A Novel Combined Resilience and Advance Care Planning Intervention for Adolescents and Young Adults With Advanced Cancer: A Feasibility and Acceptability Cohort Study

Abstract

BACKGROUND:

Few evidence-based psychosocial programs have been tested among adolescents and young adults (AYAs) with advanced cancer (AC), and early advance care planning (ACP) in this population is rare. The authors aimed to determine the feasibility and acceptability of 1) delivering an established resilience-coaching program, and 2) integrating ACP into that program, among AYAs with AC.

METHODS:

Eligible AYAs were 12 to 24 years old, diagnosed with advanced cancer (recurrent/refractory disease or a diagnosis associated with <50% survival) and fluent in English. The Promoting Resilience in Stress Management–Advanced Cancer (PRISM-AC) program included PRISM’s standard sessions targeting stress-management, goal-setting, cognitive-restructuring, and meaning-making, delivered 1:1, 1 to 2 weeks apart, plus a new session involving elements of the AYA-specific Voicing My Choices ACP guide. Participants completed surveys at baseline and 12 weeks, and exit interviews following study completion. Feasibility was defined as ≥70% completion of 1) standard 4-session PRISM and 2) the new ACP session among those completing standard PRISM. Acceptability was defined qualitatively. Trajectories of patient-reported anxiety, depression, and hope were examined descriptively.

RESULTS:

Of 50 eligible, approached AYAs, 26 (52%) enrolled and completed baseline surveys. The AYAs had a mean age of 16 years (SD = 2.7 years), and the majority were male (73%) and White/Caucasian (62%). Twenty-two AYAs (85%) completed standard PRISM, and of those, 18 (82%) completed the ACP session. Feedback was highly positive; 100% and 91% described the overall and ACP programs as valuable, respectively. Anxiety, depression, and hope were unchanged after the program.

CONCLUSIONS:

Resilience coaching followed by integrated ACP is feasible and acceptable for AYAs with AC. Participating did not cause distress or decrease hope.

LAY SUMMARY:

• Advance care planning (ACP) among adolescents and young adults (AYAs) with advanced cancer can be difficult to introduce.

• We investigated whether it is feasible and acceptable to integrate ACP into an existing resilience-coaching program for AYAs.

• In this cohort study of 26 AYAs with advanced cancer, we found the Promoting Resilience in Stress Management–Advanced Cancer program to be feasible (≥70% intervention-completion) and highly acceptable (positive post-participation feedback, no evidence of participant-distress).

• We conclude that an intervention integrating resilience coaching and ACP is feasible and acceptable among AYAs with advanced cancer.

INTRODUCTION

Adolescents and young adults (AYAs; 12–24 years of age) with cancer are at high risk of poor psychosocial outcomes, because cancer disrupts critical developmental experiences occurring at this age.^1,2 AYAs with advanced cancer (AC) have unique unmet needs that may further contribute to poor outcomes, including inadequate psychosocial and communication support despite facing challenges associated with prognostic uncertainty and missed goals.³ This group understands death may be a consequence of their disease, and most report it would be helpful, if not imperative, to discuss wishes, worries, and information with friends and family.^4,5 Only half engage in these discussions and, when they occur, discussions may be held too late.^5,6 Missed opportunities to identify and articulate hopes, worries, and corresponding treatment decisions contribute to patient suffering, parent distress, and decisional regret.^7–9 Despite guidelines for integrated psychosocial and communication support,^10,11 barriers remain, including a lack of evidence-based, AYA-specific interventions.¹²

