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. Author manuscript; available in PMC: 2025 Feb 22.
Published in final edited form as: J Autism Dev Disord. 2023 Feb 27;54(5):1849–1856. doi: 10.1007/s10803-023-05912-z

Examining the Relationship Between Anxiety Severity and Autism-Related Challenges During Cognitive Behavioral Therapy for Children with Autism

Madeleine N Fuselier 1, Andrew G Guzick 1, Jafar Bakhshaie 2, Jeffrey J Wood 3,4, Philip C Kendall 5, Connor M Kerns 6, Brent J Small 7, Wayne K Goodman 1, Eric A Storch 1
PMCID: PMC11846066  NIHMSID: NIHMS2053894  PMID: 36847894

Abstract

Purpose:

Using data from a randomized clinical trial evaluating cognitive behavioral therapy (CBT) for children with autism and co-occurring anxiety, this study examined the relationship between autism features and anxiety symptoms throughout CBT.

Methods:

Two multilevel mediation analyses were run which examined the mediating role of changes in anxiety for changes in two core features of autism, (a) repetitive and restrictive behaviors (RRBs) and (b) social communication/interaction impairments, between pre- and post-treatment.

Results:

Indirect effects between time and autism characteristics were significant for both models, indicating that as anxiety changes, so do RRBs and social communication/interaction as the outcomes respectively.

Conclusion:

Findings suggest a bidirectional relationship between anxiety and autism features. Implications of these findings are discussed.

Keywords: Autism spectrum disorder, Anxiety, Treatment, Cognitive behavioral therapy, Obsessive-compulsive disorder, Assessment

Introduction

Autism is characterized by social communication difficulties as well as restricted and repetitive behaviors (RRBs). Children with autism have higher levels of anxiety than both typically developing children (van Steensel & Heeman 2017) and children with non-autism developmental delays (Gotham et al., 2015). As many as 41% of autistic children suffer from a co-occurring anxiety disorder (Simonoff et al., 2008; Kerns et al., 2020), and as many as 84% experience impairing subclinical anxiety symptoms (White et al., 2009). Distinct presentations of anxiety that do not fit neatly into existing diagnostic categories are also common in youth with autism (Kerns et al., 2014, 2021). Co-occurring anxiety in children with autism is associated with functional impairment above and beyond the presence of autism alone (MacNeil et al., 2009) and predicts decreased quality of life (Adams et al., 2019), suicidal ideation (Bal et al., 2021), and self-injury (Kerns et al., 2015a).

Although the heightened prevalence and adverse impact of anxiety in individuals with autism is well established, less is known about the specific mechanisms at play in the relationship between autism and anxiety. However, a variety of noteworthy longitudinal associations have been found between autism features and anxiety that suggest a strong connection between these constructs. In their longitudinal study spanning from toddlerhood to adolescence, Ben-Itzchak et al. (2020) found that repetitive and restrictive behaviors (RRBs) demonstrated by toddlers with autism predicted the severity and types of anxiety symptoms observed once these individuals reached adolescence. Specifically, more severe RRBs in toddlerhood predicted elevated separation anxiety in adolescence, while elevated social anxiety in adolescence correlated with lower RRBs in adolescence. In a cross-sectional study, Briot et al. (2020) found that social anxiety predicted increased impairments in social reciprocity but not repetitive behaviors, concluding that acute differences in social motivation and social communication were indicative of co-occurring social anxiety and autism rather than autism alone. In another longitudinal study of children with autism, Pickard et al. (2017) observed that early social and communication difficulties predicted later social anxiety. Factor et al. (2022), examining the effects of a 16-week social skills intervention for individuals with autism, observed decreases in both social anxiety and social impairment, finding that greater improvements in social impairment were associated with more change in social anxiety. In another longitudinal study, Baribeau et al. found that the trajectories of insistence on sameness (a common feature of autism) and anxiety symptoms tend to be similar in severity and direction, suggesting these features may be tightly linked rather than distinct, independent features (Baribeau et al., 2021). RRBs were also found to be an early predictor of later anxiety in the same cohort (Baribeau et al., 2020). Conversely, Duvekot et al. (2018) found no significant paths from early RRBs to later anxiety (or vice versa) in their longitudinal study but did find that anxiety symptoms predicted social communication challenges over time (Duvekot et al., 2018).

