Anxiety and Phobias in Individuals with Intellectual Disabilities

Moskowitz, Lauren J.; Braconnier, Megan; Jeffay, Melissa

doi:10.1007/978-3-030-20843-1_44

Cited by 4 publications

(4 citation statements)

References 79 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…It is known that individuals with intellectual disability experience anxiety at higher rates than the general population, with an increased risk as the individual ages ( Reid et al, 2011 ; Green et al, 2015 ; Moskowitz et al, 2019 ). However, our study finds that more than one-third of children and 80% of adults with KS either scored over the threshold for an anxiety disorder and/or are being treated with medication for anxiety, which is significantly higher than what is published in general ID.…”

Section: Discussionmentioning

confidence: 99%

Neurobehavioral phenotype of Kabuki syndrome: Anxiety is a common feature

et al. 2022

View full text Add to dashboard Cite

Kabuki syndrome (KS) is a Mendelian Disorder of the Epigenetic Machinery (MDEM) caused by loss of function variants in either of two genes involved in the regulation of histone methylation, KMT2D (34–76%) or KDM6A (9–13%). Previously, representative neurobehavioral deficits of KS were recapitulated in a mouse model, emphasizing the role of KMT2D in brain development, specifically in ongoing hippocampal neurogenesis in the granule cell layer of the dentate gyrus. Interestingly, anxiety, a phenotype that has a known association with decreased hippocampal neurogenesis, has been anecdotally reported in individuals with KS. In this study, anxiety and behavior were assessed in a cohort of 60 individuals with molecularly confirmed KS and 25 unaffected biological siblings, via questionnaires (SCARED/GAS-ID and CBCL/ABCL). Participant age ranged from 4 to 43 years old, with 88.3% of participants having a pathogenic variant in KMT2D, and the rest having variants in KDM6A. In addition, data was collected on adaptive function and positive affect/quality of life in participants with KS using appropriate online surveys including ABAS-III and PROMIS Positive Affect. Survey scores were compared within the KS participants across age groups and between KS participants and their unaffected siblings. We found that children with KS have significantly higher anxiety scores and total behavior problem scores than their unaffected siblings (p = 0.0225, p < 0.0001). Moreover, a large proportion of affected individuals (22.2% of children and 60.0% of adults) surpassed the established threshold for anxiety; this may even be an underestimate given many patients are already treated for anxiety. In this sample, anxiety levels did not correlate with level of cognitive or adaptive function in any KS participants, but negatively correlated with positive affect in children with KS (p = 0.0005). These findings indicate that anxiety is a common neurobehavioral feature of KS. Providers should therefore carefully screen individuals with KS for anxiety as well as other behavioral issues in order to allow for prompt intervention. Neurobehavioral anxiety measures may also prove to be important outcome measures for clinical trials in KS.

show abstract

Section: Discussionmentioning

confidence: 99%

Neurobehavioral phenotype of Kabuki syndrome: Anxiety is a common feature

et al. 2022

View full text Add to dashboard Cite

show abstract

“…While our study findings may offer preliminary support to further explore affective symptoms in those with WSS, application of multiple instruments dedicated to measuring these features among those with cognitive and/or sensorimotor impairment is necessary, as anxiety‐related behaviors may present differently in these individuals. For example, the SCARED may not adequately pick up some behavioral features of anxiety that may be more prevalent among neurodevelopmental disorders, such as ritualistic and compulsive behaviors, perseverative tendencies, and restricted interests (Lidstone et al, 2014; Moskowitz et al, 2019; Oakes et al, 2016; Russell et al, 2019; Uljarević & Evans, 2017).…”

Section: Discussionmentioning

confidence: 99%

“…behaviors may present differently in these individuals. For example, the SCARED may not adequately pick up some behavioral features of anxiety that may be more prevalent among neurodevelopmental disorders, such as ritualistic and compulsive behaviors, perseverative tendencies, and restricted interests (Lidstone et al, 2014;Moskowitz et al, 2019;Oakes et al, 2016;Russell et al, 2019;Uljarevi c & Evans, 2017).…”

