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Table 1.  Participant Characteristics
Participant Characteristics
Table 2.  Quality Domains and Subdomains With Relevant Interview Excerpts
Quality Domains and Subdomains With Relevant Interview Excerpts
Table 3.  Quality Domains, Descriptions, Potential Indicators, and Recommendations for Development
Quality Domains, Descriptions, Potential Indicators, and Recommendations for Development
1.
Bleyer  A.  The adolescent and young adult gap in cancer care and outcome.   Curr Probl Pediatr Adolesc Health Care. 2005;35(5):182-217. doi:10.1016/j.cppeds.2005.02.001 PubMedGoogle ScholarCrossref
2.
National Cancer Institute. Cancer Epidemiology in Older Adolescents and Young Adults 15 to 29 Years of Age Including SEER Incidence and Survival: 1975-2000. National Cancer Institute; 2006.
3.
National Cancer Policy Forum; Board on Health Care Services; A Livestrong and Institute of Medicine Workshop; Institute of Medicine. Identifying and Addressing the Needs of Adolescents and Young Adults With Cancer: Workshop Summary. National Academies Press (US); 2013.
4.
Nass  SJ, Beaupin  LK, Demark-Wahnefried  W,  et al.  Identifying and addressing the needs of adolescents and young adults with cancer: summary of an Institute of Medicine workshop.   Oncologist. 2015;20(2):186-195. doi:10.1634/theoncologist.2014-0265 PubMedGoogle ScholarCrossref
5.
Adolescent and Young Adult Oncology Progress Review Group.  Closing the Gap: Research and Care Imperatives for Adolescents and Young Adults With Cancer. National Institutes of Health and National Cancer Institute; 2006.
6.
Zebrack  B, Mathews-Bradshaw  B, Siegel  S; LIVESTRONG Young Adult Alliance.  Quality cancer care for adolescents and young adults: a position statement.   J Clin Oncol. 2010;28(32):4862-4867. doi:10.1200/JCO.2010.30.5417 PubMedGoogle ScholarCrossref
7.
Thomas  DM, Seymour  JF, O’Brien  T, Sawyer  SM, Ashley  DM.  Adolescent and young adult cancer: a revolution in evolution?   Intern Med J. 2006;36(5):302-307. doi:10.1111/j.1445-5994.2006.01062.x PubMedGoogle ScholarCrossref
8.
Albritton  KH, Wiggins  CH, Nelson  HE, Weeks  JC.  Site of oncologic specialty care for older adolescents in Utah.   J Clin Oncol. 2007;25(29):4616-4621. doi:10.1200/JCO.2006.08.4103 PubMedGoogle ScholarCrossref
9.
Howell  DL, Ward  KC, Austin  HD, Young  JL, Woods  WG.  Access to pediatric cancer care by age, race, and diagnosis, and outcomes of cancer treatment in pediatric and adolescent patients in the state of Georgia.   J Clin Oncol. 2007;25(29):4610-4615. doi:10.1200/JCO.2006.07.6992 PubMedGoogle ScholarCrossref
10.
Parsons  HM, Harlan  LC, Seibel  NL, Stevens  JL, Keegan  TH.  Clinical trial participation and time to treatment among adolescents and young adults with cancer: does age at diagnosis or insurance make a difference?   J Clin Oncol. 2011;29(30):4045-4053. doi:10.1200/JCO.2011.36.2954 PubMedGoogle ScholarCrossref
11.
Schnurr  C, Pippan  M, Stuetzer  H, Delank  KS, Michael  JW, Eysel  P.  Treatment delay of bone tumours, compilation of a sociodemographic risk profile: a retrospective study.   BMC Cancer. 2008;8:22. doi:10.1186/1471-2407-8-22 PubMedGoogle ScholarCrossref
12.
Pollock  BH, Krischer  JP, Vietti  TJ.  Interval between symptom onset and diagnosis of pediatric solid tumors.   J Pediatr. 1991;119(5):725-732. doi:10.1016/S0022-3476(05)80287-2 PubMedGoogle ScholarCrossref
13.
Parsons  HM, Schmidt  S, Harlan  LC,  et al; AYA HOPE Collaborative.  Young and uninsured: insurance patterns of recently diagnosed adolescent and young adult cancer survivors in the AYA HOPE study.   Cancer. 2014;120(15):2352-2360. doi:10.1002/cncr.28685 PubMedGoogle ScholarCrossref
14.
Robbins  AS, Lerro  CC, Barr  RD.  Insurance status and distant-stage disease at diagnosis among adolescent and young adult patients with cancer aged 15 to 39 years: National Cancer Data Base, 2004 through 2010.   Cancer. 2014;120(8):1212-1219. doi:10.1002/cncr.28568 PubMedGoogle ScholarCrossref
15.
Keegan  TH, Tao  L, DeRouen  MC,  et al; AYA HOPE Study Collaborative Group.  Medical care in adolescents and young adult cancer survivors: what are the biggest access-related barriers?   J Cancer Surviv. 2014;8(2):282-292. doi:10.1007/s11764-013-0332-4 PubMedGoogle ScholarCrossref
16.
Pritchard  S, Cuvelier  G, Harlos  M, Barr  R.  Palliative care in adolescents and young adults with cancer.   Cancer. 2011;117(10)(suppl):2323-2328. doi:10.1002/cncr.26044 PubMedGoogle Scholar
17.
Wein  S, Pery  S, Zer  A.  Role of palliative care in adolescent and young adult oncology.   J Clin Oncol. 2010;28(32):4819-4824. doi:10.1200/JCO.2009.22.4543 PubMedGoogle ScholarCrossref
18.
Hinds  PS, Drew  D, Oakes  LL,  et al.  End-of-life care preferences of pediatric patients with cancer.   J Clin Oncol. 2005;23(36):9146-9154. doi:10.1200/JCO.2005.10.538 PubMedGoogle ScholarCrossref
19.
Mack  JW, Chen  K, Boscoe  FP,  et al.  High intensity of end-of-life care among adolescent and young adult cancer patients in the New York State Medicaid program.   Med Care. 2015;53(12):1018-1026. doi:10.1097/MLR.0000000000000437 PubMedGoogle ScholarCrossref
20.
Mack  JW, Chen  LH, Cannavale  K, Sattayapiwat  O, Cooper  RM, Chao  CR.  End-of-Life care intensity among adolescent and young adult patients with cancer in Kaiser Permanente Southern California.   JAMA Oncol. 2015;1(5):592-600. doi:10.1001/jamaoncol.2015.1953 PubMedGoogle ScholarCrossref
