Development and pilot testing of internet-delivered, family-based cognitive behavioral therapy for anxiety and obsessive-compulsive disorders in autistic youth

Abstract

Cognitive behavioral therapy adapted for autistic youth with anxiety and/or OCD has a strong evidence base, but few have access. A 12-week family-based, Internet-delivered cognitive behavioral therapy (iCBT) program for 7–15 year-old autistic youth with anxiety and/or OCD was developed as a potential method to address this problem. Quantitative and qualitative feedback from stakeholders (parents, youth, clinicians) was gathered on an initial draft of content before conducting a pilot trial. This feedback suggested high quality, engagement, usability, and informativeness of the material. Suggestions were incorporated into the treatment program that was tested in a pilot trial. Eight families were randomized to the iCBT program with either 1) weekly email support or 2) weekly email support plus biweekly telehealth check-ins, and seven of these families completed pre- and post-treatment assessments. An average reduction of 39% in anxiety severity was found, with six of the seven being classified as responders. Preliminary evidence suggests that family-based iCBT is an acceptable and promising treatment for autistic youth with anxiety and/or obsessive-compulsive disorders that should be further modified and tested in future work.

Almost half of autistic youth are estimated have an anxiety or obsessive-compulsive disorder (OCD) (van Steensel et al., 2011). Clinically significant anxiety has been linked with increased self-harm behavior, suicidal ideation, family stress, functional impairment, and poorer long-term trajectories in autistic youth (Gotham et al., 2015; Kerns et al., 2015). Thus, it is critical to develop effective and scalable treatments for anxiety in this population.

Cognitive behavioral therapy (CBT) is an efficacious therapy for anxiety and obsessive-compulsive disorders in autistic youth, with a recent meta-analysis of 19 randomized trials showing a large effect favoring CBT relative to control conditions (Sharma et al., 2021). In one recent large, randomized trial, Wood et al. (2020) found that CBT personalized for autistic youth as well as standard CBT were both superior to usual care, with 92% and 81% treatment response rates, respectively. They also found a significant advantage of personalized CBT relative to standard (Wood et al., 2020).

Unfortunately, most youth do not have access to CBT (Salloum et al., 2016). Therapists who specialize in CBT are in high demand, and families often have to withstand months-long waiting lists for treatment. This access gap is even more pronounced for autistic youth, as many practitioners do not feel that they have clinical expertise (Adams & Young, 2021). Coupled with practical barriers such as cost, geography, and other family obligations, it can be especially difficult for families to access CBT.

Internet-delivered CBT (iCBT) has been increasingly studied as a strategy to improve the dissemination of CBT, as this modality automates content through text, videos, and interactive activities. This approach is distinct from CBT delivered via videoconferencing in that iCBT delivers therapeutic content in an automated fashion with minimal professional support; iCBT programs that have been tested in the literature range from being entirely self-guided, to having occasional written communication with a therapeutic “coach,” to having brief telehealth sessions to supplement online material (Grist et al., 2019; Mitchell et al., 2021). Relative to traditional CBT, iCBT has the potential to significantly reduce therapist time and costs without sacrificing efficacy, and thus has been proposed as a model to improve access (Grist et al., 2019; Mitchell et al., 2021). It has strong initial evidence for anxiety and OCD in youth, especially when therapists provide support and parental involvement is emphasized (Aspvall et al., 2021; Grist et al., 2019). Despite the high rates of impairing OCD and anxiety in autistic youth, minimal work has been done to develop or test iCBT for this population specifically. Conaughton et al. (2017) tested a non-adapted internet-based CBT protocol for anxiety among 8 to 12-year-old autistic youth with social anxiety disorder, generalized anxiety disorder, or a specific phobia. In this trial, 39% of child participants no longer met criteria for their primary anxiety disorder diagnosis by the 3-month follow up assessment. They also found large, significant improvements in several measures of global functioning and anxiety severity between baseline and follow-up. While these results are encouraging, they were less robust when benchmarked against non-autistic youth utilizing the same treatment (Conaughton et al., 2017). In another recent example, Wickberg and colleagues (2022) pilot tested an adapted iCBT program for 22 autistic youth with OCD, finding 60% response rate and 50% remission rate at 3-month follow-up. Although these results were slightly less robust when benchmarked against outcomes from 22 youth treated in expert, multidisciplinary treatment (70% responder rate; 61% remission rate), they are quite encouraging when considering the iCBT protocol required just over 3 hours of total therapist time per patient, relative to approximately 24 hours of total clinician time in the comparison sample.