The Promoting Resilience in Stress Management (PRISM) intervention¹³ has the potential to improve the psychosocial well-being of AYAs with advanced disease. Informed by stress-and-coping theories and designed to be delivered in a brief, cost-efficient manner, PRISM aims to improve self-perceived resilience through teaching individual and existential resilience skills including stress-management, goal-setting, cognitive-reframing, and meaning-making.^13,14 In a phase 2 randomized-controlled trial of AYAs with cancer, PRISM was feasible, highly valued, and efficacious in improving AYA resilience and quality of life and minimizing distress.^14,15 A limitation of that study, however, was that it focused on AYAs with newly diagnosed cancer; we were unable to fully assess PRISM’s feasibility and acceptability among AYAs with progressive or refractory disease.^14,16 The few AYAs who enrolled with AC reported additional unmet needs they thought PRISM could help them meet. Specifically, they shared that PRISM had helped them to identify their values, legacies, identities, and goals, and they now wanted an opportunity to share those thoughts with loved ones.

The current study had 3 goals: 1) to determine the feasibility and acceptability of the original PRISM program among AYAs with AC; 2) to explore the feasibility and acceptability of an additional PRISM-component designed to facilitate advance care planning; and 3) to describe trajectories of patient-reported anxiety, depression, and hope pre- to post-intervention to a) inform future trials and b) ensure the program did not increase distress or minimize hope.

MATERIALS AND METHODS

Participants and Procedure

We recruited participants from Seattle Children’s Cancer and Blood Disorders Center. All procedures were approved by the local institutional review board. AYAs were eligible if they were 12 to 24 years old, able to speak and read English (including as a second language), and diagnosed with advanced cancer at least 2 weeks before enrollment. “Advanced cancer” definitions were adapted from the Center for the Advancement of Palliative Care and guidelines to optimize early palliative care in pediatric oncology: “progressive, recurrent, or refractory disease, or any diagnosis with an estimated survival <50%.”^17,18 Individuals with cognitive impairments and those who previously received PRISM were excluded.

Consecutive AYAs were recruited over 7 months (September, 2018 to April, 2019). Clinic rosters were screened and eligibility confirmed through medical record review.^14,15 AYAs (and their parents, if under 18) were approached in clinic or during an inpatient stay. Those who provided informed consent or assent with corresponding parent consent (12–17 years old) were offered the PRISM–Advanced Cancer (PRISM-AC) program. At enrollment and 12 weeks post-enrollment, participants completed a demographic survey and patient-reported outcome instruments. They received a $25 gift card at each time point. Following completion of PRISM-AC, participants were asked to complete a 1:1 follow-up interview. Two female bachelor’s level research assistants conducted interviews; 1 had minor prior contact with participants related to other study activities (eg, administering surveys) and the other had no prior contact. Interviews were conducted in-person or via phone, audio-recorded, and lasted approximately 20 minutes. Recordings were transcribed verbatim by 2 additional trained research assistants.

Intervention Development and Delivery

PRISM is a scripted, skills-based coaching program comprised of 4 core sessions targeting “resilience resources” (stress-management, goal-setting, cognitive-reframing, and meaning-making) followed by an optional family meeting where AYAs can share what they learned with family and loved ones.^13–15 Sessions are conducted 1:1 and last approximately 30 to 50 minutes. We expanded PRISM’s content for AYAs with AC based on input from AYA stakeholders who expressed interest in opportunities to articulate goals and meaning from their cancer experience. The new PRISM component was designed to introduce simple, evidence-based, and age-appropriate advance care planning exercises using the validated AYA-specific Voicing my Choices Advance Care Planning Tool (https://agingwithdignity.org).^19,20

PRISM-AC thus included 6 coaching sessions: the standard PRISM sessions 1 through 4,^13,14 a new 5th session using Voicing My Choices, and an optional family meeting (Supporting Table 1). During the new 5th session, AYAs were guided through up to 4 select pages from Voicing My Choices: page 4, “My Comfort,” page 5, “My Support,” page 8, “My Friends and Family to Know,” and page 9, “My Spiritual Thoughts.” These pages reflect PRISM skills and were vetted by AYAs, parents, medical, and psychosocial clinicians as most relevant to the program. To ensure that participants were prepared for these topics, coaches followed standard pediatric oncology practice and initiated a brief conversation with either the participant (for AYAs 18+) or privately with a parent (for AYAs <18) to introduce Voicing My Choices. When AYAs or parents expressed concerns or raised questions, coaches wrote memos in the study chart to document the discussion; if no question/ concerns were raised, no memos were written.