21.
Dy  SM, Lorenz  KA, O’Neill  SM,  et al.  Cancer quality-ASSIST supportive oncology quality indicator set: feasibility, reliability, and validity testing.   Cancer. 2010;116(13):3267-3275. doi:10.1002/cncr.25109 PubMedGoogle ScholarCrossref
22.
Dy  SM, Kiley  KB, Ast  K,  et al.  Measuring what matters: top-ranked quality indicators for hospice and palliative care from the American Academy of Hospice and Palliative Medicine and Hospice and Palliative Nurses Association.   J Pain Symptom Manage. 2015;49(4):773-781. doi:10.1016/j.jpainsymman.2015.01.012 PubMedGoogle ScholarCrossref
23.
National Quality Forum. Palliative and End-of-Life Care Performance Measures. National Institutes of Health; 2016.
24.
Hanson  LC, Scheunemann  LP, Zimmerman  S, Rokoske  FS, Schenck  AP.  The PEACE project review of clinical instruments for hospice and palliative care.   J Palliat Med. 2010;13(10):1253-1260. doi:10.1089/jpm.2010.0194 PubMedGoogle ScholarCrossref
25.
Campion  FX, Larson  LR, Kadlubek  PJ, Earle  CC, Neuss  MN.  Advancing performance measurement in oncology: quality oncology practice initiative participation and quality outcomes.   J Oncol Pract. 2011;7(3)(suppl):31s-35s. doi:10.1200/JOP.2011.000313 PubMedGoogle Scholar
26.
Lorenz  KA, Rosenfeld  K, Wenger  N.  Quality indicators for palliative and end-of-life care in vulnerable elders.   J Am Geriatr Soc. 2007;55(suppl 2):S318-S326. doi:10.1111/j.1532-5415.2007.01338.x PubMedGoogle ScholarCrossref
27.
Levy MH, Back A, Bazargan S, et al; National Comprehensive Cancer Network. Palliative care. clinical practice guidelines in oncology.  J Natl Compr Canc Netw. 2006;4(8):776-818. doi:10.6004/jnccn.2006.0068PubMed
28.
Earle  CC, Landrum  MB, Souza  JM, Neville  BA, Weeks  JC, Ayanian  JZ.  Aggressiveness of cancer care near the end of life: is it a quality-of-care issue?   J Clin Oncol. 2008;26(23):3860-3866. doi:10.1200/JCO.2007.15.8253 PubMedGoogle ScholarCrossref
29.
Goodman  D, Morden  N, Chang  C, Fisher  E, Wennberg  J. Trends in cancer care near the end of life: a Dartmouth atlas of health care brief. September 4, 2013. Accessed July 13, 2021. http://www.medicalhomeoncology.org/UserFiles/Cancer_brief_090413.pdf
30.
Peppercorn  JM, Smith  TJ, Helft  PR, et al; American Society of Clinical Oncology. American Society of Clinical Oncology statement: toward individualized care for patients with advanced cancer.  J Clin Oncol. 2011;29(6):755-760. doi:10.1200/JCO.2010.33.1744PubMed
31.
Wright  AA, Mack  JW, Kritek  PA,  et al.  Influence of patients’ preferences and treatment site on cancer patients’ end-of-life care.   Cancer. 2010;116(19):4656-4663. doi:10.1002/cncr.25217PubMedGoogle ScholarCrossref
32.
Delivering High-Quality Cancer Care: Charting a New Course for a System in Crisis. Institute of Medicine; 2013.
33.
Tong  A, Sainsbury  P, Craig  J.  Consolidated criteria for reporting qualitative research (COREQ): a 32-item checklist for interviews and focus groups.   Int J Qual Health Care. 2007;19(6):349-357. doi:10.1093/intqhc/mzm042 PubMedGoogle ScholarCrossref
34.
Wiener  L, Zadeh  S, Battles  H,  et al.  Allowing adolescents and young adults to plan their end-of-life care.   Pediatrics. 2012;130(5):897-905. doi:10.1542/peds.2012-0663 PubMedGoogle ScholarCrossref
35.
Krippendorff  K.  Content Analysis: An Introduction to its Methodology. 2nd ed. SAGE Publications; 2004.
36.
Emerson  R, Fretz  RI, Shaw  RI.  Writing Ethnographic Fieldnotes. University of Chicago Press; 1995. doi:10.7208/chicago/9780226206851.001.0001
37.
Spiggle  S.  Measuring social values: a content analysis of Sunday comics and underground comix.   J Consum Res. 1986;13(1):100-113. doi:10.1086/209050 Google ScholarCrossref
38.
Kamihara  J, Nyborn  JA, Olcese  ME, Nickerson  T, Mack  JW.  Parental hope for children with advanced cancer.   Pediatrics. 2015;135(5):868-874. doi:10.1542/peds.2014-2855 PubMedGoogle ScholarCrossref
39.
Brand  SR, Fasciano  K, Mack  JW.  Communication preferences of pediatric cancer patients: talking about prognosis and their future life.   Support Care Cancer. 2017;25(3):769-774. doi:10.1007/s00520-016-3458-x PubMedGoogle ScholarCrossref
40.
Anil U, Elbuluk AM, Ziegler J, Schwarzkopf R, Long WJ. Hospital consumer assessment of healthcare providers and systems scores do not predict outcomes after total hip arthroplasty.  J Arthroplasty. 2018;33(2):337-339.E6. doi:10.1016/j.arth.2017.09.042
41.
Degner  LF, Sloan  JA.  Decision making during serious illness: what role do patients really want to play?   J Clin Epidemiol. 1992;45(9):941-950. doi:10.1016/0895-4356(92)90110-9 PubMedGoogle ScholarCrossref
42.
Mack  JW, Block  SD, Nilsson  M,  et al.  Measuring therapeutic alliance between oncologists and patients with advanced cancer: the Human Connection Scale.   Cancer. 2009;115(14):3302-3311. doi:10.1002/cncr.24360 PubMedGoogle ScholarCrossref
43.
Teno  JM, Gozalo  PL, Bynum  JP,  et al.  Change in end-of-life care for Medicare beneficiaries: site of death, place of care, and health care transitions in 2000, 2005, and 2009.   JAMA. 2013;309(5):470-477. doi:10.1001/jama.2012.207624 PubMedGoogle ScholarCrossref
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    Original Investigation
    Oncology
    August 23, 2021

