The comfort ability program for adolescents with sickle cell pain: Evaluating feasibility and acceptability of an inpatient group-based clinical implementation

Abstract

Background:

Integration of nonpharmacological therapies, such as cognitive and behavioral pain management strategies, is recommended to support comprehensive disease and pain management among children and adolescents with sickle cell disease (SCD). The Comfort Ability Program for Sickle Cell Pain (CAP for SCP) introduces psychological and biobehavioral pain management strategies to children and adolescents with SCD. This study aimed to pilot the implementation of the CAP for SCP in a group setting to children and adolescents hospitalized for SCD pain examining feasibility, acceptability, and preliminary effectiveness on improving pain knowledge and coping efficacy.

Method:

Adaptation of CAP for SCP into a three-session group format was guided by four phases of the Dynamic Adaptation Process model: Exploration, Preparation, Implementation, and Sustainment.Youth with SCD (n=57) hospitalized for pain participated in at least one session and completed self-report of knowledge of pain management skills, pain coping efficacy, and treatment acceptance. Completion rates of sessions and qualitative feedback were gathered to evaluate feasibility and acceptability.

Results:

Feasibility of conducting inpatient group sessions was suboptimal; however, patients and medical providers reported moderate to high levels of treatment acceptance. Patients also reported significant improvements in knowledge of pain management skills following session 1.

Conclusions:

CAP for SCP is a patient-centered first-line psychoeducational intervention that can be integrated into clinical practice settings to introduce youth to cognitive and behavioral pain management strategies to support SCD pain management.

1 ∣. INTRODUCTION

Pain is a prevalent feature of pediatric sickle cell disease (SCD) that significantly contributes to declines in health-related quality of life.¹ Several evidence-based nonpharmacological treatments, such as behavioral health approaches, are recommended to support comprehensive pain care.^2,3 Behavioral health intervention, including cognitive-behavioral therapy (CBT) for pain, is efficacious in reducing pain intensity immediately following treatment among youth with a variety of pain conditions (e.g., musculoskeletal pain, chronic headache) and in reducing pain-related disability up to 12 months later.⁴ Although the feasibility, acceptability, and efficacy of CBT among youth with acute and recurrent SCD pain is understudied,⁵ recent evidence suggests that CBT is highly acceptable to adolescents and parents when administered during inpatient hospitalizations or in conjunction with routine outpatient clinic visits.⁶ Additionally, youth with SCD who established care using CBT for chronic pain management demonstrated reductions in emergency department reliance, faster reductions in inpatient admissions and length of stay over the following 12 months relative to comparisons who did not initiate CBT, and reported improvements in daily functioning, improved coping efficacy, and lower pain intensity.⁷

CBT for pain focuses on improving daily functioning and coping by targeting a combination of maladaptive thoughts, feelings, and behaviors that may maintain the negative impact of pain on quality of life. CBT is usually taught by licensed mental health professionals with expertise in pain management in an outpatient practice for 6–12 weekly sessions. Implementation of evidence-based treatments like CBT into clinical practice is often challenging, especially in the pediatric SCD setting where behavioral health resources are often limited due to various factors, such as limited provider availability, lack of institutional support, geographic limitations, scheduling difficulties, financial or insurance constraints, limited knowledge of the efficacy of behavioral interventions, and patient/family experience of stigmatization related to mental health referrals.^8-10 These barriers can significantly interfere with the provision of effective behavioral health services to children and families who would most benefit from them.

To help facilitate the implementation of CBT in clinical practice, the Comfort Ability Program for Sickle Cell Pain (CAP for SCP) was developed as a structured 2.5-hour psychoeducation and CBT intervention for pain management.⁶ It is an interactive video-based intervention designed to introduce psychological and biobehavioral pain management strategies to youth with SCD and their parents in a culturally sensitive and nonstigmatizing format. In a previous feasibility study, CAP for SCP was administered individually to adolescents by a psychologist or trained child life specialist over the course of a brief hospital stay (1–3 days) or during a routine outpatient hematology clinic appointment. The program was found to have high levels of acceptability and satisfaction among adolescents and parents and resulted in increased patient knowledge of nonpharmacological pain management strategies.⁶ The initial pilot demonstrates potential for further implementation and adaptation to increase accessibility of this evidence-based intervention.