Autistic youth could benefit further from a program that incorporates their needs as a neurodiverse population, as prior work suggests a slight but significant incremental benefit when CBT is adapted for autism (Wood et al., 2020). For example, modifications may include providing social coaching during social anxiety exposures and adapting CBT exercises for sensory issues that may also contribute to avoidance behavior and anxious apprehension. Further, understanding the optimal level of therapist support is an important step in iCBT development, as level of therapist involvement has been shown to impact treatment outcomes (Grist et al., 2019), though minimal work has directly compared iCBT programs for anxious youth with different levels of therapist support.

To address these gaps, this two-phase study aimed to 1) develop a stakeholder-informed, family-based iCBT program for anxiety and OCD adapted specifically for autistic youth, and 2) pilot test the intervention to provide preliminary evidence of feasibility and effectiveness in order to guide further refinement. The pilot test also compared treatment outcomes, therapist time, and family engagement across two levels of therapist support.

2. Method

2.1. Phase 1: Content Development and Stakeholder Feedback

2.1.1. Procedures.

2.1.1.1. Intervention Development

The study team involved in content development for the iCBT program, called the Learning to Understand and Navigate Anxiety (LUNA) Program for Autistic Youth consisted of psychologists with expertise in pediatric anxiety, OCD, and autism; post-doctoral fellows; doctoral students; and post-baccalaureate and undergraduate research assistants. First, key topics of interest (e.g., psychoeducation, exposure) were identified based on a review of existing efficacious CBT protocols of anxiety adapted for autistic youth (Chalfant et al., 2007; Storch et al., 2020; Wood et al., 2020). Obsessive-compulsive disorder was conceptualized as an anxiety disorder in this study given their highly overlapping cognitive-behavioral mechanisms (Abramowitz & Jacoby, 2015). Given the central role of exposure and parental involvement in the treatment of anxiety and obsessive-compulsive disorders in autistic youth (Guzick et al., 2022; Perihan et al., 2020; Storch et al., 2020; Wood et al., 2020), an exposure-focused and family-based approach was developed. All topics had at least 5 iterations, during which each study team member drafted, edited, and reviewed the content. Additional modules were developed to include materials related to working on multiple exposure hierarchies at the same time, adapting exposure hierarchies that involve anxiety symptoms related to autism (e.g., sensory sensitivities, insistence on sameness), social coaching during social exposures, parent behavior management strategies for disruptive behavior, and adapting exposure for anxiety related to the COVID-19 pandemic. Exposure therapy psychoeducation was provided from the framework of exposure and response prevention, with a specific emphasis on both facing feared stimuli and reducing safety behaviors or compulsions. Parent-facing, child-facing (7–11 years-old), and adolescent-facing (12–15 years-old) materials were developed for each module. To make the manual more engaging for youth, a space theme was adopted with related terminology (e.g., “mission plan” for exposure hierarchy). Videos, GIFs, and worksheets were created to diversify the delivery medium of content.

After a thorough review of available software options and firms, final content materials were implemented on Qualtrics (Qualtrics, Provo, UT), a survey platform that is compliant with the Unites States Health Information and Privacy Protection Act (HIPAA). It was chosen due to its security, ability to contain interactive components (e.g., drag-and-drop), user-friendliness, and suitability to data collection. Using Qualtrics allowed therapists to review family responses to interactive components (e.g., point-and-click exercises, text fill activities). Virtual communication was conducted on e-mail separate from the Qualtrics platform.

Several steps were taken to attempt to make materials as universally applicable as possible. These included weekly cartoon videos of two youth struggling with common anxiety and obsessive-compulsive symptom domains (phobias, anxiety related to sensory sensitivities, anxiety related to change in routines, OCD related to contamination, social anxiety, separation anxiety, worries about performance, being on time). Additional examples were provided for a range of fears and anxieties in written text to help families generate personally relevant ideas. Sample content is included in Supplement 2, including screenshots of several features of the program. An introduction video for the first week can be viewed here: https://www.youtube.com/watch?v=JjRFJf0efG4

Therapist training materials were developed after the content was developed. See Table 1 for a summary of LUNA modules

Table 1.

Summary of topics for each Internet-delivered cognitive behavioral therapy weekly module

Module	Topic
1	Orientation to the program, psychoeducation, goal setting
2	Psychoeducation, rewards
3	Coping
4	Exposure hierarchy development
5	Exposure therapy – executing exposures
6	Exposure therapy – challenging cognitions through exposure
7	Exposure therapy – addressing safety behaviors and family accommodation
8–11	Exposure therapy: self-select modules Working on multiple exposure hierarchies simultaneously Social coaching for social anxiety exposures Modifying exposures for anxiety related to sensory sensitivities and insistence on sameness/fear of change Doing exposure during the COVID-19 pandemic Managing oppositional behavior (focus on antecedent management)
12	Review and planning for the future

2.1.1.2. Stakeholder Evaluation

Stakeholders were provided with PDF files of the treatment content (text, video scripts, fillable worksheets/forms) and were asked to review in two weeks. Parents evaluated the parent and youth version that would correspond to their child’s age, youth evaluated their age-appropriate youth version, and clinicians evaluated parent and youth versions selected at random by the study team to ensure an equal number of clinicians would review both youth versions. At the end of each weekly module, stakeholders would find a link to an anonymous, online 5-minute survey that asked about different qualities of content materials. Once stakeholders completed their review, they had a 1-hour virtual semi-structured interview with a study team member to provide their opinions on the content. After the interviews were conducted, the content materials were revised to incorporate the feedback of stakeholders after consultation with the study team. The local institutional review board approved the study.

