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To determine the safety of engaging HIV-positive (HIV+) adolescents in a Family Centered Advance Care (FACE) planning intervention.
We conducted a 2-armed, randomized controlled clinical trial in 2 hospital-based outpatient clinics from 2006–2008 with HIV+ adolescents and their surrogates (n = 76). Three 60–90 minutes sessions were conducted weekly. FACE intervention groups received: Lyon FCACP Survey©, the Respecting Choices® interview, and completion of The Five Wishes©. The Healthy Living Control (HLC) received: Developmental History, Healthy Tips, Future Planning (vocational, school or vocational rehabilitation). Three-month post-intervention outcomes were: completion of advance directive (Five Wishes©); psychological adjustment (Beck Depression, Anxiety Inventories); quality of life (PedsQL™); and HIV symptoms (General Health Self-Assessment).
Adolescents had a mean age, 16 years; 40% male; 92% African-American; 68% with perinatally acquired HIV, 29% had AIDS diagnosis. FACE participants completed advance directives more than controls, using time matched comparison (P < 0.001). Neither anxiety, nor depression, increased at clinically or statistically significant levels post-intervention. FACE adolescents maintained quality of life. FACE families perceived their adolescents as worsening in their school (P = 0.018) and emotional (P = 0.029) quality of life at 3 months, compared with controls.
Participating in advance care planning did not unduly distress HIV+ adolescents.
Most youth with HIV are now expected to live past 21 years,1,2 yet mortality rates are 30 times higher than for the general US pediatric population.3 In 2006, in a perinatally infected cohort the mean age of death was 18 years.4 Overwhelming infection due to immune system compromise contributes to morbidity.4 Thus, incorporating advance care planning during the “antecedent period of decision making”5 may be valuable in preparing for end-of-life (EOL) care. Quality advance care planning includes discussing death, but differs from advance directives6 (ie, documenting who you want to make health care decisions for you when you can’t make them for yourself, the kind of medical treatment you want or do not want). Concern about possible emotional distress has impeded families7,8 and providers from initiating conversations regarding EOL for teens living with a life-threatening condition.9 Yet, talking about death and dying and decision-making with such teens may benefit them and their families.10–12 Teens want involvement in their own EOL decisions.13 Guidelines11,14 recommend conversations about advance care planning: 1) begin early or at diagnosis of a life-threatening condition; 2) be shared among the adolescent, family and health care provider; and 3) be routine and structured. Goals for EOL care frequently are unfulfilled, partially, because providers are concerned that talking about death and dying can provoke negative moods in the teen,15 as well as families.11,12 Our goal was to develop/adapt a structured, safe intervention that would provide an opportunity to talk about death and dying while medically stable: planning for the worst, hoping for the best.
Guided by a community-based participatory research framework,16 the Family Centered Advance Care (FACE) planning development and proximal outcomes are reported elsewhere.17,18 This report presents the 3-month FACE post-intervention outcomes: plans and actions, psychological adjustment, and quality of life. The FACE intervention is based on Leventhal’s theory of self-regulation19–21 and Folkman and Lazarus’s theory of transactional stress and coping22 (See Figure 1), postulating that interventions can change the appraisal of an illness from a death threat to a challenge with potential for growth and mastery.
We hypothesized that at 3 months, post-intervention families in the FACE intervention study would be significantly more likely to: 1) complete an advance directive (AD); 2) maintain or have improved psychological adjustment; 3) maintain or have enhanced quality of life; and 4) maintain or decrease HIV symptom severity, compared to families in the Healthy Living Control (HLC) time-matched comparison.
Between July 1, 2006 and May 31, 2008, 40 HIV-infected adolescents and 40 adult surrogates (n = 80) were recruited from the Children’s National Medical Center (CNMC) and St. Jude Children’s Research Hospital (SJCRH) outpatient adolescent HIV specialty clinics. Recruitment criteria for adolescents included: aged 14–21 years, with an available legal guardian (if under 18 years) or adult surrogate at least 21 years old who knew the adolescent’s HIV status, not in foster care, no developmental delays, severe depression, suicidal or homicidal ideation, dementia or psychosis.23–26 Additional family members were permitted to participate, however, their data were excluded from analyses. The FACE study was Institutional Review Board approved at both institutions. All participants provided written assent/consent.
