A Behavioural Assessment of Social Anxiety and Social Motivation in Fragile X, Cornelia de Lange and Rubinstein-Taybi Syndromes

Hayley Crawford; Joanna Moss; Laura Groves; Robyn Dowlen; Lisa Nelson; Donna Reid; Chris Oliver

doi:10.1007/s10803-019-04232-5

A Behavioural Assessment of Social Anxiety and Social Motivation in Fragile X, Cornelia de Lange and Rubinstein-Taybi Syndromes

Hayley Crawford
, Joanna Moss
, Laura Groves
, Robyn Dowlen
, Lisa Nelson
, Donna Reid
, Chris Oliver

University of Birmingham

Research output: Contribution to journal › Article › peer-review

34 Citations (Scopus)

214 Downloads (Pure)

Abstract

Unique socio-behavioural phenotypes are reported for individuals with different neurodevelopmental disorders. Here, the effects of adult familiarity and nature of interaction on social anxiety and social motivation were investigated in individuals with fragile X (FXS; n = 20), Cornelia de Lange (CdLS; n = 20) and Rubinstein-Taybi (RTS; n = 20) syndromes, compared to individuals with Down syndrome (DS; n = 20). The Social Anxiety and Motivation Rating Scale was employed whilst participants completed four social tasks, each administered separately by a familiar adult, and also by an unfamiliar adult. Compared to participants with DS, those with FXS and RTS exhibited high levels of social anxiety but similar levels of social motivation. Participants with CdLS showed heightened social anxiety and reduced social motivation only during interactions with an unfamiliar adult when active participation was voluntary.

Original language	English
Pages (from-to)	127-144
Number of pages	18
Journal	Journal of Autism and Developmental Disorders
Volume	50
Issue number	1
Early online date	21 Sept 2019
DOIs	https://doi.org/10.1007/s10803-019-04232-5
Publication status	Published - Jan 2020

Funder

Economic and Social Research council (Grant Number: ES/I901825/1) and by Cerebra

Funding

The authors would like to thank all participants and their families. The authors are indebted to the Fragile X Society, the Cornelia de Lange Foundation UK & Ireland, and the Rubinstein-Taybi Syndrome Support Group for their assistance with recruitment of participants. This study was funded by a grant from the Economic and Social Research council (Grant Number: ES/I901825/1) and by Cerebra.

Funders	Funder number
Cornelia de Lange Syndrome Foundation
Rubinstein-Taybi Syndrome Support Group
Economic and Social Research Council	ES/I901825/1

Keywords

Cornelia de Lange syndrome
Down syndrome
Fragile X syndrome
Rubinstein-Taybi syndrome
Social anxiety
social motivation

ASJC Scopus subject areas

Developmental and Educational Psychology

Access to Document

10.1007/s10803-019-04232-5

Authors Accepted ManuscriptAccepted author manuscript, 405 KBLicence: Unspecified

Cite this

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title = "A Behavioural Assessment of Social Anxiety and Social Motivation in Fragile X, Cornelia de Lange and Rubinstein-Taybi Syndromes",

abstract = "Unique socio-behavioural phenotypes are reported for individuals with different neurodevelopmental disorders. Here, the effects of adult familiarity and nature of interaction on social anxiety and social motivation were investigated in individuals with fragile X (FXS; n = 20), Cornelia de Lange (CdLS; n = 20) and Rubinstein-Taybi (RTS; n = 20) syndromes, compared to individuals with Down syndrome (DS; n = 20). The Social Anxiety and Motivation Rating Scale was employed whilst participants completed four social tasks, each administered separately by a familiar adult, and also by an unfamiliar adult. Compared to participants with DS, those with FXS and RTS exhibited high levels of social anxiety but similar levels of social motivation. Participants with CdLS showed heightened social anxiety and reduced social motivation only during interactions with an unfamiliar adult when active participation was voluntary.",

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AU - Groves, Laura

AU - Dowlen, Robyn

AU - Nelson, Lisa

AU - Reid, Donna

AU - Oliver, Chris

PY - 2020/1

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N2 - Unique socio-behavioural phenotypes are reported for individuals with different neurodevelopmental disorders. Here, the effects of adult familiarity and nature of interaction on social anxiety and social motivation were investigated in individuals with fragile X (FXS; n = 20), Cornelia de Lange (CdLS; n = 20) and Rubinstein-Taybi (RTS; n = 20) syndromes, compared to individuals with Down syndrome (DS; n = 20). The Social Anxiety and Motivation Rating Scale was employed whilst participants completed four social tasks, each administered separately by a familiar adult, and also by an unfamiliar adult. Compared to participants with DS, those with FXS and RTS exhibited high levels of social anxiety but similar levels of social motivation. Participants with CdLS showed heightened social anxiety and reduced social motivation only during interactions with an unfamiliar adult when active participation was voluntary.

AB - Unique socio-behavioural phenotypes are reported for individuals with different neurodevelopmental disorders. Here, the effects of adult familiarity and nature of interaction on social anxiety and social motivation were investigated in individuals with fragile X (FXS; n = 20), Cornelia de Lange (CdLS; n = 20) and Rubinstein-Taybi (RTS; n = 20) syndromes, compared to individuals with Down syndrome (DS; n = 20). The Social Anxiety and Motivation Rating Scale was employed whilst participants completed four social tasks, each administered separately by a familiar adult, and also by an unfamiliar adult. Compared to participants with DS, those with FXS and RTS exhibited high levels of social anxiety but similar levels of social motivation. Participants with CdLS showed heightened social anxiety and reduced social motivation only during interactions with an unfamiliar adult when active participation was voluntary.

KW - Cornelia de Lange syndrome

KW - Down syndrome

KW - Fragile X syndrome

KW - Rubinstein-Taybi syndrome

KW - Social anxiety

KW - social motivation

UR - https://www.scopus.com/pages/publications/85074028751

U2 - 10.1007/s10803-019-04232-5

DO - 10.1007/s10803-019-04232-5

M3 - Article

SN - 0162-3257

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JO - Journal of Autism and Developmental Disorders

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ER -

A Behavioural Assessment of Social Anxiety and Social Motivation in Fragile X, Cornelia de Lange and Rubinstein-Taybi Syndromes

Abstract

Funder

Funding

Keywords

ASJC Scopus subject areas

Access to Document

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Cite this