The overall goal of this project was to pilot a flexible application and adaptation of the CAP for SCP. The Dynamic Adaptation Process (DAP) conceptual model provided structured guidance for flexibly adapting CAP for SCP while maintaining the fidelity of the treatment to meet local institutional needs.¹¹ Adaptation was indicated as provision of psychoeducation and CBT on an individual basis to patients with SCD during inpatient admissions for pain crisis, is time- and resource-intensive when behavioral health availability is already limited. Delivery of the same content in a structured group format has the potential to maintain the intervention’s impact, improve efficiency, and reach more patients.¹² This project illustrates a systematic approach to behavioral health intervention modification that can enhance dissemination and inform clinical practice in other pediatric SCD settings.

This pilot aimed to improve the clinical utility and accessibility of CAP for SCP by using a group setting of children and adolescents hospitalized for SCD pain.The primary aim assessed feasibility and acceptability of the CAP for SCP group format. We expected high feasibility, defined as successful administration of ≥75% of planned sessions in a group format with ≥50% of patients completing the full program. Acceptability was expected to be moderate to high, defined as ≥75% patient retention during sessions (i.e., patient stayed for the entire session), patient-reported treatment acceptance, and positive qualitative feedback from patients and providers. Benchmarks were informed by previous studies of CBT intervention for youth with SCD or chronic pain.^6,13 The secondary aim assessed preliminary effectiveness of CAP for SCP on patient-reported knowledge of pain management skills and pain-coping self-efficacy from the pre- to post-intervention sessions. Patients were expected to report improvements in knowledge of pain management skills and coping self-efficacy.

2 ∣. MATERIALS AND METHODS

2.1 ∣. Setting and participants

The comprehensive pediatric SCD program serves over 2000 children annually across three tertiary care hospital settings and employs hematologists, advance practice providers, and a multidisciplinary family support team, lending itself well to support a group intervention. Eligibility criteria included: patient with SCD of any genotype, aged 8–18 years, fluent in English, admitted to the hospital for sickle cell-related pain with an anticipated length of stay >1 day. Initial exclusion criteria included: admitted to the hospital for chronic pain or acute pain superimposed on chronic pain, diagnosed with significant developmental delays that would interfere with group activity, active suicidal ideation, previously completed all CAP sessions, or medical providers identified a contraindication to group participation (e.g., contact precautions). After 3 months of project initiation, patients hospitalized with chronic pain or acute pain superimposed on chronic pain who had not previously engaged in CBT for pain were interested in participating after hearing about the program during hospitalization. Chronic pain or acute pain superimposed on chronic pain was removed from the exclusion criteria.

2.2 ∣. Intervention adaptation

The DAP guided the adaptation of the CAP for SCP to support modification to a group format and effective evidence-based implementation. Strategies used to support phases of exploration, preparation, and implementation are described (see Figure 1). The DAP model presented in Figure 1 outlines the different components, perspectives, stakeholders, and considerations made during each phase of development, while highlighting that these phases are iterative and learnings from one phase can inform upon previous phases, creating a feedback loop. Outcomes related to the Sustainment phase are detailed in Section 3.

FIGURE 1.

Dynamic Adaptation Process model phases that guided adaptation of the Comfort Ability Program for Sickle Cell Pain

2.2.1 ∣. Phase 1: Exploration

As outlined in Figure 1, system- and organization-level considerations were explored to help determine possible implementation of CAP for SCP. The primary investigator (PI) and intervention developer discussed potential strategies of implementing the CAP for SCP at the local institution. The PI met with the respective division-wide leadership teams to discuss interest and buy-in for the program to use time and resources to implement the group intervention. As inpatient pain is a common referral problem in SCD, the leaders were receptive to piloting the group to benefit patients and families. Project activities were reviewed by the hospital’s Institutional Review Board (IRB), which determined that the activities represented a quality improvement initiative not requiring informed consent for research from participants because it was integrated into routine clinical services and unique to the institution. IRB approval was obtained for retrospective medical chart review to gather patient demographics. Securing private space to conduct the groups was a primary logistical barrier. The hospital has dedicated spaces for teenagers on the inpatient floor, which was utilized to conduct the groups. Inclusion and exclusion criteria were set and agreed upon with the SCD medical team. Stakeholders (e.g., child life specialists, music therapists, medical team) were engaged to help with patient engagement before and during the groups. All were receptive to the implementation of this group intervention.