Collectively, the findings suggest that the relationships between autism features and anxiety may be bidirectional (see also Marker et al., 2013). For example, a child with strong insistence on sameness may experience significant anxiety in anticipation of changes to their typical routine (e.g., traveling to visit family). This child may argue with their parents and successfully avoid this visit. Without the opportunity to practice flexibility, the insistence on sameness may become more pronounced. Similarly, a child who struggles to understand how to navigate complicated social interactions may fear exclusion or bullying; this fear could lead to social isolation and fewer opportunities to develop social communication skills. In other cases, it may be that anxiety leads more directly to social communication challenges or RRBs. For example, many youths engage in stereotyped behaviors to regulate intense emotions. If they experience fear or anxiety, they may be more likely to engage in more frequent and noticeable stereotypies. If anxiety does contribute to more pronounced autism-related challenges, treating anxiety in youth with autism would be expected to result in subsequent improvements in these areas, including those related to social communication as well as RRBs. This pattern appears to be the case, as autism-related challenges have significantly improved following anxiety-focused cognitive behavioral therapy (CBT; Storch et al., 2013; Wood et al., 2009; Wood et al., 2020). It is unclear, however, whether changes in anxiety were associated with changes in autism features in these studies. The observational findings outlined above suggest strong connections between anxiety and autism, but little research has examined the relationship between the two with an interventional design. A recent intervention study reported a significant association between improvements in social impairment and social anxiety following intervention (Factor et al., 2022), though this study evaluated a social skills intervention (rather than an anxiety-focused treatment) and did not use a mediational analytic approach. While applied behavior analysis (ABA) was more commonly employed as a treatment modality to reduce autism-related RRBs in the past, growing acceptance of CBT as a feasible treatment option for autistic individuals has led to the increased use of CBT for addressing autism-related challenges (Wood et al., 2021). The present study examined the relationship between change in anxiety symptoms and change in core autism features in a cohort of youths with co-occurring anxiety and autism receiving CBT for anxiety. We hypothesized that reduction in anxiety would mediate reduction in social communication impairments and RRBs.

Methods

The current study is a secondary analysis of data from a three-site randomized clinical trial (Wood et al., 2020). The protocol of the clinical trial was approved by the institutional review boards of each of the three data collection sites: University of California Los Angeles, University of South Florida, and Temple University. See Kerns et al. 2016 for a full description of study procedures.

Participants

Participants included 167 children between the ages of 7 and 13 (inclusive) with a formal autism diagnosis, maladaptive and interfering anxiety, and a minimum IQ score of 70 points (± SEM). Autism diagnoses were confirmed by the study’s diagnostic research evaluation using gold standard measures (including the Autism Diagnostic Observation Schedule – Second Edition and the Child Autism Rating Scale-Second Edition), and the presence of maladaptive and interfering anxiety was determined by a Pediatric Anxiety Rating Scale (PARS; Research Units on Pediatric Psychopharmacology Anxiety Study Group, 2002) score of at least 14 points (Ginsburg et al., 2011; Kerns et al., 2015). Participants with comorbid depression, tic disorders, and disruptive behavior disorders were eligible as long as anxiety symptoms were primary (after autism-related features). Participants taking psychotropic medications were not excluded from the study as long as the dose was stable and both the child’s family and prescriber confirmed that there were no plans to alter the dose or change medications. Participants had a mean (SD) age of 9.9 (1.8) years. See Table 1 for demographic information [Table 1].

Table 1.

Demographic Information by Treatment Group (Wood et al., 2020)

Characteristics Participants,No./TotalNo.(%)
Adapted CBT Group Standard CBT Group Treatment as Usual Group
Female 21/75 (28) 13/72 (18) 0/19 (0)
Latino or Latina 12/63 (19) 15/54 (28) 3/19 (16)
Race
 African American/African 7/75 (9) 2/71 (3) 3/19 (16)
 Asian/Pacific Islander 6/75 (8) 3/71 (4) 1/19 (5)
 White 46/75 (61) 48/71 (68) 11/19 (58)
 Native American or Alaskan Multiracial 2/75 (3) 1/71(1) 0/19 (0)
  African American and white 0/75 (0) 2/71 (3) 1/19 (5)
  Asian and white 1/75 (1) 0/71 (0) 0/19 (0)
  Unspecified 1/75 (1) 0/71 (0) 0/19 (0)
Total household income <$40,000 15/72 (21) 13/71 (18) 5/19 (26)
Father’s education
 ≤High school diploma 14/72 (19) 12/69 (17) 5/17 (29)
 ≥4-y College degree 40/72 (56) 40/69 (60) 9/17 (53)
Mother’s education
 ≤High school diploma 6/74 (8) 5/70 (7) 2/19 (11)
 ≥4-y College degree 47/74 (64) 50/70 (71) 13/19 (68)
Parents currently married 58/75(77) 52/71 (73) 12/19 (63)
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Measures

The following measures were administered at pre-, mid-, and post-treatment.