2.1.2. Participants.

The goal of the first study phase was to develop and evaluate family-based iCBT content using a stakeholder-informed approach. Ten stakeholders were sought for feedback after completing a written draft of materials (see “Content Development” section). Each one of them fell within three categories of participants: 1) parents of autistic children; 2) autistic youth with self/parent-reported significant anxiety, and between the ages of 7 – 15; and 3) licensed mental health clinicians who specialize in autism and/or anxiety. All participants were English-speaking residents of the state where this study was conducted. Recruitment sources included social media, families who previously participated in an anxiety treatment study for autistic children conducted by the study team, and non-profit organizations. Parents and clinicians consented to participated and youth provided verbal assent.

Quantitative feedback:

Each weekly content questionnaire included 5-point Likert scale questions on overall quality, difficulty, informativeness, engagement and length of weekly materials.

Qualitative feedback:

Each weekly content questionnaire had four open-ended questions asking about the repetitiveness of the materials, what stakeholder did or did not like about the materials, areas of improvement and any additional comments stakeholders wanted to provide. During the semi-structured interview, participants were asked to provide their feedback on overall content in terms of its quality, informativeness, engagement, satisfaction, areas of improvement, theme, feasibility, intention to use/recommend treatment to others and their treatment history.

2.1.4. Analytic plan.

Descriptive statistics (response distribution, median, mode) were provided for responses to quantitative items soliciting feedback on each module. Qualitative feedback provided by stakeholders and subsequent changes that were made to the iCBT program were also summarized. This qualitative review was done practically for iterative improvements to the iCBT program but were not analyzed using systematic qualitative methods (e.g., thematic analysis). We have provided a summary of all domains of feedback in Supplemental Material 1.

2.2. Phase 2: Pilot Test of iCBT Program

2.2.1. Procedures.

Recruitment

The goal of the pilot trial was to provide preliminary evidence for the iCBT program, and to receive additional feedback from therapists and families in order to prepare for a larger follow-up trial (currently underway). The local institutional review board approved the trial.

Participants were recruited from a variety of sources throughout the state of Texas, including autism-related community organizations, clinics, non-profits, advocacy groups, and Facebook groups. Recruitment for the pilot trial began in March 2022 and continued through July 2022.

Parents were first pre-screened during a 30-minute phone call to determine likely eligibility prior to scheduling a virtual baseline assessment. Electronic parental consent/child assent was obtained prior to beginning baseline assessments, which included interview-rated assessments and questionnaires. Participants eligible to participate in treatment after the baseline were randomized.

iCBT Treatment

Treatment Conditions

Families were randomized to one of two iCBT conditions with different levels of therapist contact (iCBT-Email, iCBT-Video). In both conditions, parent-child dyads were sent a set of links each week, one for the child and one for the parent, to access the modules that correspond to the week of the program they were on. Families participated for 12 weeks and were encouraged to complete one module per week. Parent and child/teen materials generally aligned, with slight variations in focus, as the parent content was meant to teach parents how they could help their child overcome their anxieties while the child content was meant to teach youth strategies directly. Therapists were either a licensed psychologist (first author), a post-doctoral fellow (sixth author), or a doctoral student supervised and trained by the first author using readings, didactics, modeling, and observation. The trial was registered on ClinicalTrials.gov (Identifier NCT05284435).

In the iCBT-Email condition, links to the treatment modules were sent to families on a weekly basis with therapists checking in once per week, via email. Therapists reviewed participants’ progress on the previous week’s materials prior to emailing the family to provide feedback on how they could continue to improve and implement the program in their everyday lives.

Participants in the iCBT-Video condition also had six bi-weekly 30-minute video calls to provide additional support. Content covered during video calls included: checking changes in functioning since prior visit, answering questions about previous week’s content, checking homework materials, troubleshooting, identifying potential barriers to participation, and providing feedback about CBT exercises.

After the pilot trial, therapists provided feedback to potential barriers families may have experienced in relation to content engagement.

