Cognitive Behavior Therapy Tailored to Anxiety Symptoms Improves Pediatric Functional Abdominal Pain Outcomes: A Randomized Clinical Trial

Abstract

Objectives:

To evaluate the feasibility of a stepped care model, and establish the effect of a tailored cognitive behavioral therapy (CBT), the Aim to Decrease Anxiety and Pain Treatment (ADAPT), compared with standard medical treatment as usual on pain-related outcomes and anxiety.

Study design:

Eligible patients between the ages of 9 and 14 years with FAPD (n = 139) received enhanced usual care (EUC) during their medical visit to a gastroenterologist. Those that failed to respond to EUC were randomized to receive either a tailored CBT (ADAPT) plus TAU, or TAU only. ADAPT dose (4 sessions of pain management or 6 sessions of pain and anxiety management) was based on presence of clinically significant anxiety. Outcomes included feasibility, based on recruitment and retention rates. Response to ADAPT+TAU versus TAU on pain-related outcomes and anxiety measures was also investigated using a structural equation modeling (SEM) equivalent of a MANCOVA. Anxiety levels and ADAPT dose as moderators of treatment effects were also explored.

Results:

Based on recruitment and retention rates, stepped care was feasible. EUC was effective for only 8% of youth. Participants randomized to ADAPT+TAU showed significantly greater improvements in pain-related disability, but not pain levels, and greater improvements in anxiety symptoms compared with those randomized to TAU only. Anxiety and ADAPT treatment dose did not moderate the effect of treatment on disability nor pain.

Conclusions:

Tailoring care based on patient need may be optimal for maximizing the use of limited psychotherapeutic resources while enhancing care.

Trial registration

ClinicalTrials.gov: NCT03134950

Pediatric functional abdominal pain disorders (FAPD) are common (1, 2), debilitating (3–5), and can persist for years (6, 7). Youth with FAPD can experience psychological problems (8–11) and social and academic difficulties (4, 5). Anxiety corresponds to higher pain and disability (10, 12, 13) over the long term (14, 15). Early psychological intervention could improve patient outcomes (16); however, it is infrequently offered during standard medical care.

Stepped care a feasible method for receiving psychological treatment during routine care, approaches initially provide less intensive treatments to the majority of patients, which are brief and fully embedded within the medical visit. (17, 18) For patients that do not respond, care can be “stepped up” (eg, more intensive intervention outside of a medical visit with a trained provider). Stepped care has been shown to be feasible (18); however, it has not yet been tested in pediatric FAPD.

For those requiring stepped-up care, cognitive behavior therapy (CBT) is effective for treating pediatric chronic pain (19), including FAPD (20–22). CBT includes cognitive and behavioral approaches to improve pain-related functioning (19). However, a substantial proportion (~40%) of youth with FAPD fail to respond (20). This may be because anxiety attenuates the effect of pain-focused CBT (23). Bolstering pain-focused CBT with anxiety management techniques for those with anxiety may enhance treatment outcomes. Our research team has initiated anxiety screening and enhanced usual care (EUC) processes into gastroenterology clinics of a large children’s hospital (12). The team also developed and pilot tested the Aim to Decrease Anxiety and Pain Treatment (ADAPT) (21), a tailored CBT to target pain and anxiety in youth with FAPD. In the present study, those who failed to respond to enhanced care were eligible to receive ADAPT plus medical treatment as usual (TAU).

We examined feasibility (recruitment and retention rates) of stepped care, and response to ADAPT+TAU. We predicted that those who completed ADAPT+TAU would show greater reductions in functional disability, pain intensity, and anxiety than the TAU group.

Methods

Patients were recruited from outpatient pediatric gastroenterology clinics of a large academic medical center (Figure 1). Consent/assent from the primary caregiver and child were obtained for this IRB-approved study. Children ages 9–14 years-old with FAPD diagnosed by a pediatric gastroenterologist using ROME-IV guidelines (24) were eligible. To qualify, participants also had to show evidence of more than minimal disability (defined as a score of > 7 on the Functional Disability Inventory; FDI). This cut-off was previously established as a criterion for enrollment in prior pediatric pain behavioral trials (25). After study enrollment, gastroenterologists continued to offer TAU to all participants, which could include a referral for CBT. Given the wait for CBT generally exceeded the anticipated study period, this was not a barrier to participating, and study staff ensured clinical CBT and ADAPT did not overlap. The study was registered on ClinicalTrials.gov (NCT03134950).

