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


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.

Publication DOI: https://doi.org/10.1007/s10803-016-2736-2
Divisions: College of Health & Life Sciences
College of Health & Life Sciences > Applied Health Research Group
Additional Information: 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.
Uncontrolled Keywords: Working memory Short-term memory Rubinstein–Taybi syndrome Typically developing children Dissociation
Publication ISSN: 1573-3432
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Related URLs: https://link.sp ... 0803-016-2736-2 (Publisher URL)
PURE Output Type: Article
Published Date: 2016-06-01
Published Online Date: 2016-03-24
Accepted Date: 2016-03-23
Authors: Waite, Jane (ORCID Profile 0000-0002-8676-3070)
Beck, Sarah
Heald, Mary
Powis, Laurie
Oliver, Chris



Version: Published Version

License: Creative Commons Attribution

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