2.2.2 ∣. Phase 2: Preparation

To develop the Implementation Resource Team (Figure 1), members of the psychology consultation-liaison team for hematology/oncology set aside time for training in the intervention to ensure consistent treatment implementation and fidelity and served as the primary interventionists. Academic researchers identified the methods of assessment for monitoring outcomes and assessment timing schedule. Intervention developers adapted CAP for SCP into a three-session program and gathered and reviewed feedback for iterative changes (see Intervention and Modifications). Patients were each given individual folders with intervention materials requiring administrative and financial support to generate and maintain these materials. Program education was provided to all individuals involved in inpatient care (e.g., medical providers, nurse manager, and nurses) via presentations and ongoing conversations to assist in patient education and encouragement from the team for patient participation.

2.2.3 ∣. Phase 3: Implementation

Implementation of CAP for SCP was integrated into the routine clinical services provided by the psychology consultation team. On days on which there was only one available psychology team member, Interventionists prioritized demands for patient care (e.g., emergent patient consultations) and conducting CAP for SCP sessions, requiring flexibility for intervention delivery schedule. Interventionist support was provided via monthly team meetings to discuss any implementation or logistical issues and to provide problem-solving opportunities. Feedback was gathered from inpatient care team about their perceptions of implementation, perceptions shared with them by patients, families, and other nurses, and potential barriers and facilitators to continued implementation during weekly SCD team meetings and individual patient care discussions. Iterative feedback informed ad hoc adaptations to implementation (see Figure 1). Patients provided feedback on feasibility, acceptability, and preliminary effectiveness.

2.3 ∣. Intervention and modifications

CAP for SCP is an interactive, multimedia intervention that includes (1) a 63-page fillable workbook with resource guide; (2) a 40-minute video designed to provide psychoeducation, SCD health education, social support (via patient stories), and modeling/instruction of relaxation-based pain management skills; and (3) eight audio-based guided relaxation exercises to support in vivo learning and practice of skills. CAP for SCP was modified from its original format of a 1-day workshop lasting 2.5 hours into three, 1-hour modules delivered over 3 days to adapt to the setting for implementation. Table 1 describes intervention content by session.

TABLE 1.

Comfort Ability Program for Sickle Cell Pain intervention content

Session	Session content
1	• Education about sickle cell disease and pain • Self-management strategies to stay healthy • Exacerbating factors for pain, including stress • Diaphragmatic breathing
2	• Identifying negative and positive thoughts • Linking thoughts, feelings, and behaviors • Benefits of relaxation for pain and stress • Muscle relaxation
3	• Guided imagery • Body/mind techniques • Pleasant and distracting activities • Identifying signs of depression and anxiety • Sleep habits • Provision of additional resources

2.4 ∣. Procedures

The CAP for SCP intervention was administered in a private communal space in the hospital. Sessions were provided in an open-group format, such that patients could attend any session. Patients were encouraged to participate in sessions consecutively, but to support patient inclusion and participation, patients were able to attend group sessions in any order. Sessions were generally planned for the first and third week of every month for 11 months. Interventionists identified eligible patients during rounds and conferred regarding their eligibility with the SCD treatment team. The interventionist shared a group flyer detailing the date, time, and location with the medical team for a team member (e.g., nurse practitioner) to provide to the patient during family-centered rounds. Prior to starting the group, the interventionist stopped by the patient’s room to confirm their participation. Child life specialists and nursing staff helped to prepare patients for the group and move them to the private space for the group. Group sessions were conducted by a CAP for SCP-trained psychology provider.

Sessions included a combination of patients watching video-based program content that introduced pain management topics, completing workbook exercises, and group discussion. Patients reported on their knowledge of pain management topics and pain coping efficacy at the beginning and end of each session. Patients also reported on session acceptability at the end of each session. Patient participation in session was clinically documented as appropriate in the electronic medical record (EMR). A one-page educational summary document was shared with parents/caregivers after completion of the session.

2.5 ∣. Metrics and measures

2.5.1 ∣. Demographic and medical characteristics

Patient characteristics were abstracted from the EMR and included age, sex, race, ethnicity, SCD genotype, and pain intensity rating (0–10 numerical rating scale) reported within 4 hours prior to session.