Figure 1.

Consort Study Diagram. Two hundred and three children were approached, 139 were enrolled, and 110 remained eligible for stepped-up care after rescreening. Of those, 89 were randomized to ADAPT + TAU (n = 40 completing) or medical treatment as usual (TAU; n = 39 completing).

Procedures

This study evaluated the feasibility of a stepped care approach. All participants received Step 1. Participants that did not demonstrate a clinical response then received Step 2.

• Step 1: Screening and Enhanced Usual Care (EUC; Table I [available at www.jpedscom]). Patients were introduced to the study by a medical staff (12). A research coordinator then met with interested families. After consent, participants received Step 1, EUC: a brief pain-focused psychoeducation/relaxation training administered during their medical visit by a research coordinator or trained nurse (see https://steppedcare.research.cchmc.org/) (12). This website was also provided to participants for home practice. Usual care (part of EUC and TAU) at this center also consisted of use of pharmacotherapies that demonstrate at least some evidence-base for the management of FAPD pain (e.g., antispasmodic agents) by the GI provider. As part of usual care, providers had the autonomy to tailor treatment based on the needs of the patient. Two weeks later, participants were rescreened by a research coordinator via telephone. Those reporting low levels of disability (FDI ≤ 7) were considered EUC “responders” and completed the study. “Non-responders” (FDI >7) were invited to complete the next phase.

• Step 2: Assessment and ADAPT. Qualifying participants underwent an assessment consisting of a diagnostic interview (Anxiety Disorder Interview Schedule-IV [ADIS] (26)) and additional psychosocial measures (see Measures). If participants qualified following the assessment, they were randomized to either stepped-up care, consisting of ADAPT, a tailored CBT treatment (Table 1 and below) + TAU, or TAU alone. Eight weeks after randomization, all participants completed a follow-up assessment to re-assess outcomes.

Table 1.

Stepped Care and ADAPT Intervention Components for Youth with FAPD

Intervention	Provider	Platform	Protocol Used (Focus of Skill)
Step 1: Enhanced Usual Care a
	Clinic Nurse or Research Coordinator	In-person Web	Psychoeducation (Pain) Relaxation (Pain)
Step 2: ADAPT Intervention
Session 1	Psychologist	In-person	Program Overview Psychoeducation (Pain and Anxiety) Parent Guidelines (Pain) Deep Breathing/Guided Imagery (Pain)
Session 2	Psychologist	In-person	Progressive Muscle Relaxation (Pain) Calming Statements (Pain) Activity Pacing (Pain)
Session 3	Psychologist	Web and phone	Pleasant Activity Scheduling (Pain) Problem Solving (Pain)
Session 4b	Psychologist	Web and phone	Cognitive Restructuring (Anxiety)
Session 5b	Psychologist	Web and phone	Guided Exposure (Anxiety) Assertiveness Training (Anxiety)
Session 6	Psychologist	Web and phone	Maintenance Planning (Pain and Anxiety)

^a https://steppedcare.research.cchmc.org/

^bOptional modules administered over two sessions depending on presence of clinically significant anxiety. Participants without clinical anxiety received four weekly sessions of pain-focused ADAPT intervention only (i.e., two in-person and two web-based modules with interventionist phone support). Participants with clinical levels of anxiety received a total of six weekly sessions (i.e., two in-person and four web-based with interventionist phone support) consisting of both pain and anxiety management strategies.

Note. ADAPT = Aim to Decrease Anxiety and Pain Treatment; FAPD = Functional abdominal pain disorders; All ADAPT sessions were approximately 60 minutes. Web sessions of ADAPT were completed by child participant independently (30 minutes) followed by phone support (15–30 minutes), with the patient’s psychologist. Phone sessions were conducted primarily between the interventionist and child participant, with a brief portion of time reserved to update summarized content with the parents.