    Patient, Family, and Clinician Perspectives on End-of-Life Care Quality Domains and Candidate Indicators for Adolescents and Young Adults With Cancer

    Author Affiliations
    • 1Division of Population Sciences, Dana-Farber Cancer Institute, Boston, Massachusetts
    • 2Department of Pediatric Oncology, Dana-Farber Cancer Institute, Boston, Massachusetts
    • 3Division of Research, Kaiser Permanente Northern California, Oakland
    • 4Department of Research and Evaluation, Kaiser Permanente Southern California, Pasadena
    • 5Brown Medical School, Providence, Rhode Island
    • 6Divisions of Pediatric Oncology and Palliative Care, Emory University and Aflac Cancer & Blood Disorders Center at Children’s Healthcare of Atlanta, Atlanta, Georgia
    • 7Department of Medical Oncology, Dana-Farber Cancer Institute, Boston, Massachusetts
    • 8Now with Agios Pharmaceuticals, Cambridge, Massachusetts
    • 9Lacuna Loft, Oakland, California
    • 10Department of Pediatric Oncology, Kaiser Permanente Southern California, Pasadena
    • 11Department of Psychosocial Oncology and Palliative Care, Dana-Farber Cancer Institute, Boston, Massachusetts
    • 12Division of Medical Oncology, Kaiser Permanente Northern California, Oakland
    • 13Department of Palliative Care, Kaiser Permanente Southern California, Pasadena
    • 14Psychosocial Support and Research Program, National Cancer Institute, Bethesda, Maryland
    JAMA Netw Open. 2021;4(8):e2121888. doi:10.1001/jamanetworkopen.2021.21888
    Key Points

