Temperament factor structure in fragile X syndrome: the children's behavior questionnaire.
The CBQ works for FXS—just know that high fear/shyness scores are real anxiety markers, not a flaw in the test.
01Research in Context
What this study did
Van Hanegem et al. (2014) asked parents to fill out the Children’s Behavior Questionnaire (CBQ).
The team ran a factor analysis on answers from boys with fragile X syndrome.
They wanted to see if the usual three CBQ factors still showed up in this group.
What they found
The three CBQ factors held together, so the tool is still valid.
Fear and shyness items loaded higher than normal, pointing to a syndrome-specific profile.
In plain words, boys with FXS score extra high on fear and shyness, and that is real, not a testing glitch.
How this fits with other research
Crawford (2023) extends this idea by showing that social anxiety in FXS is driven by sensory hyper-arousal and emotion dys-regulation. The CBQ fear factor is the parent-view mirror of those same mechanisms.
Sullivan et al. (2007) found that parents and teachers could spot anxiety in FXS by watching for arguing, task refusal, and staring. These behaviors line up with the higher fear/shyness scores on the CBQ.
Hall et al. (2006) observed that social escape behaviors jump when tasks demand eye contact or conversation. The CBQ fear factor predicts who will show those escape behaviors under pressure.
Why it matters
You can keep using the CBQ with FXS clients; just expect big fear and shyness numbers. Treat those scores as real anxiety signals, not error. Pair the CBQ with brief direct observation during social tasks to confirm which kids need anxiety supports first.
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02At a glance
03Original abstract
Early patterns of temperament lay the foundation for a variety of developmental constructs such as self-regulation, psychopathology, and resilience. Children with fragile X syndrome (FXS) display unique patterns of temperament compared to age-matched clinical and non-clinical samples, and early patterns of temperament have been associated with later anxiety in this population. Despite these unique patterns in FXS and recent reports of atypical factor structure of temperament questionnaires in Williams Syndrome (Leyfer, John, Woodruff-Borden, & Mervis, 2012), no studies have examined the latent factor structure of temperament scales in FXS to ensure measurement validity in this sample. The present study used confirmatory factor analysis to examine the factor structure of a well-validated parent-reported temperament questionnaire, the Children's Behavior Questionnaire (Rothbart, Ahadi, Hershey, & Fisher, 2001), in a sample of 90 males with FXS ages 3-9 years. Our data produced a similar, but not identical, three-factor model that retained the original CBQ factors of negative affectivity, effortful control, and extraversion/surgency. In particular, our FXS sample demonstrated stronger factor loadings for fear and shyness than previously reported loadings in non-clinical samples, consistent with reports of poor social approach and elevated anxiety in this population. Although the original factor structure of the Children's Behavior Questionnaire is largely retained in children with FXS, differences in factor loading magnitudes may reflect phenotypic characteristics of the syndrome. These findings may inform future developmental and translational research efforts.
Research in developmental disabilities, 2014 · doi:10.1016/j.ijdevneu.2004.05.0