ADAPT.

ADAPT is brief, tailored intervention to treat pain and anxiety, and uses a blend of in-person visits and web modules with phone support to ensure consistent delivery (21). ADAPT was developed with NIH funding (F32HD078049, Cunningham PI) to address the unique needs of youth with FAPD. ADAPT teaches evidence-based cognitive behavioral strategies to cope with pain (25) and anxiety (27, 28), given that anxiety commonly co-occurs in this population (9–12, 16, 29) and is predictive of poor outcomes (14, 15). Pilot testing indicated good evidence of feasibility (21). ADAPT was delivered by doctoral psychologists supervised by licensed providers.

For this study, ADAPT (Table 1) was delivered in a stratified manner to be optimally tailored to the needs of youth with FAPD. All youth received coping skills training for pain as part of ADAPT, and additional components for anxiety management were offered if indicated. Specifically, youth with FAPD without evidence of clinical anxiety received four weekly sessions of pain-focused ADAPT (See Measures section for additional information). Two sessions were delivered in-person, and two were delivered via web modules with weekly psychologist telephone support. Youth with clinical anxiety received six weekly sessions total (two in-person and four web-based with weekly telephone support) consisting of pain management and anxiety management.

Treatment Integrity.

Treatment integrity was assessed using audio recordings of the in-person sessions by independent evaluators (e.g., doctoral and postdoctoral psychology trainees). Integrity scores (0–100%) were obtained. Items assessed included delivery of session-specific skills and assessment of participant comprehension.

Measures

Demographic and background information (including medication/supplement usage) were collected from the parent and from the patient’s electronic medical chart. Furthermore, clinically sensitive and psychometrically validated child informant measures were used, as youth with FAPD are more sensitive informants of their own symptoms (10).

Outcome Measures

Pain-Related Disability.

The Functional Disability Inventory – Child Version (FDI) is a 15-item measure of difficulty in performing activities in the past several days (30), is valid for youth with chronic pain (31), and is used in FAPD (10, 12, 21, 32). Higher scores indicate greater disability. Healthy youth on average have a total FDI score of 3.5 (30). Therefore, a score of >7 (double that value) was determined to be indicative of at least some disability due to pain for enrollment in the current study. An FDI score decrease of ≥ 7.8 points denotes a clinically meaningful treatment response (33) which was used for comparing ADAPT+TAU versus TAU outcomes, and there are cutoffs categorizing mild (<13), moderate (13–29), and severe (30+) disability (31). The FDI (all α’s ≥ .85) was completed at screening, re-screening, and post-treatment.

Pain Intensity.

Average pain over the past two weeks was assessed via a Visual Analog Scale (VAS), anchored with the words, “no pain,” and “worst pain,” (34). This was collected at screening, baseline, and post-assessment. A ≥ 3/10 is clinically significant (35).

Clinical Anxiety Measures.

The Screen for Child Anxiety Related Disorders/Child Report (SCARED) is a 41-item, measure of anxiety over three months (36, 37), and is widely used in pediatric pain research (12, 38–41). Higher scores indicate greater anxiety. A cut-off of ≥ 25 is clinically significant (37). The SCARED was administered at screening (α = .93) and post-assessment (α = .95). A 50% reduction in scores at post-treatment is considered a clinically meaningful indictor of improvement/remission (42).

The Anxiety Disorder Interview Schedule-IV (ADIS; Child Version) is a clinician-administered interview assessing psychiatric disorders in childhood (26, 43). Trained-to-criterion, blind-to-treatment-condition clinicians administered the ADIS. Diagnoses and Clinician Severity Ratings (CSR) ranging from 0–8 (scores ≥ 4 indicate a diagnosable condition) were obtained. The ADIS was administered at baseline and post-assessment. Inter-rater reliabilities were examined by reviewing audio recordings based on established guidelines (44). Agreement for no diagnosis (κ Range = .67–1.0) and the presence of an anxiety disorder at both pre- and post-treatment were appropriate (κ Range = .83–1.0). The κ coefficients obtained for matching principal anxiety disorders (GAD, Social Phobia, SAD, and specific phobias) were in the good to excellent range at pre- (κ = .64–1.0) and post-treatment (κ = .87–1.0).