    Question  What do adolescents and young adults with cancer, their families, and clinicians find important in end-of-life care?

    Findings  This qualitative study of interviews with 23 adolescents and young adults, 28 family caregivers, and 29 clinicians identified 7 distinct end-of-life priority domains: attention to physical symptoms, attention to quality of life, psychosocial and spiritual care, communication and decision-making, relationships with clinicians, care and treatment, and independence. Although some domains reflected existing quality domains for adults, some domains were unique or had distinct manifestations for this young population.

    Meaning  The priority domains identified in this study as relevant to adolescents and young adults with advanced cancer may be used for quality measurement.

    Abstract

    Importance  End-of-life care quality indicators specific to adolescents and young adults (AYAs) aged 12 to 39 years with cancer have not been developed.

    Objective  To identify priority domains for end-of-life care from the perspectives of AYAs, family caregivers, and clinicians, and to propose candidate quality indicators reflecting priorities.

    Design, Setting, and Participants  This qualitative study was conducted from December 6, 2018, to January 5, 2021, with no additional follow-up. In-depth interviews were conducted with patients, family caregivers, and clinicians and included a content analysis of resulting transcripts. A multidisciplinary advisory group translated priorities into proposed quality indicators. Interviews were conducted at the Dana-Farber Cancer Institute, Kaiser Permanente Northern California, Kaiser Permanente Southern California, and an AYA cancer support community (lacunaloft.org). Participants included 23 AYAs, 28 caregivers, and 29 clinicians.

    Exposure  Stage IV or recurrent cancer.

    Main Outcomes and Measures  Care priorities.

    Results  Interviews were conducted with 23 patients (mean [SD] age, 29.3 [7.3] years; 12 men [52%]; 18 White participants [78%]), 28 family caregivers (23 women [82%]; 14 White participants [50%]), and 29 clinicians (20 women [69%]; 13 White participants [45%]). Caregivers included 22 parents (79%), 5 spouses or partners (18%), and 1 other family member (4%); the 29 clinicians included 15 physicians (52%), 6 nurses or nurse practitioners (21%), and 8 social workers or psychologists (28%). Interviews identified 7 end-of-life priority domains: attention to physical symptoms, attention to quality of life, psychosocial and spiritual care, communication and decision-making, relationships with clinicians, care and treatment, and independence. Themes were consistent across the AYA age range and participant type. Although some domains were represented in quality indicators developed for adults, unique domains were identified, as well as AYA-specific manifestations of existing domains. For example, quality of life included global quality of life; attainment of life goals, legacy, and meaning; support of personal relationships; and normalcy. Within communication and decision-making, domains included communication early in the disease course, addressing prognosis and what to expect at the end of life, and opportunity for AYAs to hold desired roles in decision-making. Care and treatment domains relevant to cancer therapy, use of life-prolonging measures, and location of death emphasized the need for preference sensitivity rather than a standard path. This finding differs from existing adult indicators that propose that late-life chemotherapy, intensive measures, and hospital death should be rare.

