“Intolerance of uncertainty” mediates the relationship between social profile and anxiety in both Williams syndrome and autism

Abstract

Anxiety is the most significant mental health concern for both Williams syndrome (WS) and autism. Whilst WS and autism are characterized by some syndrome‐specific social differences, less is known about cross‐syndrome profiles of anxiety symptoms. Previous research has shown that Intolerance of Uncertainty (IU) is a core mechanism of anxiety maintenance for clinically anxious populations and for autistic children, adolescents, and adults. The only published study in this area for WS has shown some similar patterns—with an added emphasis on the role of sensory sensitivities—in a sample of older teens and adults (mean age = 24), with the authors highlighting the need for younger samples to consider developmental influences. Here we report a cross‐syndrome, cross‐sectional mediation analyses of children diagnosed with WS or autism, including data from parent surveys of 90 children with WS (n = 48) or autism (n = 42). Group differences showed higher trait levels on all measures for the autism group. Importantly, the relationship between social profile and anxiety was fully mediated by IU level for both groups. This suggests possible similar core mechanisms underlying anxiety in these conditions, and the possibility of generalized intervention approaches especially related to managing distress related to uncertainty in multiple contexts. Lay Summary Autism and Williams Syndrome share some similarities in social profile and also in anxiety traits, but there are also some key differences as well. Comparing them side‐by‐side at the same time improved identification of ways to reduce feelings of anxiety. We found that the intolerance of uncertainty affected the relationship between social profile and anxiety in the same way for young children diagnosed with autism or Williams syndrome, meaning that intervention approaches could be similar for both.

Understanding the prevalence and manifestation of anxiety and other socio-emotional and behavioural difficulties in children with Developmental Language Disorder

“…Intolerance of uncertainty (IU) is a trait characterised by preference for predictability and reacting with emotional distress when presented with the unknown or unfamiliar [23]. It is recognised as a transdiagnostic risk factor in the formulation of a number of anxiety disorders [43][44][45]. Children with DLD could be suggested to have further vulnerability to IU, as their language difficulties render instructions and cues difficult to understand [46].…”

Heterogeneity of Autism Characteristics in Genetic Syndromes: Key Considerations for Assessment and Support

“…Visuo-spatial processing difficulties and auditory hypersensitivity [ 56 , 123 ]. Obsessive preoccupations [ 57 ] and increased anxiety are reported [ 58 , 59 ] • Hypersociability [ 61 ] and social vulnerability [ 62 ] • Auditory hypersensitivity [ 56 ] • Repetitive behaviours [ 57 ] • Intolerance of uncertainty [ 60 •] • Age-related increase in anxiety [ 58 , 59 ] Prader-Willi syndrome (PWS) Loss of the paternally expressed genes on the q11–13 region of chromosome 15, due to deletion or inheritance of two maternal copies [ 73 ] Mild to moderate degree of ID is common . Greater cognitive difficulties are associated with the UPD subtype than the deletion [ 73 ] Hyperphagia can cause excessive eating and food stealing, which requires strict food management [ 73 ].…”

“…People with WS also experience significant anxiety, which increases with age and results in lower social motivation [ 58 , 59 ]. Similarly to autistic individuals and those with CdLS, intolerance of uncertainty mediates the relationship between anxiety and autism characteristics in WS [ 60 •]. However, unlike CdLS, social interactions are not influenced by degree of familiarity with a partner in WS [ 61 ] resulting in increased social vulnerability [ 62 ].…”

An exploratory study on emotion regulation strategy use in individuals with Williams syndrome, autism spectrum disorder and intellectual disability

“…Anxiety is also reported to be one of the most common psychopathologies in individuals with WS ( 29 , 30 ) with higher levels of anxiety reported than in individuals with Down syndrome, Prader Willi-syndrome, and Intellectual Disability, for example ( 30 – 33 ). Some studies also point to higher anxiety levels in autistic individuals compared to individuals with WS ( 26 , 34 ). Interestingly, the type of anxieties appears to be different: while autistic people may have social and non-social anxieties, individuals with WS less often show social anxieties compared to autistic people ( 26 ).…”