Other Measures

Depressive Symptoms.

The Childhood Depression Inventory, Second Edition - (CDI 2) is a 28-item self-report measure of depression (45). Those with severe symptoms (CDI 2: T score > 80), or active suicidal ideation at baseline (α = .89) were excluded and referred for treatment.

Power and Analytic Plan

Power (.80) to detect a treatment effect difference (d = .50 for FDI) was n=40 per group. Descriptive statistics using SPSS v. 25 were computed to assess youth participation and response rate to the initial dose of stepped care. Missing data were minimal (≥ 96% of item-level data obtained) and were handled using maximum likelihood estimation (46) in Mplus. A structural equation modeling (SEM) equivalent of a MANCOVA was conducted to assess differences between ADAPT+TAU versus TAU completers on the primary outcome (FDI), and secondary outcome (VAS pain intensity), controlling for baseline outcome scores and age, as age has been shown to be positively correlated with higher FDI scores (10). Differences in anxiety symptoms controlling for baseline anxiety scores were also explored. Indicators of clinically significant change were also used to estimate the strength of the treatment effects.

Treatment effect moderation by anxiety and by number of sessions was also explored via a SEM equivalent MANCOVA and independent samples t tests. Effect sizes (95% CI) were calculated to understand effect magnitude. Differences in types and numbers of medications/supplements prescribed between ADAPT+TAU and TAU groups was also examined via chi-squared tests and an independent samples t test.

Results

Demographic information is presented in Table 2 (available at www.jpeds.com). The total sample (n = 139) ranged from ages 9–14 years (M = 11.65, SD = 1.68), and included 84 females (60%). The most common diagnoses based on the ROME IV checklist (24) completed by the medical provider were FAPD Not Otherwise Specified ([NOS]; n = 85, 61%) and Irritable Bowel Syndrome (n = 50, 36%). Most primary caregivers were mothers who completed high school. On average, youth presented with moderate levels of pain, functional disability, and anxiety (Table 3; available at www.jpeds.com).

Table 2.

Sample Characteristics

	Total Enrolled (n = 139)	Treatment Completers (n = 79)
Gender
Female	84 (60%)	47 (59%)
Male	55 (40%)	32 (41%)
Age
9 years	21 (15%)	12 (15%)
10 years	21 (15%)	13 (16%)
11 years	17 (12%)	9 (11%)
12 years	28 (20%)	12 (15%)
13 years	30 (22%)	18 (23%)
14 years	22 (16%)	15 (19%)
Race
White or European American	129 (93%)	71 (90%)
Asian American	0 (0%)	0 (0%)
American Indian	1 (1%)	1 (1%)
African American	4 (3%)	3 (4%)
Biracial	2 (1%)	1 (1%)
Other	1 (1%)	1 (1%)
Ethnicity
Hispanic/Latino	2 (1%)	2 (3%)
Pain Diagnosis
FAPD	85 (61%)	45 (57%)
IBS	50 (36%)	31 (39%)
Functional Dyspepsia	2 (1%)	1 (1%)
Abdominal Migraine	2 (1%)	2 (3%)
Participating Caregiver
Mother	108 (78%)	64 (81%)
Father	13 (9%)	7 (9%)
Grandmother	13 (9%)	5 (6%)
Grandfather	2 (1%)	1 (1%)
Foster Parent	3 (2%)	2 (3%)
Caregiver High School Completion
Mother	---	79 (100%)
Father	---	75 (95%)

Note. “Treatment” indicates completion of ADAPT+TAU or TAU. No significant differences in demographics between enrolled and treatment completers were detected. Parent high school completion was only assessed at baseline (not enrollment).

Table 3.