AYAs and/or parents were invited to review the Voicing My Choices guide and choose whether they and/or their child would receive the full version (including pages that would not be used in the session) or only the 4 select pages. Coaches recorded which version was selected. All AYAs kept the selected booklet and/or pages following the session and were encouraged to continue exercises on their own, with family, or medical staff.

PRISM-AC was delivered by 4 coaches, each with a bachelor’s degree or an advanced degree in psychology, social work, or public health. They completed >8 hours of training including supervised role-playing and practice scenarios. Sessions were delivered 1:1, audio-recorded, and monitored for fidelity by a clinical psychologist; for each coach, the first 5 and then 1 of 5 randomly selected subsequent sessions were scored with a standardized tool.¹³ In this study, all sessions were delivered with 100% fidelity.

In-person delivery was prioritized; phone or videoconference was available if needed to address logistical constraints (eg, participant returned home). Sessions were scheduled 1 to 2 weeks apart; if a session was missed, coaches attempted to reschedule for up to 3 months.

Measures

Demographic and medical information

Demographic information (gender [male/female/other (fill-in-the-blank)], race, and ethnicity) was collected via survey. Medical information (diagnosis, diagnosis date) and patient ages were extracted from electronic health records.

Patient-reported outcomes

Hope was measured with the Snyder Hope Scale.²¹ This measure contains 8 core items scored on an 8-point Likert scale that assess overall perception that one’s goals can be met, with higher scores indicating greater hope.²¹ Anxiety and depression symptoms were measured with the Hospital Anxiety and Depression Scale.²² Seven questions assess anxiety symptoms and 7 assess depression symptoms. Each item is scored from 0 to 3, for a total range of 0 to 21 points per subscale.

Data Analysis

Feasibility and acceptability of the original PRISM program

First, we assessed the feasibility and acceptability of the original PRISM program among AYAs with advanced cancer. Feasibility was defined as ≥70% of participants completing sessions 1 through 4. Acceptability was assessed qualitatively; participants who finished all sessions (including session 5) or had not received a session in >3 months were approached for feedback interviews. Those who agreed were first asked whether and/or how the core PRISM (sessions 1–4) was helpful and, “Should everyone who has cancer get PRISM?” Feedback was summarized to inform whether further refinement was needed. Qualitative methods and presentation of results followed the Standards for Reporting Qualitative Research.²³ Three coders (K.M.F., M.B.O., and K.S.B.) conducted directed content analysis in Dedoose.²⁴ First, we developed an a priori codebook based on questions from the interview guide. Next, each coder independently coded 4 interviews, and additional emergent codes were added. Once the codebook was finalized, the primary coder (K.M.F.) and secondary coder (M.B.O.) sequentially coded each transcript. When discrepancies occurred, the third coder (K.S.B.) analyzed relevant text to reach consensus. We tallied the total number of participants reporting in each code.

Feasibility and acceptability of session 5 using Voicing My Choices

Next, we explored the feasibility and acceptability of a 5th session focused on early advance care planning using Voicing My Choices. Feasibility was defined as ≥70% of active participants (those who completed PRISM 1–4) completing session 5. Following methods described above, acceptability was assessed via post-participation feedback, this time focused specifically on this session. To explore whether parent- or AYA-reactions to Voicing My Choices were barriers to delivery, we assessed the proportion of AYAs and/or parents who selected each version of the guide (full or abbreviated) and reviewed conversation memos for any notable patterns.

