The application of attentional control theory for anxiety in Cornelia de Lange syndrome

Laura Groves, Joanna Moss, Hayley Crawford, Rachel Royston, Jane Waite, Andrea Thomas, Kira Moss, Chris Oliver

Research output: Contribution to journalConference abstractpeer-review


Background: DSM‐5 anxiety symptomology and autism‐specific anxiety traits (e.g. Intolerance to Uncertainty [IU]) are prevalent in individuals with Cornelia de Lange syndrome (CdLS). Previous research has implicated executive functioning (EF) impairments in the emergence and maintenance of anxiety, specifically attention shifting and inhibition deficits (attention control theory). However, no research has explored whether this theory underpins anxiety in CdLS. In this study, associations between EF and subthreshold DSM‐5 anxiety and IU were investigated. Method: Caregivers of individuals with CdLS (n = 24) completed direct and informant report measures of EF, subthreshold DSM‐5 anxiety and IU. Individuals with fragile X syndrome (FXS) (n = 28) comparable for level of ability, severity of autism symptomology and age were included as a contrast group.Results: Subthreshold DSM‐5 anxiety was present in both groups (CdLS = 91.8%; FXS = 100.0%) with high rates of co‐morbidity (CdLS = 81.6%; FXS = 88.9%). Autism‐specific anxiety was also prevalent (CdLS = 43.8%, FXS = 35.48% above cut‐off), with specific difficulties for IU identified. Analyses showed presence of social anxiety, specific phobias and IU were associated with greater attention shifting deficits in CdLS (all p < 0.05) but not FXS. Additional analyses revealed that greater scores for IU were associated with presence of generalised anxiety in both groups. Conclusions: Overall, the findings suggest that in CdLS, anxiety is a difficulty for a number of individuals and may be driven by attention shifting difficulties as proposed by attentional control theory. This was not observed for all anxiety types or for anxiety in FXS which may be driven by some other underlying mechanism. However, the association between generalised anxiety and IU is interesting and may suggest that IU acts as a mediating factor between attention shifting deficits and generalised anxiety in CdLS. Understanding the cognitive underpinnings of anxiety in these groups has important implications for clinical practice to maximise the effectiveness of interventions.
Original languageEnglish
Pages (from-to)1081-1082
JournalJournal of Intellectual Disability Research
Issue number9
Early online date13 Aug 2019
Publication statusPublished - Sept 2019
Event The Society for the Study of Behavioural Phenotypes (SSBP) 22nd Educational Day and Research Symposium - Aston University, Birmingham, United Kingdom
Duration: 4 Sept 20196 Sept 2019
Conference number: 22


  • Anxiety
  • Attentional control theory
  • Cornelia de Lange syndrome
  • Executive functioning
  • Fragile X syndrome


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