Full details of the study methods are reported elsewhere.17,18 In brief, three 60–90 minute semi-structured family interview sessions were conducted by a trained/certified interviewer at weekly intervals: Session 1: Lyon Advance Care Planning Survey© – Adolescent and Surrogate Versions: Session 2: The Respecting Choices Interview® Session 3: Completion of The Five Wishes.©
HLC subjects were also administered in three weekly 60–90 minute sessions and family format to control for time, attention and/or related Hawthorn effects. Session 1: Developmental History.27 Session 2: Safety Tips.28 Session 3: School and Career Planning interview.29
Five Wishes© (Towey and Aging with Dignity) is a legal document that facilitates expression of treatment preferences, if they were unable to communicate their wishes and includes the selection of a surrogate decision-maker. It can also serve as a tool to facilitate the participation of adolescents under age 18,30 but must be signed by their parent/legal guardian to be legally sufficient.
Statement of Treatment Preferences31 expresses values and goals related to future decision making regarding frequently occurring scenarios common to individuals with complications of AIDS.32 It was used to document specific treatment preferences of patients and their surrogates’ understanding of what the patient would want. Patients and surrogates chose one of three options, “to continue all treatment and keep fighting,” “to stop all treatment to prolong my life,” and “don’t know.”
Beck Anxiety Index (BAI)24 is a 21-item measure of anxiety rated on a 4-point Likert scale of symptoms over the past week. The BAI has demonstrated adequate reliability and validity to clinically assess anxiety in individuals aged 17 to 80. We extended downward to age 14 to allow for consistency of data collected by a single measure of anxiety.
Beck Depression Inventory-II25 is a 21-item scale self-report measure to assess presence of symptoms of depression over the past two weeks on a 4-point Likert scale for adolescents >13 years of age.
The Pediatric Quality of Life Inventory™ 4.033–35 is a 23-item modular measure of health-related quality of life in children and adolescents. Four dimensions of functional quality of life (physical, emotional, social, school) are assessed. It has the strongest norms, validity, and reliability of the measures available. Norms exist for up to age 18.9 years, which was extended upward to allow for one assessment measure across participants.
General Health Assessment for Children36 is a self-report measure for adolescents aged 12–20 years. The HIV-related symptom subscale was used to assess the degree of distress caused by HIV-related physical symptoms. It is comprised of 18 items rated on a six-point Likert scale (“Not at All” to “Extremely”).
Measures were administered separately to both the adolescent and surrogate, at baseline and at the 3-month post-intervention follow-up.
Stage of Illness was measured by the CDC (Centers for Disease Control and Prevention) classification system in place at the time of the study.37
Adolescent/surrogate dyads were randomly assigned to one of two study groups, FACE versus HLC. Randomization utilized permuted block design, based on study site, to ensure the same numbers would be recruited to FACE and HLC. All participants were given a brochure with information on advance care planning and received standard of care. Follow-up data were collected face-to-face at 3-months post-intervention with two exceptions. A research assistant, not the facilitator, administered follow-up questionnaires orally to control for literacy. Progress of participants through the trial is illustrated in Figure 2.
To assess whether the FACE intervention participants established an advance directive, medical charts were reviewed for documented completion.
Before we implemented parametric analyses, we evaluated the normality and variance homogeneity assumptions and when necessary implemented data transformations to ensure the data met these assumptions. Following analyses involving data transformations, the results were back transformed to return the estimates to their original scale and units. In this event, adjusted means are reported which also control for baseline levels of scores.
To evaluate changes in psychological adjustment and quality of life, ANCOVA (analysis of covariance) was used to estimate and compare group means, with 95% confidence intervals, by study group and assess the effect of the two groups. Each estimate was derived controlling for baseline anxiety and depression scores, as well as any covariates found in preliminary analyses to differ between groups. The slope coefficient for group was used to estimate the magnitude of any differences. A t-test was employed to assess statistical significance.
In secondary analyses, logistic regression models were used to test for a trend in the outcome based on the level of severity of illness using CDC criteria.37 It was unnecessary to code for intervention exposure, because all subjects attended 100% of Sessions 1–3. Estimates were derived separately for surrogate and adolescent. However, when possible, overall estimates were derived by treating the dyad as a cluster, again controlling for baseline levels, as well as any covariates found in preliminary analyses to differ between groups.
Randomization produced an acceptable balance respecting group assignment by sample characteristics at baseline.17 Of eligible families, 97% chose to participate. As planned, 40 dyads were randomized. However, two dyads were excluded from analyses due to withdrawal from the study prior to the start of Session 1. See Figure 2. Data analyses were based on 38 dyads, using an intent-to-treat design, which included the 2 misallocated dyads. The characteristics of these 38 adolescents are presented in Table 1. There were no adverse events.
