A behavioural assessment of social anxiety and social motivation in fragile X, Cornelia de Lange and Rubinstein-Taybi syndromes

Research output: Contribution to journalArticlepeer-review


  • Hayley Crawford
  • Laura Groves
  • Robyn Dowlen
  • Lisa Nelson
  • Donna Reid

Colleges, School and Institutes

External organisations

  • Coventry University
  • Manchester University Hospitals NHS Foundation Trust
  • Derby Royal Hospital, Derby, United Kingdom.


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 languageEnglish
Pages (from-to)127-144
Number of pages18
JournalJournal of Autism and Developmental Disorders
Issue number1
Publication statusPublished - 21 Sep 2019


  • Adult, De Lange Syndrome/psychology, Down Syndrome/psychology, Female, Fragile X Syndrome/psychology, Humans, Male, Motivation, Phobia, Social, Rubinstein-Taybi Syndrome/psychology, Social Behavior, Young Adult