Patient-reported outcomes

This pilot study was not designed or powered to examine intervention efficacy. To inform future clinical trials, we summarized patient-reported outcomes descriptively at each time point for the full sample and for those who completed the new 5th session. Among AYAs for whom both baseline and follow-up scores were available, we calculated change scores.

RESULTS

Sample Characterstics

Sixty-one patients were eligible over the study period. Of these, 50 were approached, 30 (60%) enrolled, and 26 (52% of those approached, 87% of those enrolled) completed baseline surveys (Fig. 1). There were no detectable demographic differences between AYAs who enrolled and declined; however, those who enrolled had been diagnosed with cancer for more time than those who declined (4.9 vs 2.9 years). Participants were mean-aged 16.0 years (SD = 2.7; range, 12–22), majority 12 to 17 years old (65%), male (73%), White (62%), non-Hispanic (89%), and spoke English as a first language (89%) (Table 1). The majority were diagnosed with a brain tumor (35%) or sarcoma (31%) and had histories of progressive and/or refractory disease (58%).

Figure 1.

Recruitment and completion of the Promoting Resilience in Stress Management for Patients With Advanced Cancer intervention. LTFU indicates lost to follow-up; PRISM, Promoting Resilience in Stress Management; VMC, Voicing My Choices.

TABLE 1.

Participants’ Demographic Information and Instrument Scores by Study Component Completion

Characteristic	Completed Baseline (n = 26)	Completed PRISM 1–4 (n = 22)	Completed PRISM 1–5 (n = 18)
Age at enrollment, y, No. (%)
12–17	17 (65)	14 (64)	12 (67)
18–24	9 (35)	8 (36)	6 (33)
Gender
Male	19 (73)	15 (68)	12 (67)
Female	7 (27)	7 (32)	6 (33)
First language, No. (%)
English	23 (89)	19 (86)	16 (89)
Other	3 (11)	3 (14)	2 (11)
Race, No. (%)
White	16 (62)	14 (64)	13 (72)
Black/African American	1 (4)	1 (5)	1 (6)
Asian	2 (8)	2 (9)	2 (11)
American Indian, Alaska Native	1 (4)	1 (5)	0 (0)
Native Hawaiian, Pacific Islander	1 (4)	0 (0)	0 (0)
Mixed	5 (19)	4 (18)	2 (11)
Ethnicity, No. (%)
Hispanic	3 (11)	3 (14)	2 (11)
Non-Hispanic	23 (89)	19 (86)	16 (89)
Diagnosis, No. (%)
Acute lymphoblastic leukemia	7 (27)	6 (27)	3 (17)
Central nervous system	9 (35)	7 (32)	7 (39)
Non-Hodgkin’s lymphoma	2 (8)	1 (5)	1 (6)
Sarcoma	8 (31)	8 (36)	7 (39)
Time since diagnosis, median (IQR)
Years since initial diagnosis	4.9 (1.2–6.4)	4.9 (1.2–6.4)	4.8 (1.2–6.5)
Initial vs recurrent cancer, No. (%)
Initial	11 (42)	10 (45)	9 (50)
Recurrent	15 (58)	12 (55)	9 (5)
Patient-reported outcomes at baseline, mean (SD)
Anxiety	6 (4)	6 (4)	6 (4)
Depression	4 (3)	4 (3)	4 (3)
Hope	48 (11)	48 (12)	48 (11)

Abbreviations: IQR, interquartile range; PRISM, Promoting Resilience in Stress Management.

Feasibility and Acceptability of PRISM for AYAs with AC

Twenty-two participants (73% of 30 enrolled, 85% of 26 who completed baseline surveys) completed the core PRISM program. The median number of days between sessions was 14.5 (interquartile range [IQR] = 9–27 days), and time from enrollment to completion of sessions 1 through 4 was 67.5 days (IQR = 51–95 days). The majority of sessions were delivered in person (61%) during an inpatient hospitalization or outpatient clinic visit, followed by phone (36%) and videoconference (3%). Eight (27%) participants stopped study-participation before completing session 4, including 4 who died or became too ill (Fig. 1).

