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An experimental study of executive function and social impairment in Cornelia de Lange syndrome
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Nelson, Lisa, Crawford, Hayley, Reid, Donna, Moss, Joanna and Oliver, Chris (2017) An experimental study of executive function and social impairment in Cornelia de Lange syndrome. Journal of Neurodevelopmental Disorders, 9 (33). doi:10.1186/s11689-017-9213-x ISSN 1866-1947.
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Official URL: http://dx.doi.org/10.1186/s11689-017-9213-x
Abstract
Background
Extreme shyness and social anxiety is reported to be characteristic of adolescents and adults with Cornelia de Lange syndrome (CdLS); however, the nature of these characteristics is not well documented. In this study, we develop and apply an experimental assessment of social anxiety in a group of adolescents and adults with CdLS to determine the nature of the social difficulties and whether they are related to impairments in executive functioning.
Methods
A familiar and unfamiliar examiner separately engaged in socially demanding tasks comprising three experimental conditions with a group of individuals with CdLS (n = 25; % male = 44; mean age = 22.16; SD = 8.81) and a comparable group of individuals with Down syndrome (DS; n = 20; % male = 35; mean age = 24.35; SD = 5.97). Behaviours indicative of social anxiety were coded. The Behavior Rating Inventory of Executive Function-Preschool version, an informant measure of executive function, was completed by participants’ caregivers.
Results
Significantly less verbalisation was observed in the CdLS group than the DS group in conditions requiring the initiation of speech. In the CdLS group, impairments in verbalisation were not associated with a greater degree of intellectual disability but were significantly correlated with impairments in both planning and working memory. This association was not evident in the DS group.
Conclusions
Adolescents and adults with CdLS have a specific difficulty with the initiation of speech when social demands are placed upon them. This impairment in verbalisation may be underpinned by specific cognitive deficits, although further research is needed to investigate this fully.
| Item Type: | Journal Article | |||||||||
|---|---|---|---|---|---|---|---|---|---|---|
| Subjects: | H Social Sciences > HV Social pathology. Social and public welfare Q Science > QM Human anatomy R Medicine > RC Internal medicine R Medicine > RD Surgery R Medicine > RG Gynecology and obstetrics R Medicine > RJ Pediatrics |
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| Divisions: | Faculty of Science, Engineering and Medicine > Medicine > Warwick Medical School > Health Sciences Faculty of Science, Engineering and Medicine > Medicine > Warwick Medical School > Health Sciences > Mental Health and Wellbeing Faculty of Science, Engineering and Medicine > Medicine > Warwick Medical School |
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| Library of Congress Subject Headings (LCSH): | De Lange's syndrome , Intellectual disability , Social phobia , People with mental disabilities | |||||||||
| Journal or Publication Title: | Journal of Neurodevelopmental Disorders | |||||||||
| Publisher: | BioMed Central Ltd. | |||||||||
| ISSN: | 1866-1947 | |||||||||
| Official Date: | 11 September 2017 | |||||||||
| Dates: |
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| Volume: | 9 | |||||||||
| Number: | 33 | |||||||||
| DOI: | 10.1186/s11689-017-9213-x | |||||||||
| Status: | Peer Reviewed | |||||||||
| Publication Status: | Published | |||||||||
| Access rights to Published version: | Open Access (Creative Commons) | |||||||||
| Date of first compliant deposit: | 24 January 2020 | |||||||||
| Date of first compliant Open Access: | 24 January 2020 | |||||||||
| RIOXX Funder/Project Grant: |
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