Descriptive Statistics and Bivariate Correlations of Study Outcome Variables

	M	SD	Range	2	3	4	5	6	7	8
Pre
1. FDI	17.87	8.17	8 – 47	.38**	.28*	.05	.39***	.25*	.30**	.21
2. VAS	3.75	1.86	0 – 8.7	--	.08	−.12	.27*	.53***	.04	.29*
3. SCARED	35.12	15.68	4 – 67		--	.34*	.24*	.02	.62***	.31*
4. CSR	6.08	1.31	4 – 8			--	.18	.13	.36**	.23 †
Post
5. FDI	11.33	9.48	0 – 41				--	.54***	.67***	.25 †
6. VAS	2.73	2.22	0 – 8.6					--	.26*	.32*
7. SCARED	25.46	16.56	1 – 63						--	.43**
8. CSR	3.79	2.23	0 – 8							--

Note. FDI (Functional Disability Inventory) assesses child report of functional disability. VAS (Visual Analogue Scale) assesses child report average pain intensity. SCARED (Screen for Child Anxiety Related Disorders) assesses child report of anxiety symptoms. CSR (Clinician Severity Rating) measures clinician perception of severity of the primary anxiety diagnosis during the Anxiety Disorders Interview Schedule. Table contains means and standard deviations for participants used in primary and exploratory analyses.

^†p < .10,

^*p < .05,

^**p < .01,

^***p < .001.

Stepped Care

Step 1: EUC

A total of 203 youth were approached between 2015 and 2018, and 139 (68.5%) enrolled and underwent EUC. The average FDI score was 17.9 (SD = 8.3). Research staff re-screened 120 youth via telephone approximately 2 weeks after their screening/gastroenterology clinic visit. Of those, only 10 (8%) reported their FDI levels reduced to no more than minimal (i.e., FDI ≤ 7; See Measures). Those with more than minimal disability (i.e., FDI > 7) after two weeks were scheduled for a baseline assessment (n = 110), of which 89 completed and remained eligible for randomization.

Step 2: ADAPT+TAU versus TAU

Qualifying youth were randomized (based on computer generated list) to ADAPT+TAU (n = 44) or TAU alone (n = 45), and 79 of those youth completed the study by the end of 8 weeks (ADAPT+TAU, n = 40; TAU, n = 39). The assignment was provided to research coordinators by the study statistician. There were no significant group differences in baseline variables.

Study outcome data are presented in Table 4. Participants who received ADAPT+TAU demonstrated significantly lower FDI levels at post-assessment compared with those randomized to TAU (b = −4.26, SE = 2.04, t = −2.09, p = .036; Figure 2). The effect size difference was moderate (Cohen d = 0.45). Moreover, there was an average 9.7-point reduction in FDI scores for ADAPT completers (compared with a 2.9-point reduction in the TAU group), which is indicative of clinically meaningful change (e.g., >7.8-point decrease) (33). In addition, 60% of those completing ADAPT+TAU had a clinically significant reduction in FDI scores compared with 28.1% of those completing TAU. Similarly, ADAPT+TAU completers had a 12.3% average VAS pain reduction, whereas TAU completers had a 5.5% average VAS pain reduction. The effect size difference was moderate (Cohen d = 0.30). However, the differences between groups on pain intensity was not statistically significant (b = −0.67, SE = 0.41, t = −1.61, p = .108; Figure 3).

Table 4.

Post-Treatment Changes in Child Symptoms Following ADAPT+TAU

	${\overline{X}}_{\mathit{\text{difference}}}$	SE	t-value	p
Primary Study Aims
Functional Disability at Post-Treatment
Age	−0.15	0.59	−0.26	.792
FDI (Pre-treatment)	0.46	0.14	3.22	.001
ADAPT+TAU subgroup	−4.26	2.04	−2.09	.036
Pain at Post-Treatment
Age	−0.17	0.12	−1.43	.153
VAS Pain (Pre-treatment)	0.60	0.10	6.13	.000
ADAPT+TAU subgroup	−0.67	0.41	−1.61	.108
Exploratory Study Aims
Anxiety Severity at Post-Treatment
CSR Anxiety Diagnosis (Pre-treatment)	0.29	0.20	1.46	.145
ADAPT+TAU subgroup	−0.51	0.55	−0.93	.350
Anxiety Symptoms at Post-Treatment
SCARED (Pre-treatment)	0.62	0.08	7.33	.000
ADAPT+TAU subgroup	−6.36	2.85	−2.23	.026