FACE adolescents completed the Five Wishes© with their families at a higher rate than the HLC adolescents (90% versus 11%, P < 0.001; Table 2). At Baseline, one adolescent in the FACE group previously completed an AD and 5 were unsure whether they had. At 3-month post-intervention uncertainty was resolved. Five Wishes© completion did not significantly differ by gender, race, ethnicity, education, employment status, CDC stage of illness, number of guardians present, length of time with known diagnosis or age at diagnosis. However, teens whose surrogates were not biological family members (n = 8/10; 80%) were more likely to complete the Five Wishes© than those whose surrogates who were biological relatives (11/28; 40%, P = 0.031).
On the Statement of Treatment Preferences the majority of adolescents chose to continue all treatments, regardless of quality of life. Ten percent of adolescents in Situation 1, 26% in Situation 2, and 24% in Situation 3 chose to discontinue extraordinary treatment. FACE adolescents appeared to be no more likely than HLC adolescents to discontinue treatment across the three situations, but the absence of statistical significance in this small sample is not absence of an effect. See Table 2. Non-African-American adolescents were significantly more likely to choose discontinue treatment (P = 0.026). A trend emerged for non-African-American adolescents to be more likely to discontinue treatment under Situation 2 (P = 0.060), if they were to become physically disabled and require nursing home care. No associations reached significance between the three Situations by gender, education, employment status, age, length of time known diagnosis or age learned HIV status.
FACE adolescents who talked about their own prospective death and dying with their families in facilitated conversations by a trained facilitator were no more depressed or anxious than HLC adolescents at 3-months post-intervention (Table 3). Surrogates in the FACE group demonstrated slight increases in depressed and anxious mood compared to controls at 3-months post-intervention, controlling for baseline levels. However, increases were not clinically or statistically significant, but in minimal ranges and could be due to chance. Baseline levels of depression and anxiety predicted depression (adolescents, P = 0.002; surrogates, P < 0.001) and anxiety (adolescents, P = 0.003; surrogates P < 0.001) at 3-months post-intervention. Overall, adjusted mean levels of adolescent and surrogate depression and anxiety (Table 3) at 3-month post-intervention fell in the minimal clinical range. Depression and anxiety at 3-month post-intervention, controlling for baseline, did not differ significantly by gender, race, ethnicity, education, employment status, age at diagnosis, length of time with known diagnosis, family type or number of guardians present. Adolescents assigned to the intervention had statistically significantly higher depressed mood compared to controls at baseline, although mood levels were clinically insignificant in the minimal range.
Quality of life as measured by the PedsQL™ Total score was maintained for adolescents in both FACE and HLC and there were not significant differences by group or on any of the subscales (Table 4).
However, surrogates’ perception of their adolescents’ Total quality of life was significantly lower for surrogates in the FACE versus HLC group (P = 0.032) at 3-months, controlling for baseline levels. FACE surrogates rated their adolescents as having poorer school quality of life at 3-months post-intervention (66.9 versus 80.0, P = 0.011) and poorer emotion quality of life (74.8 versus 85.7, P = 0.029), compared to HLC surrogates. The physical and social subscale scores were not significantly different by group, controlling for baseline levels.
On the GHAC trends towards more HIV-specific symptoms emerged in FACE vs HLC adolescents for the following symptoms (See Table 5): rash or itching; fatigue or weakness; and trouble sleeping. However, the scores reflect very low levels of symptoms, if examined using the midpoint cut-off for reporting symptoms which distressed them, moderately, very much or extremely. There were no statistically significant differences between FACE and HLC teens for any other HIV related symptoms 3-months post-intervention. There also was no relationship between Treatment Preferences and symptom score (P = 0.39).
In secondary analysis using a regression model examining all adolescents regardless of treatment arm, those in CDC Category B (symptomatic, not AIDS) had lower physical scores (more symptoms) than those in Category A (asymptomatic) or C (AIDS) (P = 0.005) and they scored higher on Total Symptoms (P = 0.015).
Using a rigorous randomized 2-arm design and intent-to-treat analysis, the FACE intervention demonstrated that advance care planning can be safely administered to HIV+ teens, ie, did not cause significant emotional distress or adverse events. FACE prepared surrogate decision makers for their role in EOL discussions and treatment decisions.
