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Lookup NU author(s): Emerita Professor Jacqueline Rodgers
This work is licensed under a Creative Commons Attribution 4.0 International License (CC BY 4.0).
© 2025 The Author(s). Journal of Intellectual Disability Research published by MENCAP and John Wiley & Sons Ltd.Background: Cornelia de Lange syndrome (CdLS) and fragile X syndrome (FXS) are associated with co-occurring autism and anxiety. In autistic people, intolerance of uncertainty (IU) mediates the relationship between autistic characteristics and anxiety, but it is not known whether this relationship is evident in these genetic syndromes. Understanding the relationship between autism, anxiety and IU is essential to informing the theoretical frameworks of anxiety in rare genetic syndromes and improving clinical interventions. Method: Sixty participants with CdLS or FXS participated in a questionnaire-based study to examine the association between autistic characteristics, anxiety and IU. Results: IU mediated the association between autism and anxiety in participants with CdLS but not in participants with FXS. Conclusions: These results suggest that other factors may contribute to the autism-anxiety relationship in FXS, and highlight the merit of syndrome-specific approaches to the study of anxiety. Recommendations are made for intervention-based research to ameliorate anxiety in CdLS.
Author(s): Smith K, Perry V, Groves L, Moss J, Oliver C, Knight E, Patterson T, Rodgers J, Waite J, Crawford H
Publication type: Article
Publication status: Published
Journal: Journal of Intellectual Disability Research
Year: 2025
Pages: epub ahead of print
Online publication date: 29/08/2025
Acceptance date: 24/07/2025
Date deposited: 08/09/2025
ISSN (print): 0964-2633
ISSN (electronic): 1365-2788
Publisher: John Wiley and Sons Inc
URL: https://doi.org/10.1111/jir.70024
DOI: 10.1111/jir.70024
Data Access Statement: The data that support the findings of this study are available from the corresponding author upon reasonable request. The data are not publicly available because of privacy or ethical restrictions
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