Contrasting Relationships Between Anxiety and Intolerance of Uncertainty in Cornelia de Lange and Fragile X Syndromes.
Kayla Smith, Victoria Perry, Laura Groves and 7 others
PMID 40878447WHAT IT FOUND
Intolerance of uncertainty fully explained the link between autistic traits and anxiety in Cornelia de Lange syndrome, but not in Fragile X syndrome.
Both groups showed similar levels of anxiety and uncertainty, suggesting that uncertainty-focused interventions may help CdLS but not FXS.
Key findings
01Intolerance of uncertainty fully mediated the relationship between autistic characteristics and anxiety in the CdLS group, but not in the FXS group.
02There were no significant differences between the CdLS and FXS groups in levels of autistic characteristics, anxiety symptom severity, or intolerance of uncertainty.
03Autistic characteristics and intolerance of uncertainty significantly predicted anxiety symptom severity, explaining 52% of the variance, while syndrome group did not add predictive value.
STILL TO COME
How it was doneWhat they found
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What it does not show
The study was underpowered to detect small or medium effects, with statistical power of 0.12 for small effects and 0.68 for medium effects. All data were collected via parent or carer report, which may not reflect the individual's internal experience, and self-report was not feasible due to the participants' level of intellectual disability. The sample varied widely in age (children and adults), which may affect how anxiety presents. The ADAMS measure has not been validated specifically for autistic people, and the SRS-2 T-scores may have reduced sensitivity to differences in males with FXS who often score at the upper limit. Females with FXS were excluded, so the findings apply only to males with FXS.
Declared interests
The authors declared no conflicts of interest. The study was funded by the Baily Thomas Charitable Fund, Cerebra, and the Cornelia de Lange Syndrome Foundation UK and Ireland.
The easy way to misread this
Do not assume that interventions targeting intolerance of uncertainty will reduce anxiety in people with Fragile X syndrome. The study found no mediating role for uncertainty in the FXS group, unlike in CdLS.
Summarised by AI from the full paper, without a clinician reviewing it. Check it against the source before it changes what you do. Read it on PubMed →