Our study challenges earlier findings38–47 with 100% of our primarily African-American FACE group completing an advance directive. The fact that several families spontaneously requested an extra copy of the Five Wishes© to complete for themselves suggests this outcome was not a result of the Hawthorne Effect (desire to please the researcher). Our results are consistent with a report by Washington, who in a study of African-American primary caregivers who made decisions about EOL care, found strong support for hospice care.48
Participating in the FACE intervention did not influence decisions to discontinue treatment. Our goal was to safely assist adolescents with HIV/AIDS to make decisions with their families about their EOL preferences, while still cognitively intact.49–51 People sometimes change their AD as the disease progresses, for example, deciding to “Allow a Natural Death,” rather than to continue aggressive treatment(s). Thus, EOL care decision making is best under-stood as an ongoing process, rather than a one-time event, and should be revisited.52–57
Consistent with earlier adult studies44,45,58–61 African-American adolescents in our cohort preferred to continue all treatments at EOL at rates significantly higher than non-African-American adolescents, however, because of our small sample size this result may not be stable. This finding differs from that of Hinds and colleagues62 who reported race did not influence DNR (do not resuscitate) status in a pediatric oncology sample. This difference could be due to the timing of decision making, which in the Hinds study was during what Hansen5 calls the “central period” of decision making (where EOL is eminent), rather than the “antecedent period” as was done in our study. Our African-American families were willing to think and talk about EOL issues and were prepared for their role as decision-makers.17,18
FACE achieved rates of recruitment in excess of those reported in adults in which only 30%–47% of eligible patients participated in EOL studies.63–66 Nevertheless, 54% of potential participants approached prior to screening for eligibility, declined to participate. Dyadic/family studies pose a challenge as sometimes the patient wants to participate, but the family member does not have time, wants to protect the patient, or is uninterested.67
Several factors may explain the success of the FACE intervention. First, we integrated processes identified with successful HIV interventions,68–70 specifically, community-based participation in development and adaptation to ensure cultural sensitivity and to reduce health disparities.16,17,71,72 Second, we used a competency-based program to ensure consistency in the delivery of the intervention as measured by two components: 1) facilitator certification and 2) patient and family ratings of quality of communication, consistent with the recommendations of Dickens.72 Third, the high rate of retention suggests participants were highly motivated to seek education and improve long-term care. Fourth, FACE was highly family-centered. Finally, research assistants were hired and retained only if they committed to flexibility in scheduling sessions and if successfully certified (Respecting Choices® interview).
In contrast to Bakitas and colleagues,63 we did not find improvements in quality of life or mood for the intervention group, rather healthy levels of mood and quality of life were maintained. Floor effects for mood and ceiling effects for quality of life limited the power to detect differences, and differences that we found were within healthy ranges and therefore not clinically significant. The reason(s) higher levels of depressed mood were found at baseline for adolescents randomly assigned to FACE versus HLC are unclear. Baseline assessment occurred before randomization, so assignment to the intervention group did not impact mood.
Unexpectedly, FACE surrogates perceived their adolescents’ school and emotional quality of life was lower compared to HLC surrogates. This may reflect increased knowledge of school related and emotional problems for their teens that emerged during the facilitated conversation. Also, FACE surrogates reported more sadness and anxiety at 3-month post-intervention, than FACE adolescents who reported less compared to baseline levels. Although the changes were not clinically or statistically significant, they suggest that adolescents may have found talking about their own death and dying less anxiety provoking and sad than their families did. This finding needs further study.
The FACE results begin to fill the gap on ethnocultural factors that influence EOL decision making and preferences among HIV-positive adolescents, who demonstrated a willingness to discuss difficult and emotionally-laden issues with their surrogate decision makers and complete advance directives, potentially “breaking the ice” for future EOL conversations. Going beyond measuring the effect of communication on satisfaction, we studied outcomes related to advance care planning, quality of life and mood using standardized measures. The FACE results suggest that research into this sensitive area is safe. Probable benefits at the time of dying for teens and families, or after death for families, remain to be demonstrated in future studies.
We wish to thank our families for their participation and the community for their help in developing this protocol, especially Ebony Johnson. We also thank Connie Trexler and Keith Selden who facilitated the community advisory boards and focus groups. We extend our gratitude to our research assistants who worked with our families: Stephanie Lee, Portia Pieterse, Yolanda Peele, Ellin Kao, LaQuisha Mark, Mackenzie Nowell, Megan Banet, Megan L Wilkins, Ericka Midgett, J Christopher Young, Elizabeth Kolivas. We thank Jennifer Marsh and Saeid Goudarzi for help with statistical support and data management early in the study. We thank our consultants Drs Beatrice Krauss, Mary Ann McCabe, Bruce Rapkin and Robert Washington who helped during the developmental phase of the study and Dr Tomas Silber for his ethics consultations. We thank the health care providers and case managers who referred families to our study, especially Drs Hans Spiegel and Natella Rakhmanina.
The study was funded by grant 5R34MH072541-03 from the National Institute of Mental Health. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institute of Mental Health or the National Institutes of Health. This trial has been registered at www.clinicaltrials.gov Identifier #NCT00723476.
Linda Briggs, receives royalties from the Respecting Choices® interview. However, the remaining authors have no financial relationships or other conflicts of interest relevant to this article to disclose.