Methods. Individuals with RTS (N = 32) and typically developing (TD) children (N = 89) completed a battery of WM tasks. Participants with RTS also completed an IQ assessment and parent/carers completed the Vineland Adaptive Behavior Scales (VABS). A crosssectional trajectory analysis was conducted.Results. The RTS group showed significant WM deficits relative to mental age on measures of WM span in both verbal and visuo-spatial domains. However, whilst better performance on verbal WM span tasks was positively correlated with higher mental age in RTS, this association was not observed on the visuo-spatial span task despite being evident in the TD comparison group.Conclusions. Individuals with RTS are likely to have difficulties with tasks that rely on WM, above and beyond difficulties predicted by overall ability. In addition, there is a dissociation between the cross-sectional trajectories for verbal and visuo-spatial skills in RTS. Interventions and education strategies for individuals with RTS may need to be tailored to reduce or accommodate these difficulties.",
keywords = "Working memory Short-term memory Rubinstein–Taybi syndrome Typically developing children Dissociation",
author = "Jane Waite and Sarah Beck and Mary Heald and Laurie Powis and Chris Oliver",
note = "Copyright: The Author(s) 2016. Open Access - This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://crea tivecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. Funding - Cerebra and the Rubinstein–Taybi Syndrome Support Group.",
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Dissociation of Cross-Sectional Trajectories for Verbal and Visuo Spatial Working Memory Development in Rubinstein Taybi syndrome. / Waite, Jane; Beck, Sarah; Heald, Mary; Powis, Laurie; Oliver, Chris .

In: Journal of Autism and Developmental Disorders, Vol. 46, No. 6, 01.06.2016, p. 2064-2071.

Research output: Contribution to journalArticle

TY - JOUR

T1 - Dissociation of Cross-Sectional Trajectories for Verbal and Visuo Spatial Working Memory Development in Rubinstein Taybi syndrome.

AU - Waite, Jane

AU - Beck, Sarah

AU - Heald, Mary

AU - Powis, Laurie

AU - Oliver, Chris

N1 - Copyright: The Author(s) 2016. Open Access - This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://crea tivecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. Funding - Cerebra and the Rubinstein–Taybi Syndrome Support Group.

PY - 2016/6/1

Y1 - 2016/6/1

N2 - Background. Impairments in working memory (WM) might amplify behavioural difference in genetic syndromes associated with intellectual disability (ID) and account for variability in behavioural phenotypes. Murine models of the genetic disorder Rubinstein-Taybi syndrome (RTS) evidence memory impairments but there is limited research on memory in this syndrome. We examine the cross-sectional trajectory of domains of WM development in RTS.Methods. Individuals with RTS (N = 32) and typically developing (TD) children (N = 89) completed a battery of WM tasks. Participants with RTS also completed an IQ assessment and parent/carers completed the Vineland Adaptive Behavior Scales (VABS). A crosssectional trajectory analysis was conducted.Results. The RTS group showed significant WM deficits relative to mental age on measures of WM span in both verbal and visuo-spatial domains. However, whilst better performance on verbal WM span tasks was positively correlated with higher mental age in RTS, this association was not observed on the visuo-spatial span task despite being evident in the TD comparison group.Conclusions. Individuals with RTS are likely to have difficulties with tasks that rely on WM, above and beyond difficulties predicted by overall ability. In addition, there is a dissociation between the cross-sectional trajectories for verbal and visuo-spatial skills in RTS. Interventions and education strategies for individuals with RTS may need to be tailored to reduce or accommodate these difficulties.

AB - Background. Impairments in working memory (WM) might amplify behavioural difference in genetic syndromes associated with intellectual disability (ID) and account for variability in behavioural phenotypes. Murine models of the genetic disorder Rubinstein-Taybi syndrome (RTS) evidence memory impairments but there is limited research on memory in this syndrome. We examine the cross-sectional trajectory of domains of WM development in RTS.Methods. Individuals with RTS (N = 32) and typically developing (TD) children (N = 89) completed a battery of WM tasks. Participants with RTS also completed an IQ assessment and parent/carers completed the Vineland Adaptive Behavior Scales (VABS). A crosssectional trajectory analysis was conducted.Results. The RTS group showed significant WM deficits relative to mental age on measures of WM span in both verbal and visuo-spatial domains. However, whilst better performance on verbal WM span tasks was positively correlated with higher mental age in RTS, this association was not observed on the visuo-spatial span task despite being evident in the TD comparison group.Conclusions. Individuals with RTS are likely to have difficulties with tasks that rely on WM, above and beyond difficulties predicted by overall ability. In addition, there is a dissociation between the cross-sectional trajectories for verbal and visuo-spatial skills in RTS. Interventions and education strategies for individuals with RTS may need to be tailored to reduce or accommodate these difficulties.

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JO - Journal of Autism and Developmental Disorders

T2 - Journal of Autism and Developmental Disorders

JF - Journal of Autism and Developmental Disorders

SN - 0162-3257

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