Section: Reported History Of Anxiety Disorder Correlates With Parent ...mentioning

confidence: 99%

Anxiety in Wiedemann–Steiner syndrome

Björnsson

Fahrner

et al. 2022

American J of Med Genetics Pt A

View full text Add to dashboard Cite

This study examined anxiety in Wiedemann–Steiner syndrome (WSS). Eighteen caregivers and participants with WSS completed the parent‐ and self‐report versions of the Screen for Child Anxiety Related Disorder or the adapted version of the Screen for Adult Anxiety Related Disorder. Approximately 33.33% of parents and 65% of participants with WSS rated in the clinical range for overall anxiety. Across anxiety subtypes, parents primarily indicated concerns with Separation Anxiety (72%), which was also endorsed by the majority of participants with WSS (82%). The emergent trend showed Total Anxiety increased with age based on parent‐informant ratings. The behavioral phenotype of WSS includes elevated anxiety. Clinical management should include incorporating early behavioral interventions to bolster emotion regulation given the observed risk of anxiety with age.

show abstract

“…While for typically developing children anxiety symptoms relating to specific phobias decrease over time with development (Costello et al, 2011 ), children with Williams syndrome and/or co-occurring intellectual disability are likely to experience worsening anxiety symptoms across development (Maiano et al, 2018 ; Ng-Cordell et al, 2018 ). Cognitive behavioral interventions have been identified as empirically supported approaches to addressing fears and phobias in typically developing children (Hirshfeld-Becker et al, 2010 ; Ollendick and Davis, 2013 ; Whiteside et al, 2020 ) and have also shown benefit for children with ASD (Davis et al, 2007 ; Ollendick et al, 2021 ) and children with intellectual disabilities (Hagopian et al, 2001 ; Moskowitz et al, 2019 ; Dovgan et al, 2020 ; Fodstad et al, 2021 ). Furthermore, evidence is accumulating that these interventions are also useful, with developmentally appropriate incorporation of play activities, with young typically developing children (Oar et al, 2015 ; Kershaw et al, 2017 ; Farrell et al, 2018 ).…”

Section: Introductionmentioning

confidence: 99%

Addressing fears of children with Williams syndrome: therapist and child behavior in the context of a novel play-and humor-infused exposure therapy approach

Young,

Mohanty,

Levine

et al. 2023

Front. Psychol.

View full text Add to dashboard Cite

Many children with Williams syndrome struggle with fears and phobias that significantly impact their daily lives. Yet, there is sparse literature about the impact of behavioral interventions to treat anxiety and phobias among children with Williams syndrome. Using observational coding of intervention videos, the current study examines patterns of the therapist's use of play and humor and relations to child behavioral responses for four children with Williams syndrome who were identified as treatment responders to humor- and play-infused exposure therapy for fears and anxieties. Sessions were coded for therapist behaviors (exposure with or without play/humor, stimulus type used during exposure, passive or invited attention to feared stimulus, and spontaneous parent participation in exposure) as well as positive, negative, and neutral child behaviors (verbalizations and behaviors). Temporal patterns between therapist and child behaviors were analyzed using lag sequential analyses. The results showed that tolerance of feared stimuli improved for two of the four children following this play- and humor-infused exposure therapy approach, and the remaining two participants demonstrated progress beyond tolerating the feared stimulus and showed increased positive behaviors with the feared stimulus across sessions. Findings also showed patterns of therapist attunement to the child's anxiety level demonstrated through efforts to flexibly adjust the degrees of exposure. Therapist-initiated invited attention behaviors, indicative of the therapist's use of narration and priming, were associated with child tolerance and positive behaviors during exposure to the feared stimulus. Limitations of this study include a very small sample size, short duration of intervention, and a single-subject research design, which limit the generalizability of findings. Implications and future directions of this research are discussed.

show abstract

Anxiety and Phobias in Individuals with Intellectual Disabilities

Cited by 4 publications

References 79 publications

Neurobehavioral phenotype of Kabuki syndrome: Anxiety is a common feature

Neurobehavioral phenotype of Kabuki syndrome: Anxiety is a common feature

Anxiety in Wiedemann–Steiner syndrome

Addressing fears of children with Williams syndrome: therapist and child behavior in the context of a novel play-and humor-infused exposure therapy approach

Contact Info

Product

Resources

About