Pediatric Anxiety Rating Scale (PARS)

The PARS (Research Units on Pediatric Psychopharmacology Anxiety Study Group, 2002) is a clinician-rated scale assessing anxiety symptoms and associated severity and impairment over the past week. The PARS has demonstrated good reliability and validity (Caporino et al., 2013), and is psychometrically sound and treatment sensitive in samples of children with autism (Storch et al., 2012), including demonstrating treatment sensitivity (Wood et al., 2020; Storch et al., 2015). The PARS Severity Scale item scores range from 0 to 5, with higher scores representing more severe anxiety. A mean of the scores on the 7 items was calculated. Internal consistency was α = 0.61.

Social Responsiveness Scale-Parent Version (SRS-P)

The Social Responsiveness Scale parent version (SRS-P; Constantino & Gruber 2005) assesses the severity of various features associated with autism. The SRS-P has been extensively validated in populations aged 4 to 18 (Chun et al., 2021). Two SRS-P subscales were examined: one focused on repetitive and restrictive behaviors (the “Restricted Interests and Repetitive Behavior” subscale of the SRS; SRS-RRB; α = 0.77) and one focused on social communication/interaction (the “Social Communication” subscale of the SRS; SRS-SCI; α = 0.90).

Procedures

Participants were randomized with a 4.5:4.5:1 ratio to receive either (1) standard-of practice CBT (Coping cat), (2) autism-adapted CBT (Behavioral Interventions for Anxiety in Children with Autism [BIACA]), or (3) treatment as usual (TAU). Study therapists included 19 doctoral students in psychology and postdoctoral fellows who were assigned to participants based on availability. The standard-of-practice (Coping Cat) arm included 16 weekly 60-minute sessions. The main features are of Coping Cat are (1) recognizing anxious feelings and somatic reactions to anxiety, (2) identifying cognition in anxiety-provoking situations (e.g., expectations of threat), (3) developing a plan to cope (e.g., reappraisal), (4) imaginal and in vivo exposure tasks, and (5) self-reinforcement for effort. The treatment uses modeling, role-play, and contingent reinforcement. Specific homework tasks are assigned. Parent involvement in the child’s treatment includes a regular 15-minute check-in at the start of each session and 2 meetings with the therapist. The autism-adapted CBT (BIACA) arm included 16 weekly 90-minute sessions that were split evenly between children and parents. Beyond longer sessions and a higher degree of parent involvement, BIACA differs from the standard-of-practice CBT arm in the following ways: (1) BIACA uses a modular format guided by an algorithm to personalize treatment, given the multifaceted clinical presentations in ASD; (2) children’s disruptive behavior is addressed as needed with antecedent and incentive-based practices to reduce the influence of aggression and noncompliance on treatment engagement; (3) children are taught social engagement skills as needed (e.g., playdate hosting, joining peers at play) to facilitate successful peer-oriented exposure-therapy assignments; (4) the children’s focused interests are treated as an asset and incorporated into treatment to promote engagement; (5) target behaviors are reinforced with a comprehensive reward system at home and, when relevant, in school to promote motivation and treatment engagement.

The TAU arm included a list of community referrals and a continuation of usual services for four months. Families were provided referrals for therapy, but they were not given any specific treatment recommendations. Treatment fidelity was monitored through audio recordings and analyzed by the principal investigators and trained research assistants. 92 BIACA and 70 Coping Cat sessions were randomly selected for fidelity coding. Coders noted the presence or absence of required topics for each session. There was adherence to 97% and 96% of the required topics in BIACA and Coping Cat sessions, respectively. A second coder rated 14.3% of the coded tapes to assess interrater reliability. Intraclass correlations were 0.85 and 1.00 for BIACA and Coping Cat, respectively. See Kerns et al. 2016 for additional information about treatment conditions.