Note. ADAPT is Aim to Decrease Anxiety and Pain Treatment. TAU is medical treatment as usual. ADAPT+TAU subgroup represents dummy coded variable with 0 = TAU only and 1 = ADAPT+TAU. DV=dependent variable. FDI (Functional Disability Inventory) assesses child report of functional disability. VAS (Visual Analogue Scale) assesses child report average pain intensity. SCARED (Screen for Child Anxiety Related Disorders) assesses child report of anxiety symptoms. CSR (Clinician Severity Rating) measures clinician perception of severity of the primary anxiety diagnosis during the Anxiety Disorders Interview Schedule.

Figure 2.

Functional disability inventory (FDI) scores from screening to re-screening generally remained within the moderate range of disability. After randomization to ADAPT + TAU or TAU, both groups experienced a reduction in FDI scores, with the ADAPT+TAU group experiencing the most pronounced improvement. The ADAPT+TAU group had significantly lower FDI scores at post compared with the TAU group.

Figure 3.

Average scores on the Visual Analog Scale (VAS) for pain remained in the moderate range from screening to baseline. At post assessment, both ADAPT+TAU and TAU groups experienced a reduction in pain symptoms. For ADAPT+TAU completers, the reduction in pain symptoms was clinically significant but was not statistically different compared with those completing TAU.

Anxiety at post-treatment, controlling for pre-treatment anxiety, was also explored (Table 4 and Figure 4 [available at www.jpeds.com]). Children receiving ADAPT+TAU reported lower SCARED scores at post-treatment as compared with those who received TAU (b = −6.36, SE = 2.85, t = −2.23, p = .026). The effect size was moderate (Cohen d = 0.39). The average SCARED score percent decrease was 33% for ADAPT+TAU versus 16.5% for TAU; 13out of 40 (32.5%) youth experienced at least a 50% reduction in SCARED scores after ADAPT+TAU compared with 6 out of 39 (15.4%) after TAU. There were not significant differences in the primary anxiety disorder diagnosis CSR’s at post-treatment when comparing the ADAPT+TAU and TAU groups (b = −0.51, SE = 0.55, t = −0.93, p = .350). However, the effect was in the expected direction, suggesting severity of the primary anxiety disorder tended to decrease following ADAPT+TAU compared with TAU.

Figure 4 (online).

Patients with FAPD generally presented with clinically elevated levels of anxiety. Those who completed ADAPT+TAU were more likely to have statistically and clinically significant reductions in anxiety symptoms at post-assessment. Note: The SCARED was not readministered at baseline because scores are valid for 3 months following administration.

Treatment Integrity.

Treatment integrity for ADAPT was found to be excellent (M = 98%, Range = 88% to 100%).

Adherence.

Of the 44 individuals assigned to ADAPT, four were lost to attrition (9%). Of these individuals, two participated in at least one in-person session and two did not complete any component of ADAPT treatment. Of ADAPT completers (n = 40), 75% (n = 30) were assigned the six-session version and 25% (n = 10) were assigned the four-session version.

All 40 ADAPT completers participated in the two in-person sessions and weekly therapist telephone calls. Adherence data to the web portion of ADAPT were calculated based on previously established guidelines (21). The percentage of available videos and handouts viewed, and forms completed was calculated for each patient: Participants watched, on average, over half of the available videos (M = 57%, SD = 37%, Range = 0%−100%). On average, patients also downloaded 21% (SD = 29%, Range = 0%−100%) of the handouts and completed 29% (SD = 22%, Range = 0%−64%) of the online forms. Eight patients did not complete any online content.

Moderation Analyses.

Analysis showed baseline SCARED scores did not moderate effects on FDI and average pain intensity at post. Further, ADAPT dose (4 versus 6 sessions) did not moderate effects on FDI and average pain intensity at post. Baseline SCARED scores did moderate effects on post-treatment SCARED scores (t = −18(37), p < 0.001). Specifically, children who were more anxious were more likely to demonstrate greater reductions in their SCARED scores following ADAPT. These differences were expected given that ADAPT treatment type was assigned based on SCARED anxiety levels. ADAPT dose by treatment did not moderate effects on post-treatment SCARED scores.