2.5.2 ∣. Feasibility

Feasibility was assessed using metrics tracked by providers surrounding session occurrence including: (1) number of completed sessions; (2) number of completed sessions implemented in a group format (two or more patients/session); and (3) total number of intervention sessions completed by unique patients to assess patient access and engagement with the intervention across the full three-session program.

2.5.3 ∣. Acceptability

Session metrics tracked patient acceptability with respect to patients’ withdrawal from a session (i.e., leaving mid-session).

The Abbreviated Acceptability Rating Profile (AARP)¹⁴ assessed patient’s pain treatment acceptance with each intervention session. Patients rated their beliefs about the treatment from 1 (strongly disagree) to 6 (strongly agree) at the end of each session. Total scores ranged from 8 to 48, with higher scores indicative of greater acceptability. The AARP has been shown to have good internal consistency and acceptable reliability and validity across racially diverse populations of varying educational and socioeconomic backgrounds, including youth with SCD and their parents.⁶ In this sample, the internal consistency ranged from acceptable to good (α = .75 for session 1, α = .70 for session 2, α = .85 for session 3).

Qualitative feedback was gathered each month from the treatment team conducting sessions to identify barriers and inform iterative procedural refinements to enhance feasibility and acceptability. As feedback was typically provided within the context of patient care discussions, psychology team members informally documented qualitative feedback in lieu of utilizing qualitative methods (e.g., audiorecording, transcribing) to maintain patient privacy.

2.5.4 ∣. Preliminary effectiveness of the intervention

The following patient self-report measures were administered pre- and post-intervention for sessions 1, 2, and 3.

The Knowledge of Psychological Interventions for Pain¹⁵ is a 16-item measure that assessed knowledge of how pain works in the body and specific psychological and biobehavioral interventions for pain management. For the purposes of this project, the measure was divided into three separate questionnaires to assess knowledge of the content covered in each of the three sessions. Items were multiple choice with five possible answers and were completed by patients at the beginning and end of the session. This measure was previously used with the CAP to evaluate knowledge gained after participation in a pain coping skills intervention in other populations, with >75% correct considered a benchmark for adequate knowledge.¹⁵ For each session, correct items were summed and transformed to a total correct percentage score (0–100%), with higher scores suggestive of greater patient pain knowledge. Change in pre- to post-session percentage scores as well as proportion of patients exceeding the 75% correct benchmark assessed knowledge changes over the course of the session.

The Child Pain Coping Self-Efficacy Scale¹⁶ is a seven-item measure used to assess patient confidence and perceived ability to function as usual with pain (e.g., how sure they are that they can complete specific activities when in pain). Items are rated on a 5-point scale from 1 (Pretty sure) to 5 (Very unsure). Items are summed, with total scores ranging from 7 to 35, with higher scores indicating lower self-efficacy. This measure has shown excellent reliability and construct validity among samples of children with pain.^15,16 Total scores were assessed pre- and post-session for each of the three sessions.

2.6 ∣. Statistical analysis plan

Data were entered into Microsoft Excel and then exported into SPSS version 26 for analyses. Descriptive statistics on patient demographics and outcome variables were computed to determine whether the data met the underlying assumptions of the proposed analytic procedures. The t-tests and chi-square analyses evaluated patient demographics across the three hospital locations to ensure no systematic source of bias across sites.

Missing data for patients who attended sessions but did not complete self-report measures were evaluated using independent t-tests (for continuous variables) and chi-square analysis (for categorical variables) for patient demographics (i.e., age, sex, SCD genotype), pre-session pain intensity rating, and hospital campus to determine whether data were missing at random and inform inclusion of potential covariates.

Feasibility was assessed by calculating participation and completion rates of sessions. Acceptability was evaluated through descriptive statistics and item-level analysis of self-report measures of treatment acceptance and from spontaneous qualitative feedback from patients and providers. Paired t-tests evaluated pre- to post-session differences in pain knowledge and pain coping efficacy for sessions 1, 2, and 3. The proportion of patients exceeding the established >75% correct benchmark for pain knowledge was explored from pre- to postsession. Lastly, coping item-level comparisons were explored to consider descriptive trends from pre- to post-session.