Twenty-three participants were eligible for feedback interviews. Of these, 11 (48%) were available (not deceased or lost-to-follow-up) (Supporting Table 2); all had completed sessions 1 through 4 before their interview. All (11 of 11) agreed that PRISM should be offered to all patients. Most (10 of 11) found PRISM helpful. Many discussed the value of specific skills, how PRISM helped them deal with negative emotions, and how they appreciated having someone listen. Nearly half (5 of 11) suggested that skills could be focused on coping with advanced cancer, specifically.

Feasibility and Acceptability of PRISM Session 5 Using Voicing My Choices

Conversations to introduce the Voicing My Choices guide were held with all 22 PRISM-recipients who completed Session 4, including 8 young adults ≥18 years-old and 14 parents of adolescents <18. All 8 young adults selected the full guide. No memos were recorded from conversations with young adults, indicating that no debate or ambiguity occurred. Among parents, 57% (8 of 14) opted for the abbreviated and 29% (4 of 14) for the full guide. Choices from 2 parents were not recorded due to researcher error. Memos were recorded for the 7 parent-conversations with ambiguity or debate. Three parents expressed concern about portions of the full guide that explicitly mentioned death or dying (eg, funeral planning) and after discussion were comfortable with the abbreviated guide. Two parents wanted their child to receive the abbreviated guide and requested a copy of the full guide as a resource “…if it ever got to that point”; of these, 1 became tearful and ultimately endorsed the topic’s importance. Two parents wanted to talk to their child about the session content before scheduling.

In total, 18 AYAs (82% of 22 who completed sessions 1–4, 70% of 26 who completed session 1, and 78% of 23 who received session 1 and did not die or become too sick during the study) completed session 5. The median time from enrollment to completion of session 5 was 88.5 days (IQR = 65–103 days).

Ten of the 11 AYAs who completed interviews had received session 5 before their interview. Nearly all interviewed AYAs (9 of 10) said the session was valuable (Supporting Table 2). They appreciated receiving the guide, felt the session helped them be more independent and assertive in their care, helped put their thoughts into words, and assisted in sharing their thoughts and feelings with family members. Most AYAs (7 of 10) recognized the program’s value while also acknowledging it was emotionally difficult; 2 (ages 15 and 19) specifically disliked the question about where they would want to die.

Exploratory Analyses

We first calculated change-scores for all AYAs who completed both baseline and follow-up surveys (n = 19), regardless of session completion. PROs did not change substantially from baseline to 12-week follow-up (Fig. 2). We observed a slight decrease in anxiety (M_Δ = −1.0, SD = 4.5) and slight increase in hope (M_Δ = 1.1, SD = 5.9) and depression (M_Δ = 1.0, SD = 3.0). We next evaluated data for AYAs who had completed all sessions, including Voicing My Choices, before their 12-week survey (n = 9) (Fig. 2). We observed a decrease in anxiety (M_Δ = −2.6, SD = 4.2), an increase in hope (M_Δ = 2.1, SD = 6.3), and a slight increase in depression (M_Δ = 0.7, SD = 3.4).

Figure 2.

Change in patient-reported outcomes from baseline to 12-week follow-up. Mean change in patient-reported outcomes from baseline to 12-week follow-up with 95% confidence interval. The top row shows adolescent and young adult patients who completed all sessions, including the Voicing My Choices advance care planning tool before survey collection. The bottom row shows all study participants who completed surveys at both time points, regardless of session completion. Anxiety and depression symptoms were measured with the Hospital Anxiety and Depression Scale²²; hope was measured with the Snyder Hope Scale.²¹

DISCUSSION

Despite recommendations for integrated psychosocial and communication support for AYAs with AC, few evidenced-based programs exist, and few AYAs with AC have opportunities to participate in formal ACP.^4,10,12,20 We found that 1) delivering an evidence-based resilience coaching program (PRISM), and 2) adding a brief module as an introduction to ACP was feasible in this population. Qualitative comments were largely positive and similar to those observed in other PRISM studies,^13,25,26 with AYAs describing PRISM as valuable for coping with stress, medical procedures, and negative emotions. Consistent with other studies of ACP among AYAs,^7,20 most AYAs responded positively to the added session.