2.2.2. Participants.

Inclusion criteria required that 1) participants were between the ages of 7 and 15 years old; 2) the child had a diagnosis of autism using gold standard assessment procedures (e.g., Autism Diagnostic Observation Schedule-Second Edition; Childhood Autism Rating Scale-Second Edition), as verified by a written report reviewed by licensed psychologists involved with the project; 3) the child had a primary anxiety or obsessive-compulsive disorder diagnosis as determined by the Mini International Neuropsychiatric Interview for Children and Adolescents (MINI-Kid; Sheehan et al., 2010); 4) at least one parent or guardian was able and willing to participate in both assessments and treatment; 5) the child had a Full Scale intellectual quotient of at least 70 (verified by the Kaufman Brief Intelligence Test, Second Edition; (Kaufman, 2004); 5) both the parent and child were able to read and understand English; 6) the child was able to communicate verbally; and 7) participants had to reside in the state of Texas during the trial. Exclusion criteria included: 1) the child had a diagnosis of bipolar, psychotic, or conduct disorder, 2) the child had severe and current suicidal/homicidal ideations, 3) the child was receiving current psychotherapy for anxiety, or had started or changed dosages of psychotropic medications within the last 4 weeks. An initial goal of recruiting 20 parent-child dyads was set to provide initial feasibility and effectiveness data.

2.2.3. Measures.

All clinical assessments were administered to both parent and child before and after treatment began through a virtual platform. Independent evaluators (IE) leading assessments were postdoctoral fellows or advanced doctoral trainees supervised by a licensed psychologist experienced in anxiety and autism.

MINI-KID:

The Mini International Neuropsychiatric Interview for Children and Adolescents (MINI-Kid) (D. V. Sheehan et al., 2010) is a brief, semi-structured, diagnostic assessment based on DSM-5 criteria that reliably produces psychiatric diagnoses for young children and adolescents in both clinical and general populations (Duncan et al., 2018; D. Sheehan et al., 1997).

PARS-A:

The Pediatric Rating Anxiety Scale modified for Autism (PARS-A) (Maddox et al., 2020) is a short, semi-structured, clinician administered measure of anxiety. It begins with a symptom checklist of common anxiety symptoms and subsequently includes five questions that rate anxiety on a 0–5 Likert severity scale across the following domains: frequency, distress, avoidance, impairment at home, and impairment outside home. The PARS-A was modified from the Pediatric Anxiety Rating Scale to assess anxiety more accurately in youth with autism (e.g., assessing anxiety related to insistence on sameness in the checklist). In this study, obsessive-compulsive symptom items developed by the authors assessing were added to the checklist, consistent with this study’s conceptualization of OCD as an anxiety disorder. The measure shows good test-retest reliability (intraclass correlation coefficient = 0.75–0.82) and convergent validity (Maddox et al., 2020).

CGI-I:

The Clinical Global Impression-Improvement Scale is a short, 7-point, clinician-rated measurement used to assess the level of global improvement (Guy, 1976). Following treatment completion, the CGI-I was administered to determine participant’s improvement of clinical anxiety symptoms as well as overall treatment efficacy. The CGI-I has been widely used in psychiatric and psychological clinical trials and has shown strong validity and reliability across raters (Lewin et al., 2012).

Child Sheehan Disability Scale:

The Child Sheehan Disability Scale (CSDS) is a brief, three-item, self- and parent-report measure, adapted from the Sheehan Disability Scale, that was administered to identify levels of anxiety-related impairments and functional impairments in children (D. V. Sheehan et al., 1996; Whiteside, 2009). It has demonstrated strong psychometric properties (Whiteside, 2009).

RCADS:

The Revised Child Anxiety and Depression Scale (RCADS) (Chorpita et al., 2000) is a 47-item self-report instrument that measures clinical symptoms of anxiety and depression in children and adolescents (RCADS-C) with a corresponding parent version (RCADS-P). Both have shown strong psychometric properties (Chorpita et al., 2005). In this study, the anxiety and obsessive-compulsive subscales are reported.

SRS-2:

The Social Responsiveness Scale-Second Edition (SRS-2) (Constantino & Gruber, 2012) is a 65-item, parent-report measure that assesses features of autism. It shows strong psychometric properties and is a valid instrument for determining autism traits in clinical populations (Constantino & Gruber, 2012). For this study, the DSM-5-based Repetitive Behaviors and Restricted Interest (RRB) and Social Communication Impairment (SCI) subscales were used.

CSQ-8:

The Client Satisfaction Questionnaire-8 (Larsen et al., 1979) is a brief, 8-item, frequently used self-report measure that assesses the global satisfaction of clients with the treatment and services they receive. This measure demonstrates high internal consistency and strong psychometric properties (Attkisson & Zwick, 1982; Nguyen et al., 1983). It was delivered as a parent-report measure in this study.