Data Analyses

In SPSS, version 23, data were analyzed using multilevel linear modeling (MLM) using linear mixed-effects models (MIXED). The constrained maximum-likelihood estimation was used to estimate unknown parameters in the models.1 The models were divided into two levels, with repeated evaluations over time (Level 1; variables included time and anxiety symptoms) nested inside persons (Level 2). The multilevel modeling approach is one of the fundamental strategies for coping with missing data since it allows the number of observations to vary across participants (Raudenbush, 2001). We contrasted a model with a linear time variable with a model with a quadratic time variable using Akaike’s information criteria (Akaike, 1987) and the deviance statistic before undertaking mediation studies. According to the results, the linear model fits better across all study variables. A time-structured predictor was incorporated to account for the evaluation of timing irregularities. As a result, the time values matched the actual time gaps (in weeks) between each follow-up evaluation (Singer et al., 2003). From pre- to post-treatment, mediating analyses (Kenny et al., 2003) were used to assess the link between anxiety and autism features (three time-points in Fig. 1 [Fig. 1], i.e., pre-, mid-, and post-treatment). The first looked at the function of anxiety symptoms in mediating changes in repetitive and restricted behaviors from visit 1 to visit 3, with time as the predictor, anxiety symptoms as the mediator, and repetitive and restrictive behaviors as the outcome. The second model investigated anxiety symptoms’ function in mediating social communication/interaction changes, with time serving as the predictor, anxiety symptoms serving as the mediator, and social communication/interaction serving as the outcome. To assess the confidence intervals (CI) of the indirect effects, we utilized the RMediation program (Tofighi & MacKinnon, 2011). The absence of zero in the 95% confidence intervals indicates that an indirect impact is statistically significant (Preacher & Selig, 2012).

Fig. 1.

Fig. 1

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Mediational models demonstrating the relationship between the change in anxiety symptoms and the change in autism-related challenges (i.e., RRBs and social communication/interaction challenges)

Results

For model one with repetitive and restrictive behaviors as the outcome, the A path (anxiety symptoms regressed on time) was significant (b = − 0.73, SE = 0.045, t= −16.01, p < .001) indicating that anxiety symptoms significantly decreased during the treatment. Next, path B (repetitive and restrictive behaviors regressed on anxiety) was significant (b = 0.017, SE = 0.0031, t = 5.39, p < .001) indicating that within individual participants, as anxiety decreased between visit 1 to visit 3, so did repetitive and restricted behaviors. The confidence intervals for the indirect effect was significant (path a*path B), b= −0.012, 95% CI (−0.017 to −0.008), indicative of the hypothesized mediation. Path C (repetitive and restrictive behaviors regressed on time) was significant (b= −4.13, SE = 0.74, t= −5.56, p < .001) indicating that repetitive and restrictive behaviors significantly decreased between visit 1 to visit 3 (see results for the regression analyses of the adjusted model in Table 2 [Table 2]).

Table 2.

Summary of Multilevel Regression Analyses for the Mediational Model 1: Repetitive and Restrictive Behaviors; Anxiety Symptoms as Mediator

Step Path Predictor Outcome b SE t p
1 C Time repetitive and restrictive behaviors −4.133190 0.743056 −5.562 0.000
2 A Time Anxiety symptoms − 0.726941 0.045414 −16.007 0.000
3 B Anxiety symptoms repetitive and restrictive behaviors 0.016601 0.003078 5.394 0.000
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Regarding model two with social communication/interaction as the outcome, the Path B with social communication/interaction regressed on anxiety symptoms was significant (b = 0.011, SE = 0.0025, t = 4.30, p < .001), suggesting that within individual participants, changes in anxiety and social communication/interaction difficulties were significantly positively associated over time (i.e., as anxiety decreased for a participant, so did social communication difficulties). Path A was similar across both models. Next, Path C (social communication/interaction regressed on time) was significant (b= −4.87, SE = 0.91, t= −5.33, p < .001) indicating that social communication/interaction significantly decreased between visit 1 to visit 3. The confidence intervals for the indirect effect (path a*path b) was significant, 0.0078, 95% CI [−0.012 to −0.004], providing evidence for mediation (i.e., that anxiety mediated the relationship between time in treatment and fewer social communication/interaction deficits). See results for the regression analyses of the adjusted model in Table 3 [Table 3].2

Table 3.