These results align with other evidence indicating that AYAs and parents are open to ACP discussions.^7,19,20 Our findings suggest a novel way to introduce these topics. Specifically, by integrating the concepts as part of a more generalized psychosocial supportive care program, we were able to engage both AYAs and parents in discussions they may not otherwise have had. More, we identified strategies to help parents prepare for these discussions, namely, by providing them opportunities to review Voicing My Choices content beforehand and offering abbreviated options. This is important because well-intentioned parents may delay advance care planning because they worry it will upset their child. In our cohort, although some parents were concerned about topics in Voicing My Choices (eg, funeral planning) and wanted time to think or talk to their child, all (100%) ultimately agreed for their child to at least receive the abbreviated guide. Notably, although over half of parents preferred their adolescent (<18 years) receive the abbreviated guide, all young adults (18+ years) chose the full guide for themselves. Although we cannot determine whether adolescents participants would have similarly chosen the full guide for themselves, this trend aligns with prior studies demonstrating that AYAs want to be involved in decision-making.²⁷

Generally, enrollment in psychosocial and supportive care intervention-research is low among AYAs^28,29 and older adults with advanced cancer.^30,31 Our overall enrollment rate of 60% (and survey completion rate of 52%) suggests AYAs are willing to participate in programs like PRISM-AC. However, we also found that illness complications and logistical contraints led to longer-than-anticipated completion timelines and participant attrition. This suggests that delivering psychosocial supportive care to AYAs with advance illness requires flexible timelines and delivery strategies, such as truncating timelines when patients are well and maintaining regular contact with families to promote continued engagement.

This study was not designed to assess efficacy; rather, we endeavored to collect pilot data to ensure that depression, anxiety, and hope were not worsening. We found that all 3 constructs were relatively stable—with or without the ACP session. Similar trends have been observed in other studies with parents and patients, demonstrating that honest prognostic communication fosters hope and may not prevent concurrent distress.^32,33 Here, we cannot determine whether this stability is due to the intervention or other causes (eg, other psychosocial services).

Findings should be considered in light of study limiations. First, our sample was limited to English speakers and the majority were White/Caucasian and male. It was also too small to detect significant demographic- (age, sex, race, or ethnicity) or illness-related differences in willingness to enroll or intervention-completion. It is possible that findings would differ with larger and more diverse samples; we cannot describe how PRISM-AC would be received by diverse cultures. Second, although no participants opted out of session 5 or actively withdrew after being introduced to Voicing My Choices, 3 were lost to follow-up between sessions 4 and 5. We cannot conclude whether concern about the session influenced willingness to complete the study. Third, feedback interviews were completed by a subsample of participants who completed all or most PRISM-sessions. Those who withdrew sooner may have had less positive experiences, potentially biasing acceptability results. Finally, we did not collect “downstream” clinical outcomes such as AYA-engagement in later ACP, sharing of information with family and/or staff, or impact on long-term decision-making. We are now conducting a larger clinical trial to determine whether PRISM-AC is efficacious in improving quality of life for AYAs with AC and whether engaging in advance care planning exercises using Voicing My Choices increases patient-engagement in difficult conversations (NCT03668223).

In summary, the current study represents a first step toward developing and implementing evidence-based programs focused on psychosocial and communication support. AYAs with AC have unique developmental and psychosocial needs and desire to be active participants in their care. Providing support of these domains is critical to whole-patient care, with the potential to improve quality of life and reduce suffering for both AYAs with cancer and their families.