    Conclusions and Relevance  The findings of this qualitative study suggest that AYAs with cancer have priorities for care at the end of life that are not fully encompassed in existing indicators for adults. Use of new indicators for this young population may better reflect patient- and family-centered experiences of quality care.

    Introduction

    More than 70 000 adolescent and young adult (AYA) patients with cancer are diagnosed in the US each year, and cancer is their leading disease-related cause of death.1,2 This population is at risk for inferior quality cancer care,1,3-15 in part because AYAs are at transitional phases in life and thus may have unique social, educational, employment, and family concerns beyond their treatment needs.4,5

    Adolescents and young adults may be especially vulnerable to care challenges as they face death. These deaths are a departure from the natural order of life in which each generation outlives the one before it.16 In contrast to feelings that their lives should be ahead of them, dying young people face profound losses, including the opportunity to find partners, nurture families and careers, and grow old.16,17 Adolescents and young adults near death also often recognize the pain they leave behind in surviving loved ones.18 All of these issues may contribute to heightened use of intensive measures at the end of life in this population.19,20

    To date, end-of-life care quality indicators specific to AYAs with cancer have not been developed, challenging our ability to ascertain and measure optimal care for AYAs approaching death. Existing quality indicators for adults with cancer have at times been used to understand end-of-life care among AYAs and have identified high rates of inpatient care, care in the intensive care unit or emergency department, and chemotherapy near death.19,20 However, we do not know whether these indicators, and other indicators developed for adults, speak to the values of AYAs. In addition, existing quality indicators for adults often take a health care systems perspective, using health care utilization data, with a focus on management of pain and symptoms,21-27 limited use of intensive measures,23,25,26,28,29 and involvement of palliative care and hospice.21,23,25-27,30,31 Although health care systems–based indicators are important, they do not speak to the individual patient and family experience of care. The 2013 National Academy of Medicine report, Delivering High-Quality Cancer Care: Charting a New Course for a System in Crisis,32 recommends use of an array of indicators defined from multiple perspectives, including process indicators based on care delivery and indicators that evaluate the patient and family experience, noting “pervasive gaps in existing cancer measures”32(p15) due to “a lack of consumer engagement in measure development.”32(p15)

    The goal of this study was to develop a patient-centered definition of high-quality end-of-life care for AYAs with cancer and to use that definition to propose quality domains and indicators. We conducted qualitative interviews with AYA patients aged 12 to 39 years with advanced (stage IV or recurrent) cancer; family caregivers of living AYAs with advanced cancer, as well as bereaved family caregivers; and multidisciplinary clinicians who care for AYAs with advanced cancer. Interviews were designed to identify domains of importance to AYAs and their family members. We then worked with a 12-member multidisciplinary advisory group, including physicians in medical and pediatric oncology and hospice or palliative medicine, pediatric oncology nurse practitioners, a medical oncology physician assistant, a patient advocate, and psychosocial health care professionals, to translate priority domains into proposed quality indicators. When possible, existing quality indicators were recommended, but in select areas, a need for new indicators was identified when existing quality indicators were not adequate to speak to identified domains.

    Methods

    Participants were recruited for this qualitative study between December 6, 2018, and January 5, 2021, from 3 sites (the Dana-Farber Cancer Institute in Boston, Massachusetts; Kaiser Permanente Northern California in Oakland; and Kaiser Permanente Southern California in Pasadena) and online via an AYA cancer support and advocacy organization, Lacuna Loft (lacunaloft.org). Eligible participants included English- or Spanish-speaking AYA patients aged 12 to 39 years with advanced (stage IV or recurrent) cancer; family caregivers of living AYAs with advanced cancer, or of AYAs with cancer who had died within the past 5 years; and clinicians (physicians, nurses or nurse practitioners, and psychosocial clinicians) who care for AYAs with advanced cancer. The age range of 12 to 39 years was chosen to cover both the World Health Organization definition (12-24 years) and National Cancer Institute definition (15-39 years) of an AYA. Eligible patients and caregivers were identified via clinic lists, administrative databases, and/or referrals by clinicians. We used purposive sampling to ensure a range of ages and racial/ethnic representation. An online advertisement on the Lacuna Loft website allowed for self-referral of patients and caregivers. Eligible clinicians were identified by investigators at each site. Signed informed consent was obtained from all participants.