3 ∣. RESULTS

3.1 ∣. Patient cohort

Fifty-seven patients with SCD between 8 and 18 years of age (mean [M] = 13.33 years, standard deviation [SD] = 2.37) participated. All patients identified as non-Hispanic Black, and most patients were female (n = 35, 61%). The most common SCD genotype among participants was hemoglobin (Hb)SS (n=42, 73.7%), followed by HbSC (n=6, 10.5%), HbSβ⁰-thal (n = 5, 8.8%), HbSβ⁺-thal (n = 3, 5.3%), and HbSβO-Arab (n = 1, 1.8%), respectively. There were no differences in demographic characteristics (e.g., child age, sex, SCD genotype) across the three hospital campus locations (all p-values >.60); therefore, data for the three hospitals were combined for all subsequent analyses.

As illustrated in Figure 1, the Sustainment phase of the DAP model integrated outcomes based on feasibility, acceptability, preliminary effectiveness, and patient and provider feedback to inform sustainability of the CAP for SCP group format for implementation in an inpatient setting.

3.2 ∣. Feasibility

3.2.1 ∣. Conducting inpatient sessions

Figure 2 illustrates the participation and completion rates of sessions. There were a total of 177 preplanned opportunities for sessions to occur (between n = 51 and 69 planned sessions per site). While inpatients were eligible for group participation during 159 (or 89.8%) of these opportunities, interventionists were available for 96 (or 54.2%) of the sessions. Primary barriers to interventionist availability were time conflict (e.g., emergent patient care activities during the time of planned session) or no trained providers were on service that day (e.g., provider vacation or illness).

FIGURE 2.

Flowchart depicting feasibility of conducting planned Comfort Ability Program (CAP) for Sickle Cell Pain (SCP) group sessions. Note: ^†indicates the denominator used for subsequent percentage calculations; N refers to the number of sessions. Group format possible indicates that more than two eligible inpatients were available to participate. Individual format only possible indicates that only one eligible inpatient was available to participate. In the case that individuals declined participation when a group format was possible, the session was conducted in the individual format, thus that session became a completed individual format session

There were 67 planned sessions (37.9%) where eligible inpatients and interventionists were available. Of these sessions, 77.6% (52/67) were successfully completed. Based on qualitative feedback, primary system-level barriers to completion included patient-prioritized involvement with inpatient medical providers (e.g., engaged in a medical procedure), completing an alternative activity (e.g., physical therapy, school, hospital-wide activity), or discharge was planned on the day of the session. Common patient-level barriers to participation included high pain intensity, sleeping, perceived movement restriction (e.g., connected to IV medication), discomfort with personal appearance, or other unknown reasons.

3.2.2 ∣. Implementing inpatient group format

When both eligible patients and interventionists were available for planned sessions, a group format was possible during 74.6% (50/67) of sessions and only an individual format (i.e., only one eligible participant was inpatient) was possible during 25.4% (17/67) of sessions. Approximately 35% of completed sessions (18/52) were conducted as groups, whereas 65% were conducted individually.

3.2.3 ∣. Patient retention across the program

A total of 159 non-unique patients were eligible for inclusion (i.e., same patient may have been eligible for multiple sessions over multiple admissions), with an overall participation rate of 46.5% in at least one of the three CAP for SCP sessions. Of the 57 unique patients who participated, 64.9% (n = 37) completed one session, 19.3% (n = 11) completed two sessions, and 7.0% (n = 4) completed all three sessions. All patients who participated in two or more sessions completed sessions consecutively.

3.3 ∣. Intervention acceptability

Over 91% of patients who attended a session completed the encounter in its entirety, whereas 8.8% (n = 5) initiated a session but discontinued mid-session secondary to acute illness or symptoms (e.g., increase in pain, fell asleep). Self-report patient ratings found moderate to high levels of treatment acceptance for sessions 1 (M = 35.91, SD = 7.23, range 22–48), 2 (M=37.65, SD=6.74, range 24–46), and 3 (M=36.63, SD = 7.89, range 26–48). Based on item-level analysis, most patients reported they “somewhat agree” to “strongly agree” that this treatment will help them (97% at session 1; 89% at session 2; 73% at session 3), they would be willing to use this treatment for their pain (91% at session 1; 78% at session 2; 82% at session 3), that this treatment is a good way to handle their pain (90% at sessions 1 and 3; 83% at session 2), they liked the treatment (88% at session 1; 89% at session 2; 82% at session 3), that the treatment should be effective in changing their pain (85% at session 1; 89% at session 2; 82% at session 3), and they learned new information (60% at session 1; 72% at session 2; 82% at session 3).

