Cost-effectiveness of cognitive-behavioral therapy versus treatment as usual for anxiety disorders in children with autism spectrum disorder
Introduction
Anxiety disorders in children with autism spectrum disorders (ASD) are among the most common comorbid disorders (e.g., Simonoff et al., 2008) and are found to be related to a higher life interference (Farrugia & Hudson, 2006), a lower quality of life (Steensel, Bögels, & Dirksen, 2012), as well as additional societal costs (Steensel, Dirksen, & Bögels, 2013). Primarily, children with ASD suffer from chronic impairments in the social, communicative and repetitive domain (APA, 2000), and these ASD characteristics may be exacerbated by comorbid anxiety disorders (e.g., Wood & Gadow, 2010).
A number of studies have examined the effectiveness of cognitive-behavioral therapy (CBT) for the treatment of anxiety disorders in ASD and all have reported positive results; a substantial percentage of children being free from their primary anxiety disorder after treatment (i.e., 71%; Chalfant, Rapee, & Carroll, 2007), a substantial percentage of children being free from all anxiety disorders at post-treatment (i.e., 53%; Wood et al., 2009), and an overall decrease in anxiety levels (Chalfant et al., 2007, Reaven et al., 2009, Reaven et al., 2012, Sofronoff et al., 2005, Sung et al., 2011, Wood et al., 2009). Several studies have also compared CBT to a waitlist condition (Chalfant et al., 2007, Reaven et al., 2009, Sofronoff et al., 2005, Wood et al., 2009) and all studies have reported CBT to be superior compared to waitlist (no treatment) based on parental report. Thus, there is a growing body of evidence supporting the effectiveness of CBT for the treatment of anxiety disorders in ASD, however, less is known about the cost-effectiveness of CBT in relation to other treatments for these patients.
In cost-effectiveness analyses, the clinical outcomes as well as the costs of an intervention are compared to an alternative intervention in order to determine which treatment is the most cost-effective. An intervention is said to dominate the other when this intervention results in higher gains and lower costs. The incremental cost-effectiveness ratio (ICER) can be calculated which represents the ratio of the net increase in costs to the net increase in effectiveness (Palmer, Byford, & Raftery, 1999). Studies that have compared both costs as well as clinical outcomes (effectiveness) – i.e., conducted a cost-effectiveness analysis – for the treatment of anxiety disorders in children with ASD are lacking. Also, we have identified only three studies that have compared the effectiveness of CBT to other treatment options for anxiety in the ASD-population (Reaven et al., 2012, Storch et al., 2013, Sung et al., 2011) which will be discussed next.
The study of Sung et al. (2011) randomly assigned children with ASD to CBT (n = 36) or a social-recreational program (n = 34) which consisted of a variety of age-appropriate leisure activities. After having followed either CBT or the social-recreational program, children with ASD reported a decrease in anxiety levels, and no evidence was found for one intervention being more effective than the other. The study of Reaven and colleagues (2012) randomly assigned children to either CBT (n = 24) or TAU (n = 26). TAU in the study by Reaven et al. (2012) consisted of (1) medication, (2) social skills training, (3) bully-proofing programs, (4) individual psychosocial interventions, or (5) family focused interventions. The authors found evidence for CBT to outperform TAU; i.e., at post-treatment clinical severity ratings of the anxiety disorders were lower in CBT compared to TAU, and while a significant reduction in the total number of anxiety disorders was found in CBT, this result was not found in TAU. Furthermore, at post-treatment, families in TAU were invited to participate in CBT and 93% of the families agreed (Reaven et al., 2012), suggesting that TAU was not sufficient for treating anxiety disorders. In the study by Storch et al. (2013) 24 children were assigned to CBT and 21 to TAU (which consisted of interventions – psychotherapy, medication management or family treatment/education – targeting anxiety or ASD symptoms. Large group differences in favor of the CBT condition were found with respect to anxiety severity. In addition, it was found that 75% of the children in CBT were treatment responders versus only 14% of the children in TAU.
In sum, there is growing evidence for CBT to be effective for the treatment of anxiety disorders in children with ASD, however, studies comparing the effectiveness of CBT to other interventions are rare. The cost-effectiveness of CBT compared to other treatments for anxiety in children with ASD has not yet been examined. The aim of this study was to investigate the cost-effectiveness of CBT, as compared to TAU, for the treatment of anxiety disorders in children with ASD.
Section snippets
Study design
Families of children referred to outpatient mental health care centers for anxiety and/or ASD-related problems were eligible for this study. Therapists contacted the research assistant if they were seeing a child with ASD and comorbid anxiety. Families were then invited for a pre-assessment in which the ADIS-C/P was administered. When children met ADIS-C/P criteria of at least one anxiety disorder, then families were enrolled in the study. In total, 49 families (49 children, 49 mothers and 32
Pre assessment comparisons
Table 1 displays the sample characteristics of the CBT- and TAU-condition with respect to the baseline assessment. No differences were found for gender, X2 (1) = 0.09; p = 0.763, or age, F (1, 47) = 0.16; p = 0.689. No differences between conditions were found for the total anxiety severity score (ADIS-C/P) based on child-report, F (1, 47) = 0.03; p = 0.861, or parent-report, F (1, 47) = 0.53; p = 0.471. In addition, no differences between CBT and TAU were found with respect to the presentation (type) of
Discussion
This was the first study to compare CBT versus TAU taking into account both effects and costs. Although both effects and costs did not differ significantly between the two conditions, the cost-effectiveness analyses demonstrated that CBT seems a cost-effective intervention to treat anxiety disorders in children with ASD.
With respect to a primary anxiety disorder-free child, the ICER demonstrated that CBT dominates TAU. The bootstrapped ICERs demonstrated that CBT had a high probability to be
Conclusions
CBT seems a cost-effective intervention to treat anxiety disorders in children with ASD when compared to TAU if being free from primary anxiety disorder is used as measure of outcome. Regarding the cost per QALY analysis, the bootstrap analysis and cost-effectiveness acceptability curve show that results are highly uncertain and do not clearly point to one treatment being more cost-effective. It will be important for future studies to use follow-ups over a longer period of time. Also,
Acknowledgement
This study was funded by a grant from the Netherlands Organization for Health Research and Development (ZonMw), project number 170881006.
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