2.2.4. Analytic plan.

The primary outcomes were the PARS-A and CGI-I, with responders being defined as those with CGI-I ratings of “much” or “very much improved.” Paired-samples t-tests were conducted to provide an effect size estimate in the degree of change on primary and secondary outcomes. P values are reported but effect sizes are emphasized in interpretation due to the underpowered nature of this study. CSQ responses were also presented. The following therapist and family utilization data were also examined: therapist self-reported time taken per family per week, number of modules completed per family, parent and child time spent per module. These data provide information on engagement, therapist time, and feasibility.

3. Results

3.1. Stakeholder evaluations

3.1.1. Participants.

Ten stakeholders were surveyed for feedback, including parents of autistic children of all ages (n = 3), autistic youth between the ages of 7 – 15 (n = 3), and licensed mental health clinicians who specialize in autism and/or anxiety (n = 4).

3.1.2. Stakeholder evaluations.

Feedback indicated generally preferable views of the iCBT content. Averaged across all sessions and content recipients (caregivers, children, and teens), responses suggested strong overall quality (median rating = “Good,” modal rating = “Excellent”), difficulty (median and modal rating = “Just Right”), informativeness (median rating = “Informative,” modal rating = “Very informative”), engagement (median and modal rating = “Engaging”), and length (median and modal rating = “Just right”). Distributions of ratings are presented in Figure 1.

Figure 1.

Stakeholder feedback on initial draft of iCBT content

When examining caregiver-facing content specifically, responses revealed an identical pattern, though for the “informativeness” item both median and modal ratings were rated as “very informative.” Child- and teen-facing content also replicated the overall findings described, though for child-facing material, the modal rating for overall quality was “Good,” and the modal response for “informativeness” was tied between “informative” and “very informative.” Teen-facing ratings were similar for overall though, the modal response for “informativeness” was “average.”.

Interviews with stakeholders and free responses indicated a few domains of improvement, including content, language, design/engagement, and usability. A few of the key specific concerns are reviewed here. In terms of content, caregivers emphasized a need for sections on self-compassion and self-celebration as well as instructions on certain therapeutic techniques (e.g., helping children process after practicing exposures). Materials for weeks 4 (exposures), 7 (safety behaviors) and 8 (managing multiple exposure hierarchies) were perceived as particularly challenging. Inclusion of developmentally sensitive examples were requested (e.g., anxious situations common in high schools for teen content, emphasis on social anxiety). In terms of language, stakeholders pointed out the complexity of the text in certain areas. In terms of the space theme, some expressed concern with certain aspects (e.g., unclear terminology [e.g., exposures as “phases”]). Similar to the survey responses, it was recommended to include more videos, graphics, pictures, and colors. In terms of usability, stakeholders suggested the need for recap paragraphs and time estimates of completion at the beginning of each weekly module. These considerations were discussed within weekly treatment development meetings and several modifications were subsequently made before delivering the content to pilot trial participants. For a summary of stakeholders’ feedback, please refer to the supplemental materials.

3.2. Pilot test

3.2.1. Participants

Sixteen participants completed a virtual baseline assessment visit. Of these, six participants were ineligible (low KBIT-2 score [n = 1], not diagnosed with an anxiety disorder [n = 2], no official ASD diagnosis [n = 1], anxiety not the primary diagnosis [n = 2]). Two families were not able to be contacted after the baseline, and eight were enrolled in treatment.

Four of the eight youth identified as male and five identified as White race, with an average age of 10.3 years-old (SD = 3.3 years). The most common diagnoses (primary and secondary) were separation anxiety disorder (n = 5), generalized anxiety disorder (n = 5), and social anxiety disorder (n = 5). No youth had obsessive-compulsive disorder. Seven families completed treatment; one family in the iCBT-Email condition dropped out after the fifth week and preferred not to complete the post-treatment assessment. For a complete summary of demographics see Table 2.

Table 2.