Qualitative feedback from patients revealed that learning new information about SCD and pain management strategies and the interactive nature of sessions (i.e., use of multimedia, interaction with other patients) contributed to their overall enjoyment of the intervention. However, some patients expressed already knowing the information presented during sessions or discomfort with attending a group-based activity given their pain intensity or concerns of physical appearance. Interventionists expressed positive feedback regarding the use and delivery of interactive materials (i.e., workbook, videos, skill building). Timely progression through sessions was often noted as challenging based on patient reading level and reading materials aloud supported patient engagement and discussion of session content. Qualitative feedback from medical providers highlighted appreciation and enthusiasm for groups, including requests for groups to be offered more regularly, expanding groups to the outpatient setting for non-hospitalized patients, and an expansion of eligibility criteria, which were integrated into the project design.

3.4 ∣. Preliminary effectiveness

Missing data for patients who attended sessions but did not complete self-report measures revealed 19% (8/42) missing self-report data for session 1, 33% (8/24) missing at session 2, and 14% (2/14) missing at session 3. Missing self-report data were not related to child age or presession pain intensity rating (t < 0.6, p > .80), patient sex, SCD genotype, or hospital campus (X² < 6.0, p > .18).

Significant improvements in pain knowledge were observed from pre-intervention (M= 48.44, SD = 22.94) to post-intervention for session 1 only (M=58.86, SD=24.31; paired t-test [33] = −3.90, p<.001) (see Table 2). Exploratory analysis of the percentage of participants who reported >75% correct on knowledge items increased for session 1 (9% at pre-session, 28% at post-session), session 2 (30% at presession, 50% at post-session), and session 3 (0% at pre-session, 15% at post-session).

TABLE 2.

Comparison of pre- and postintervention pain knowledge and coping scores by group sessions

		Pre-session		Post-session		Post–Pre (paired)
	N	Mean (SD)	Median (IQR)	Mean (SD)	Median (IQR)	Mean (SD)	p-Value
Session 1
Pain knowledge	34	48.44 (22.94)	50.00 (33.33–66.67)	58.86 (24.31)	50.00 (50.00–83.34)	10.42 (15.12)	<.001
Coping	34	22.59 (6.00)	23.00 (18.50–26.50)	23.18 (5.78)	22.50 (20.00–27.00)	0.59 (5.66)	.55
Session 2
Pain knowledge	20	60.00 (25.02)	66.67 (50.00–83.34)	64.17 (29.75)	75.00 (50.00–83.34)	4.17 (22.86)	.43
Coping	16	23.56 (5.56)	25.00 (19.50–28.75)	23.81 (5.32)	25.00 (20.00–28.50)	0.25 (2.70)	.72
Session 3
Pain knowledge	13	50.00 (16.67)	50.00 (33.33–66.67)	56.41 (24.09)	66.67 (41.67–66.67)	6.41 (12.80)	.09
Coping	12	22.54 (4.42)	22.00 (18.75–26.00)	22.42 (4.48)	22.50 (19.00–25.00)	−0.13 (2.49)	.87

Note: Full possible range of scores for pain knowledge: 0–100; and coping: 7–35.

Abbreviation: IQR, interquartile range.

No significant differences were observed regarding pre–post intervention coping scores (see Table 2). An exploratory item-level comparison for pain coping items revealed trends in increased pain coping self-efficacy from pre- to post-session for four out of seven coping behaviors. Patients endorsed feeling “pretty sure” or “very sure” they could make it through school (16% at presession to 25% at postsession), take care of themselves (25% at presession to 41% at postsession), and do activities with their family (24% at presession to 34% at postsession) after session 1, and do well in school following session 2 (5% at presession to 17% at postsession) and session 3 (0% at presession to 25% at postsession). Coping self-efficacy regarding being with friends, doing household chores, or doing homework remained relatively similar (i.e., <6% difference) from pre- to post-intervention across all sessions.

