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.
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.
| Original language | English |
|---|---|
| Pages (from-to) | 2064-2071 |
| Journal | Journal of Autism and Developmental Disorders |
| Volume | 46 |
| Issue number | 6 |
| Early online date | 24 Mar 2016 |
| DOIs | |
| Publication status | Published - 1 Jun 2016 |
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