Demographics Table and Baseline Clinical Characteristics

	Full sample n = 8	iCBT-Email n = 4	iCBT-Video n = 4
Primary diagnosis, N (%)
Social Anxiety	1 (13%)	1 (25%)	0 (0%)
Separation Anxiety Disorder	4 (50%)	2 (50%)	2 (50%)
Generalized Anxiety Disorder	3 (38%)	1 (25%)	2 (50%)
Secondary diagnosis, N (%)
Specific Phobia	3 (38%)	2 (50%)	1 (25%)
Generalized Anxiety Disorder	2 (25%)	2 (50%)	0 (0%)
Social Anxiety	4 (50%)	2 (50%)	2 (50%)
Separation Anxiety Disorder	1 (13%)	1 (25%)	0 (0%)
Panic Disorder	2 (25%)	2 (50%)	0 (0%)
Attention-Deficit/Hyperactivity Disorder	3 (38%)	2 (50%)	1 (25%)
Tourette’s Disorder	1 (13%)	1 (25%)	0 (0%)
Major Depressive Disorder (in full remission; past episode)	3 (38%)	1 (25%)	2 (50%)
Age, M (SD)	10.3 (3.3)	10.0 (2.9)	10.5 (4.0)
Child Gender, N (%)
Male	4 (50%)	2 (50%)	2 (50%)
Female	3 (38%)	2 (50%)	1 (25%)
Trans	1 (13%)	0 (0%)	0 (0%)
Race
White	5 (63%)	2 (50%)	3 (75%)
White and Asian	2 (25%)	2 (50%)	0 (0%)
African	1 (13%)	0 (0%)	1 (25%)
Hispanic or Latino Ethnicity	0 (0%)	0 (0%)	0 (0%)
Mother education
Associate’s degree, high school diploma	1 (13%)	1 (25%)	0 (0%)
Some college	0 (0%)	0 (0%)	0 (0%)
Associate’s degree	0 (0%)	0 (0%)	0 (0%)
Bachelor’s degree	4 (50%)	1 (25%)	3 (75%)
Graduate degree	3 (38%)	2 (50%)	1 (25%)
Father education
Associate’s degree, high school diploma,	1 (13%)	1 (25%)	0 (0%)
Some college	2 (25%)	1 (25%)	1 (25%)
Associate’s degree	2 (25%)	0 (0%)	2 (50%)
Bachelor’s degree	1 (13%)	0 (0%)	1 (25%)
Graduate degree	2 (25%)	2 (50%)	0 (0%)

Note: iCBT: Internet-delivered cognitive behavioral therapy; PARS-A: Pediatric Anxiety Rating Scale modified for Autism

3.2.2. Primary outcome: Clinician-rating anxiety and improvement.

Six of the seven completers were classified as responders using the CGI-I criteria of “much” or “very much improved.” Among the three youth in iCBT-Email, two were considered “much improved” on the CGI-I. Among the four youth in iCBT-Video, three were considered “much improved,” and one “very much improved” on the CGI-I. An average 39% reduction was observed on the PARS-A across participants (22% in iCBT-Email; 55% in iCBT-Video). See Figure 2 for a depiction of PARS change for each individual child.

Figure 2.

Change in PARS-A across treatment in iCBT-Email and iCBT-Video participants

Note: iCBT = Internet-delivered cognitive behavioral therapy; PARS-A = Pediatric Anxiety Rating Scale Modified for Autism

Paired-samples t-tests showed an overall large decrease in PARS-A scores (see Table 3).

Table 3.

Change in primary and secondary outcomes across iCBT

	Pre M (SD)	Post M (SD)	d	Percent change	p
PARS-A
Full sample	14.6 (3.4)	8.9 (7.4)	1.08	39.0%	.029
iCBT-Email	16.3 (3.8)	12.7 (10.4)	.522	22.1%	.461
ICBT-Video	13.3 (2.8)	6.0 (3.6)	1.84	54.9%	.035
RCADS Parent-Report: Anxiety
Full sample	39.4 (14.8)	25.6 (18.4)	1.09	35.0%	.028
iCBT-Email	46.3 (14.6)	41.3 (17.6)	.945	10.8%	.243
ICBT-Video	34.3 (14.6)	13.8 (6.1)	1.59	59.8%	.050
RCADS Child-Report: Anxiety
Full sample	40.0 (23.1)	30.0 (22.0)	1.27	25.0%	.015
iCBT-Email	56.7 (17.6)	48.3 (18.5)	1.76	14.8%	.093
ICBT-Video	27.5 (19.2)	16.3 (12.5)	1.10	40.7%	.115
RCADS Parent-Report: OCD
Full sample	4.0 (2.6)	1.4 (2.1)	1.24	65.0%	.017
iCBT-Email	4.7 (3.1)	3.0 (2.6)	2.89	36.2%	.038
ICBT-Video	3.5 (2.5)	.25 (.5)	1.24	92.9%	.090
RCADS Child-Report: OCD
Full sample	4.9 (4.1)	4.4 (3.0)	0.20	10.2%	.62
iCBT-Email	8.7 (3.1)	7.3 (1.2)	0.44	16.1%	.53
ICBT-Video	2.0 (1.6)	2.3 (1.7)	−0.19	−15.0%	.72
SDS-Parent
Full sample	22.9 (11.5)	15.7 (9.4)	0.57	31.4%	.18
iCBT-Email	21.3 (11.0)	18.0 (14.7)	.39	15.5%	.57
ICBT-Video	24.0 (13.4)	14.0 (5.0)	.64	41.7%	.29
SDS-Child
Full sample	11.9 (7.0)	13.1 (7.6)	−.16	−10.1%	.69
iCBT-Email	14.0 (5.2)	12.7 (6.7)	.13	9.3%	.85
ICBT-Video	10.3 (8.5)	13.5 (9.3)	−.50	−31.1%	.39
SRS-2-Social Communication
Full sample	90.9 (22.6)	71.3 (12.1)	−.080	21.6%	.076
iCBT-Email	84.3 (10.3)	81.3 (6.7)	.29	3.6%	.67
ICBT-Video	95.9 (29.6)	63.8 (9.2)	1.29	33.5%	.082
SRS-2-Restricted Interests and Repetitive Behaviors
Full sample	20.6 (4.9)	16.0 (4.9)	−.077	22.3%	.075
iCBT-Email	16.3 (4.0)	15.3 (4.5)	1.00	6.1%	.225
ICBT-Video	23.8 (2.2)	16.5 (5.7)	1.14	30.7%	.106