4 ∣. DISCUSSION

Recent clinical guidelines for the management of SCD pain recommend integrating nonpharmacological therapies, such as cognitive and behavioral pain management strategies, to support comprehensive disease and pain management plans.³ CAP for SCP, a culturally informed, patient-centered intervention, was flexibly adapted to a three-session inpatient group intervention format using evidenced-based strategies to support implementation and help address the needs of a large urban pediatric SCD program. Although the overall feasibility of conducting inpatient group sessions was limited by system-level barriers, CAP for SCP sessions were acceptable to patients and multidisciplinary providers, with evidence of preliminary effectiveness toward improving patient knowledge of pain management strategies.

Integrating CAP for SCP as an intervention during hospitalizations for pain offers several potential benefits. Group treatment sessions can offer effective resource allocation of provider time and services that allows one to treat a number of patients simultaneously in a more time- and cost-effective manner.¹² This may be beneficial for SCD programs that have few multidisciplinary mental health providers. From a patient-centered perspective, participation in a group treatment for pain management can serve as an opportunity for physical and behavioral activation to support out of bed movement and activity, which can be difficult for youth experiencing acute or chronic SCD pain. Group treatment provides built-in opportunities for socialization with peers who also have SCD, which can reduce feelings of isolation and stigma often experienced by adolescents with SCD during hospitalizations and among individuals with chronic pain.^17,18 Granted, social engagement embedded within a group format may not be of interest to all children and adolescents. For example, some adolescents shared concerns about joining a group session due to their physical appearance in the presence of others (e.g., not dressed properly, without makeup). Nevertheless, the benefits of these groups were reported across stakeholders, including patients, providers, and medical team members.

Additional known barriers can interfere with patients receiving and engaging in behavioral pain interventions, such as time, provider availability, financial costs, and stigma related to mental health care.^3,19 For this project, the feasibility of conducting CAP for SCP inpatient group sessions was primarily limited by system-level factors, such as patient and provider time and availability. For example, competing clinical demands or emergent consultations often limited the psychology providers’ availability to offer groups, whereas patient engagement in routine clinical care or sleepiness often interfered with patient participation. When patient and provider availability aligned, the overall feasibility of completing sessions approached 80%; however, our 35% completion rate of sessions specifically in a group format illustrates the common obstacles that accompany behavioral group interventions for pain, such as high attrition rates across group therapy sessions.^20,21 It is important to consider that meta-analyses indicate psychological treatments whether offered in an individual or group format demonstrate comparable efficacy.¹² As such, conducting CAP for SCP sessions in both individual and group formats may offer greater flexibility to meet patient needs and interests without hindering intervention fidelity or efficacy.

Valuable lessons were learned during the implementation of this project that can inform future endeavors of implementing CAP for SCP at other children’s hospitals and settings. The benefits of consistent multidisciplinary partnership emerged as a key indicator of patient buy-in and participation. Consistent provision and introduction of session flyers during family-centered rounds were likely closely related to patient participation, given the critical role that medical team communication and endorsement can have on patient adherence to pain management strategies.²² Although interventionists introduced the program in the cases where it was not introduced by the medical team, it is possible that patients and families were less inclined to participate or adhere to treatment recommendations without a familiar and trusted medical provider endorsing participation.²³ This may have been especially pronounced given the stigma associated with mental health services and providers who were leading the groups and for patients for whom CAP for SCP was their first encounter with a mental health provider.²⁴ Therefore, strategies targeting continued and consistent partnership with the inpatient team providers to facilitate multidisciplinary and nonpharmacological pain management strategies may strengthen patient buy-in and participation to optimize sustained engagement over time.

Remaining flexible and responsive to patient and team needs also emerged as important to support implementation of the program. For example, conducting intervention sessions required availability of at least two psychology providers (i.e., one to conduct group sessions and another to concurrently provide consultation service coverage) for an uninterrupted block of time. Adjusting the pace of content delivery to ensure patient comprehension and having flexibility to extend beyond the time allotted to complete intervention sessions were essential to remain responsive to individual patient needs, especially within a group setting. Notably, these barriers can be overcome with intentional planning and resources, such as flexible scheduling during times of day when there are fewer clinical care demands. Accommodations of conducting the intervention individually at bedside helped increase participation for those patients who otherwise may have refused to participate due to concerns of leaving their room. Lastly, there was a clear need to remain responsive to the variability in patient literacy. Many patients struggled with the reading level of materials. To help with time flow, avoid embarrassment, or minimize lack of true understanding of survey questions, interventionists often read aloud all materials. This feedback was shared with the intervention developer, resulting in a revision of all program materials to reduce reading complexity and improve access for English Language Learners (ELL) and patients with variable levels of health literacy.