Medication Usage.

In the ADAPT+TAU group, the most common medications and supplements prescribed by the GI provider were: antispasmodic agents (n=24, 60%), acid reduction therapies (n=14, 35%), laxatives (n=12, 30%), peppermint oil (n=6, 15%), probiotics (n=5, 12.5%), low dose psychotropic agents (n=3, 7.5%), and anti-nausea medications (n=2, 5%). Anti-diarrheal medication, anti-ulcer/protectants, and gas reducing medication were also infrequently prescribed (all n’s =1, 2.5%). The average number of GI medications/supplements prescribed was 1.75 (SD=1.0, range 0–5). In the TAU group, the most common medications and supplements prescribed by the GI provider were: antispasmodic agents (n=19, 48.7%), acid reduction therapies (n=19, 48.7%), laxatives (n=13, 33.3%), low dose psychotropic agents (n=4, 10.3%), anti-nausea medications (n=4, 10.3%), peppermint oil (n=3, 7.7%), and probiotics (n=2, 5.1%). The average number of GI medications/supplements prescribed was 1.82 (SD=0.98, range 0–4). There were no significant differences between ADAPT+TAU and TAU groups on types of medications prescribed nor numbers of medications prescribed.

Discussion

Findings of this randomized clinical trial of ADAPT suggest that a brief, stepped care approach is highly feasible for managing pain and anxiety in youth with FAPD within the context of standard medical care. Moreover, the easily accessible tailored CBT intervention (ADAPT) plus TAU may be effective for reducing pain-related disability and anxiety symptoms. These findings are critically important because FAPD are among the most common childhood pain conditions (1, 2); yet, there is currently limited evidence to support the use of pharmacologic treatments (47, 48). The most potent intervention for the management of pediatric FAPD (22) and other pediatric chronic pain conditions (19) involves the use of nonpharmacologic treatments such as CBT, which are often difficult to provide due to limited access and the need for multiple in-person visits. CBT for pain management also does not always directly target anxiety, which is highly prevalent in FAPD (8–11) and adversely impacts response to CBT (23). Thus, the ADAPT intervention, which targets pain and anxiety when appropriate, has the potential to substantially improve patient outcomes. The stepped care approach (17, 18), which begins by targeting all-comers, is practical to employ in a medical setting and provides flexibility, recognizing that patients with FAPD may require varying levels of care (15).

Based on the study findings, it may be feasible for patients with FAPD to receive psychoeducation and basic relaxation strategies for pain management as part of standard medical treatment. For those who fail to respond to the initial treatment, which in this investigation constituted the vast majority of patients, additional care may be warranted. For such patients, a brief CBT, such as ADAPT, which includes both in-person and web-based/telehealth sessions (21), may bolster patient outcomes.

Youth with FAPD demonstrated improvements in pain-related functional disability and anxiety symptoms following ADAPT+TAU. This is important because the presence of anxiety in conjunction with FAPD is quite common (8–12, 16). Further, anxiety is associated with increased pain and disability (10, 12–14), and adverse outcomes in youth with FAPD in cross-sectional (10, 13) and longitudinal studies (14, 49). Left untreated, youth with FAPD are prone to long-term mental health issues (49). This tailored approach to simultaneously address pain-related disability and anxiety in FAPD may improve patient outcomes over the long term. Indeed, the goal of CBT is to teach youth coping strategies that will allow them to function despite pain. Therefore, prior literature supports the notion that disability outcomes tend to improve before pain improves (50), which is congruent with the study results.