Note: iCBT = Internet-delivered cognitive behavioral therapy; OCD = obsessive-compulsive disorder; PARS-A = Pediatric Anxiety Rating Scale modified for Autism; SDS = Sheehan

Disability Scale; SRS-2 = Social Responsiveness Scale-Second Edition; RCADS = Revised Children’s Anxiety and Depression Scale

3.2.3. Secondary outcomes: Parent- and child-rated anxiety/obsessive-compulsive symptoms, autism-related impairments, functional impairment, client satisfaction

Large reductions in in the RCADS-Anxiety Parent and Child-report versions were found, as well as in the RCADS-OCD Parent-report subscale. A medium-sized improvement in the CSDS-Parent was also found. Child-rated CSDS and RCADS-OCD showed less change. Minimal change was found in assessments of autism features. See Table 3 for a summary.

Of the seven families who finished the program, parents reported generally high satisfaction with services based on an item-level inspection of responses to the CSQ-8. Please see Table 4 for a summary.

Table 4.

Satisfaction with iCBT program (n = 7)

	No, definitely	No, not really	Yes, generally	Yes, definitely
Did you get the kind of service you wanted?	0	0	2	5
	None of my needs have been met	Only a few of my needs have been met	Most of my needs have been met	Almost all of my needs have been met
To what extent has our program met your needs?	0	0	4	3
	No, definitely not	No, I don’t think so	Yes, I think so	Yes, definitely
If a friend were in need of similar help, would you recommend our program to them?	0	0	2	5
	Quite dissatisfied	Indifferent or mildly dissatisfied	Mostly satisfied	Very satisfied
How satisfied are you with the amount of help you received?	0	0	4	3
	No, they seemed to make things worse	No, they really didn’t help	Yes, they helped somewhat	Yes, they helped a great deal
Have the services you received helped you deal more effectively with your problems?	0	0	2	5
	Quite dissatisfied	Indifferent or mildly dissatisfied	Mostly satisfied	Very satisfied
In an overall, general sense, how satisfied are you with the service you have received?	1	0	1	5
	No, definitely not	No, I don’t think so	Yes, I think so	Yes, definitely
If you were to seek help again, would you come back to our program?	0	0	2	5

3.2.5. Utilization and Therapist Time

Across the 12-week study, parents completed an average of 6.5 modules (SD = 2.7) and youth completed an average of 5.1 (SD = 2.2), though there was a wide range for both (parents: 3–11; youth: 2–9). Among iCBT-email families, parents completed an average of 4.5 modules (SD = 1) and youth completed an average of 3.8 modules (SD = 1.5). Among iCBT-video families, parents completed an average of 8.5 modules (SD = 2.4) and youth completed an average of 6.5 modules (SD = 2.1). Parents took an average of 18.6 minutes to complete each module (SD = 7.8 minutes), children took an average of 19.0 minutes per module (SD = 10.4), and teens took an average of 13.8 minutes per module (SD = 4.4).

In addition to the biweekly 30-minute video calls for participants in the iCBT-Video group, therapists spent an average of 12.4 minutes per week on each family, including time spent reviewing materials and writing emails (SD = 3.4 minutes). The total average time spent per patient per week in the iCBT-Video group was 28.1 minutes (SD = 5.0; inclusive of the additional biweekly 30-minute video call), and the average time spent per patient per week in the iCBT-Email condition was 11.8 minutes (SD = 0.7 minutes).

3.2.5. iCBT Modifications Following Pilot

Following the pilot, several further changes were noted. These included revising the manual to be more consistent with a neurodiversity framework (e.g., closer attention paid to language used to describe autism (Bottema-Beutel et al., 2021); updating characters in the adolescent videos to make them more developmentally appropriate, and increasing clarity and consistency for clinicians, parents, and youth by giving access to subsequent modules only after youth and parents had both completed a previous module.