Alternative methods for implementation to fit the needs and structure of a SCD program or hospital setting may also support enhanced feasibility of CAP for SCP across settings. Training alternative professional disciplines (e.g., nurse practitioners, child life specialists, licensed counselors, psychiatry providers, or clinical social workers) to deliver pain management interventions or developing a training opportunity for learners may increase feasibility and access for patients. Nurse practitioner- and physical therapist-delivered cognitive and behavioral pain coping skills training was feasible and efficacious among adult chronic pain populations^25,26 and warrants further study in pediatrics. In an outpatient setting, specific treatment components could be incorporated into routine SCD clinic visits such that education and skills training is integrated early and often into ongoing medical care to support preventative pain care. Alternatively, structured group sessions could be planned in conjunction with routine clinic visits, or evening/weekend group sessions may offer increased accessibility and reduce attrition. The flexible administration platform of this structured intervention means that clinicians and institutions have the opportunity to fold this behavioral pain management treatment into an existing care pathway or generate a novel touchpoint in a child’s SCD treatment to include this intervention. Of note, in the context of COVID-19, CAP for SCP has also been administered virtually in both a one-on-one and small group setting, with high treatment satisfaction. Hybrid models of administration where a child may initiate the program during an inpatient stay or outpatient appointment and finish the program with virtual support, may also be possible.

Results of this project must be interpreted within the context of its limitations. Primarily, only a small subset of patients completed all three sessions, thereby limiting the conclusions that can be drawn from the fully delivered intervention. Based on prior work evaluating the CAP intervention, it is likely that the full intervention may have contributed to more robust improvements in pain knowledge and coping efficacy.^6,27 Although the extent to which pain knowledge may translate to behavior change to improve disease management likely depends on how much youth are responsible for their own SCD management; some research suggests that the association between knowledge and behavior change was greater for those with high coping efficacy.^28,29 Thus, future studies evaluating the potential impact of CAP for SCP on both immediate (e.g., knowledge, coping efficacy) and longer term patient outcomes (e.g., physical and emotional functioning, self-management) are needed. Possible confounders, such as medication side effects (e.g., drowsiness, fatigue), pain intensity, or cognitive or attentional factors may have interfered with patient participation, engagement, and comprehension. Reasons for nonparticipation were not routinely collected, thereby limiting the evaluation of potential systematic barriers to participation (e.g., age, SCD genotype). The overall sample size limited statistical power in detecting pre- to post-session effects and restricted additional analysis on potential intervention dose effects. Future evaluations of CAP for SCP, using an individual or group format, are needed to further support the intervention efficacy on improving psychosocial functioning and quality of life in a large clinical trial as well as generalization and implementation across various SCD programs. Future studies may also consider a cost–benefit analysis to assess the potential merits of individual versus group CBT approaches for pain management. The DAP model (Figure 1) can also serve as a framework for future iterative improvement, learning from each stage of the implementation cycle and improving upon its application in different settings.

In summary, the CAP for SCP intervention is a patient-centered intervention that includes first-line cognitive and behavioral skills and strategies to support SCD pain management. Although implementation of CAP for SCP within an inpatient group setting may come with additional system-wide challenges, the high levels of patient acceptability and preliminary improvements in knowledge of pain management strategies highlight the benefits of integrating a psychoeducational intervention into clinical practice settings to support pain management among children and adolescents with SCD.

Abbreviations:

CAP

Comfort Ability Program

CBT

cognitive-behavioral therapy

DAP

Dynamic Adaptation Process

EMR

electronic medical record

Hb

hemoglobin

IRB

Institutional Review Board

M

mean

PI

primary investigator

SCD

sickle cell disease

SCP

Sickle Cell Pain

SD

standard deviation

DATA AVAILABILITY STATEMENT

The data that support the findings of this study are available from the corresponding author upon reasonable request.