Summary of Multilevel Regression Analyses for the Mediational Model 2: Social Communication/Interaction; Anxiety Symptoms as Mediator

Step Path Predictor Outcome b SE t p
1 C Time social communication/interaction −4.870765 0.914735 −5.325 0.000
2 A Time Anxiety symptoms − 0.726941 0.045414 −16.007 0.000
3 B Anxiety symptoms social communication/interaction 0.010750 0.002503 4.295 0.000
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Discussion

The present analyses examined the relationship between the change in anxiety symptoms and change in social communication impairments and RRBs in youth with autism participating in CBT for anxiety. Our results indicate that the amelioration of anxiety symptoms mediated the reduction in autism-related challenges, including both social communication deficits and RRBs. This is noteworthy, given that CBT directly targeted anxiety and not autism-related challenges in this trial. This finding suggests that addressing anxiety in youth with autism could directly improve challenges associated with core autism features, and thus that some challenges traditionally associated with autism might be brought about by co-occurring anxiety, not solely by autism itself. Overall, our results suggest that anxiety should not be overlooked in the treatment of autism-related challenges.

The present results could be explained by the negative effects of anxiety on social communication (Sukhodolsky et al., 2008) and by the roll RRBs may play in reducing anxiety-induced distress in individuals with autism. As the understanding of autism increases, it might be determined that RRBs can be functionally understood as a susceptibility to anxiety in particular situations (e.g., when experiencing changes in routine, when experiencing certain sensory stimuli) coupled with a propensity to alleviate this anxiety in particular ways (e.g., engaging in repetitive behaviors, maintaining routines). Similarly, anxiety and social interaction/communication may be intertwined in this population; addressing feared expectations about negative evaluation or social confusion (with social skills supports, as was provided in the autism-adapted CBT condition), can lead to improvements not only in anxiety but also in social communication difficulties. CBT features present in both treatment conditions appear to be contributing to the reported benefits, and it seems likely that the observed improvements in social communication/interaction challenges and RRBs were brought about by the reduction of anxiety, as the standard-of practice CBT did not address autism-specific challenges yet improvements these domains were still observed.

Study limitations and possible future directions merit comment. All participants had IQ scores of 70 or greater, so results might not be generalizable to individuals with mild to severe cognitive impairment. Participants were majority male (28% female), which reflects the lower recognition of autism in females (Fombonne, 2009), but warrants consideration in the interpretation of the findings. Limited sociodemographic diversity and the impact of potential informant effects (e.g., a halo effect, as parents provided the information on the PARS intervention and made ratings on the SRS at each timepoint) should also be considered. Also noteworthy is the ongoing discussion as to the potentially limited specificity of the SRS (Cholemkery et al., 2014; Capriola-Hall et al., 2021). Additionally, while informative, the analyses conducted are not causal models and must be interpreted with care, as they are inherently correlational and have multiple possible interpretations. Uncertainty remains as to how reduced anxiety might lead to reduced RRBs and improved social communication. Future research should aim to determine the specific CBT intervention elements that facilitate the observed concurrent improvements in both anxiety and autism-related challenges in this population in order to contribute to the refinement of these interventions.

Acknowledgments

Research reported in this publication was supported by the Eunice Kennedy Shriver National Institute of Child Health & Human Development of the National Institutes of Health under Award Numbers 1R01HD080096, 1R01HD080098, and P50HD103555 for use of the Clinical and Translational Core facilities. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.

Footnotes

Declarations

Dr. Wood discloses the following relationships: Research support from NIH and the Department of Defense. Dr. Kendall receives royalties from the sales of materials related to the treatment of anxiety disorders in youth. Dr. Kerns receives royalties from Elsevier. In addition, she has received honoraria and consulting fees for training other researchers on the Autism Spectrum Addendum. Dr. Storch discloses the following relationships: consultant for Biohaven Pharmaceuticals and Brainsway; Book royalties from Elsevier, Springer, American Psychological Association, Wiley, Oxford, Kingsley, and Guilford; Stock valued at less than $5000 from NView; Research support from NIH, IOCDF, Ream Foundation, and Texas Higher Education Coordinating Board.

1

Constrained maximum-likelihood estimation was used because it yields unbiased and consistent estimators for variance coefficients and is less sensitive to outliers compared to maximum likelihood estimation. However, when the models were run using to maximum likelihood estimation (instead of constrained maximum-likelihood estimation), the results stayed identical.

2

While the authors were interested in a fine-grained examination of the precise subscales analyzed (Social Communication and Restricted Interests and Repetitive Behaviour) rather than the total SRS score (which includes additional Social Awareness, Social Cognition, and Social Motivation subscales), additional analyses indicated that the two subscales examined are highly correlated with the total SRS score (r > .7). The results reported above did not change when the models were run with the total SRS scores.

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