4. Discussion

This study aimed to develop, revise, and pilot test a family-based, iCBT program for autistic youth with anxiety and obsessive-compulsive disorders. Feedback from parents, youth, and clinicians on an initial draft of the content was incorporated into the next version, though overall feedback suggested the material was relevant, high-quality, developmentally appropriate, and informative. Six of the seven families who completed the iCBT program were classified as much or very much improved at the end of treatment. Large improvements in parent-and child-rated anxiety and parent-rated OCD severity were also found (though it is worth noting no youth in this trial had OCD), as well as medium improvements in parent-rated functional impairment and high parental satisfaction with treatment.

Testing two versions of the protocol (one with email and biweekly 30- minute video calls [iCBT-video], one with email support only [iCBT-email]) provided initial data to support a more formal comparison of these two iCBT formats in follow-up studies. Past iCBT trials have featured a wide range of therapeutic contact; this literature has demonstrated that iCBT programs with relatively more support tend to be more effective (Grist et al., 2019; Johansson & Andersson, 2012). These initial data suggest iCBT for anxiety in autistic youth may follow a similar pattern, as preliminary effects among this small cohort appeared more robust when the iCBT content was complimented with telehealth visits with a therapist. Future work should continue to evaluate whether there are significant differences in iCBT treatment outcomes and engagement based on the degree of therapist support, and whether certain individuals respond positively to a program without substantial therapist involvement, as this option may provide meaningful benefits to a subset of families and could be more easily scaled.

To this end, another interesting (though highly preliminary) pattern in this pilot study was that the two participants with a higher level of initial anxiety experienced less improvement across treatment relative to other participants (though it is also worth noting they were both randomized to iCBT-email). This finding hints that a lower contact CBT approach like iCBT may only be indicated for children and adolescents with less severe anxiety, while youth with more severe anxiety may require a more substantial intervention, as might be expected. This is consistent with a previous trial that showed autistic youth with less severe anxiety disorders responded positively to a parent-led bibliotherapy intervention with minimal therapist support, though youth with more severe anxiety required additional individual therapy to improve (Storch et al., 2022). Future studies with larger samples should continue to evaluate predictors and moderators of iCBT for this population.

Several suggestions were made by the stakeholders to improve the usability of the content, both after sharing initial written drafts of the content and after testing the full iCBT program with youth and their parents. This continuous solicitation of feedback led to several suggestions that were incorporated into iterative versions of the treatment program. A “co-development” approach has been recommended when developing iCBT programs broadly and for childhood anxiety specifically (Hill et al., 2018). The number of changes made in this program based on clinician, parent, and child feedback, both after an initial draft of content and after piloting the treatment program itself, highlights the importance of this recommendation in future work of this nature.

Despite the promising results of this study, it has several limitations, and several challenges were encountered. First, this was a very small pilot test that should be replicated in an adequately powered RCT, which is currently ongoing. None of the participants in this pilot had OCD despite this program being designed with both OCD and anxiety in mind, and thus potency of this exposure-focused program for youth with OCD still needs to be tested (though it is worth noting large improvements in parent-rated OCD symptoms). It is also worth highlighting two significant challenges encountered during the study. First, difficulty identifying a technology partner led to the team “back-engineering” survey software to deliver graphics, text, videos, and forms to families in a weekly fashion. Though useful for an initial test of this program, this is not a sustainable long-term solution that will actualize the potential of iCBT to improve the dissemination and implementation of evidence-based practices, which will require partnerships between clinical/academic specialists and technology experts. Another challenge from a therapist’s perspective was in shifting their view from the typical role from a highly involved stance in which they are the primary provider of services, to a hybrid role in which they rely on the automated content to do much of the work they are used to doing themselves.

5. Conclusion

Developing evidence-based digital therapeutics for anxiety and OCD is a multi-step, iterative process that benefits from involvement of both individuals with lived experience and those with professional expertise. Family-based, Internet-delivered cognitive behavioral therapy for anxiety in autistic youth that was developed with this approach appears to be a promising treatment that should be further tested in follow-up trials. The majority of participants who completed this iCBT program experienced clinically significant benefits, with an average reduction of 39% in clinician-rated anxiety. This was achieved while using less therapist time than what is typical in clinical practice (12 minutes per week in iCBT-Email, 28 minutes per week in iCBT-Video), highlighting the potential of this delivery format to reduce costs while maintaining therapeutic benefits. Randomized trials should follow-up this pilot report to generate more reliable estimates of efficacy, effectiveness, and to identify consistent predictors of treatment response.

Highlights.

• Internet-delivered CBT was developed and revised with multiple rounds of stakeholder feedback

• Two iCBT formats were tested: one with weekly emails plus biweekly video calls; one with weekly emails only

• A 39% reduction in anxiety severity was observed among seven completers (55% in the video call condition; 22% in e-mail)

• Six of seven completers were considered responders

• Therapists spent 12 minutes/week/family in the email condition and 28 minutes/week/family in the video call condition