The importance of using a CBT treatment approach integrated into medical care and includes telehealth delivery should not be understated, particularly given the COVID-19 pandemic and the increasing demand for telehealth care. Such an approach provides increased accessibility to care (particularly for vulnerable families, and those living far from the medical center) and has important implications for the future of psychological treatments for youth with chronic pain. Indeed, ADAPT offers some advantages over exclusively web-based approaches for pain management by allowing patients to build rapport with a therapist. Direct provider-patient contact for both medical and psychological needs is important for non-specific therapeutic effects, and there is support that remote patient-provider contact (e.g., phone or live video conferencing) achieves a therapeutic bond comparable with traditional in-person delivery of psychological treatments (50). In the current investigation, adherence to both the live in-person and telehealth/phone portions of ADAPT was quite high, whereas the variability in the completion of the web components of ADAPT was consistent with adherence rates generally observed during web-based interventions (52).

Given psychological providers with expertise in the management of pediatric chronic pain are limited, additional research would be valuable to determine if ADAPT can be delivered completely remotely (e.g., in person sessions converted to telehealth sessions), and if components of ADAPT can be delivered independently by a member of the medical team (e.g., nurse delivered) in certain cases. In addition, given ADAPT+TAU appears to be effective, it is important to explore mechanisms to better understand why such a treatment works. Examination of underlying neural mechanisms, for example (53), may allow for further tailoring and enhancement of treatment approaches.

Integration of behavioral health within standard medical care provides youth with increased access to the most effective treatment for pain management, de-stigmatizes mental health concerns, increases patient/family buy-in, and offers a systematic and evidence-based approach to treating youth with FAPD. For these reasons, it would be valuable to test such a treatment model in other settings, such as primary care, where behavioral health integration for the management of mental health concerns, such as anxiety, has already shown benefits (54). It would also be valuable to test and adapt such models with other pediatric chronic health conditions involving pain, such as migraines. In our study, EUC demonstrated limited benefits, but it would be helpful to explore if this “enhancement” offered any additional improvement for patients over and above standard care. It may also be beneficial to extend the length of EUC from 2 weeks to 4 weeks in a future large-scale trial. It may also be helpful to consider an approach that is initially stratified (versus stepped) –as patients would immediately be eligible to receive more intensive treatment rather than waiting until they failed stepped care if they presented with more severe levels of disability/anxiety from the outset.

This single-site pilot RCT did not have an active treatment control beyond TAU, and our sample consisted mostly of white female participants, which limits generalizability; however, findings offered preliminary evidence of the positive effects of this innovative approach to care. We offered this intervention to younger youth (9–14 years of age) with a goal of offering a targeted intervention to prevent adverse outcomes over the longer term but believe a similar program could be beneficial for older adolescents as well (21). A larger-scale, multi-site study with longitudinal exploration of outcomes (including testing if refresher/maintenance sessions would enhance outcomes) would allow for further understanding of treatment effects. It would also be informative to compare components of ADAPT to determine if certain strategies (e.g., cognitive vs. behavioral) are more effective than others and if the inclusion of additional components (e.g., attention bias training) would further enhance treatment. Finally, it would be informative to compare other active treatments such as standard CBT for pain to ADAPT, in addition to accounting for the use of specific pharmacotherapies in treatment. In the current examination, there were no differences in types of medications or supplements prescribed across ADAPT+TAU and TAU groups. Although there was not an effect based on the ADAPT dose received, the treatment dose varied within the CBT arm (25% received 4 sessions, 75% received 6 sessions). This stratified approach to treatment allowed for the heterogenous FAPD patient population to receive an intervention that was appropriately tailored to the unique symptom presentation (e.g., with or without clinical levels of anxiety), and therefore has optimal clinical utility.

Although a stepped care approach is feasible, youth with FAPD appear to benefit from the stepped-up (CBT) approach for pain and anxiety management. This approach is feasible for improving health- and mental-health related outcomes in youth with FAPD. By offering tailored CBT to affected youth, accessibility to care is enhanced for those with greatest need. Further, tailoring care based on symptoms and need may enhance outcomes while maximizing the use of limited therapeutic resources.

Abbreviations:

ADAPT

Aim to Decrease Anxiety and Pain Treatment

CBT

Cognitive Behavior Therapy

FAPD

Functional Abdominal Pain Disorders

FDI

Functional Disability Inventory

SCARED

Screen for Child Anxiety Related Disorders