    The institutional review boards of the Dana-Farber Cancer Institute and Kaiser Permanente Northern and Southern California approved the study. This report followed the Consolidated Criteria for Reporting Qualitative Research (COREQ) reporting guideline for qualitative research.33

    With the exception of those who self-referred through Lacuna Loft, permission from the patient’s oncologist was requested for contact of patients and family members. Initial contact was from investigators at the patient’s primary site. Eligible patients (or parents/guardians if younger than 18 years) were approached in person or by mail with a letter describing the study and an informed consent document. Interested patients were asked to respond to a 3-statement readiness assessment to evaluate readiness to discuss end-of-life issues34: (1) it might be helpful for me to talk about what would be important to me if treatments were no longer effective; (2) talking about what might be important to me if treatment options were limited or there were no more treatment options available would upset me very much; and (3) I feel comfortable discussing what might be important to me if treatments were no longer effective. Eligible participants were those who responded yes, no, and yes, respectively.

    We sought to recruit approximately 20 to 30 patients, 20 to 30 caregivers, and 20 to 30 clinicians, with recruitment through thematic saturation in each group and with representation across the AYA age range, race/ethnicity, caregiver relationship, and clinician type. For patients in the younger age group (12-24 years), we recruited patients and conducted interviews in person to ensure adequate detection of distress. When in-person recruitment was affected by the COVID-19 pandemic, we increased sampling of caregivers of younger patients to enhance representation of their perspectives, recognizing the limitations of caregivers to directly speak to patient perspectives. Sixty individuals declined participation, including 32 patients (or caregivers of adolescent patients who declined on their behalf), 8 caregivers, and 20 clinicians.

    Semistructured interviews were conducted at the Dana-Farber Cancer Institute in person or by phone by trained interviewers (J.W.M., B.V., G.R., and I.J.). No interviewers had preexisting relationships with participants; one interviewer, J.W.M., is a pediatric oncologist and conducted limited interviews with medical oncology patients during interviewer training but was not involved in their care. All other interviewers were research assistants. The advisory group reviewed the interview guide to ensure developmental appropriateness and sensitivity. Interviews centered on prognostic awareness; experiences with care; care priorities ; use of medical interventions; differences across the AYA age and developmental spectrum; and nonmedical priorities, such as ways to cultivate meaning (eTable in the Supplement). Patients who disclosed that they had a poor prognosis were asked about personal priorities for care. Those who did not express awareness of poor prognosis were asked to imagine what might be important to them if treatment was no longer working. Caregivers were asked to describe what they felt was most important in the care of their AYA family member, and clinicians were asked to reflect on priorities identified through general experience with AYAs as well as experiences with a recently cared-for AYA with advanced cancer. Interviewers were trained to recognize distress and respond by offering to end the interview early and/or refer the participant for psychosocial support. Some participants, especially bereaved caregivers, expressed grief and sadness; no participants wished to end the interview early or speak with a psychosocial professional.

    Interviews were audiorecorded and transcribed. Limited field notes on prognostic awareness and emotional distress were recorded by interviewers. Median interview duration was 31 minutes (range, 14-79 minutes). Transcripts were reviewed for interview quality by J.W.M., with feedback and opportunity for retraining of interviewers if needed. Interviews were offered in English and Spanish, although no participants elected to participate in Spanish. Participation ended once the interview was complete, with no additional follow-up.

    Analysis

    Directed content analysis was used to evaluate transcribed interview data35-39 using NVivo software, version 1.4 (QSR International). An initial coding schema was developed based on broad domains of high-quality end-of-life care from the literature as well as a priori conceptual categories (eg, attainment of life goals, legacy, support of relationships). Two coders (L.F. and J.W.M.) performed coding independently and then reviewed together, with discrepancies used as opportunities to clarify or expand the coding scheme. New themes were added as they emerged from texts. Coders identified core themes expressed across all interviews, and themes that differed by age, stage in development, and stakeholder (ie, patient, caregiver, or clinician). Participant checking, ie, where interview participants are asked to review study results and comment on them, was not used. The advisory group reviewed results, including select quotations from transcripts, to create an expansive list of end-of-life care priorities.

    In a series of virtual meetings, the advisory group then translated care priorities into potential quality indicators, considering ways that high-quality care might support identified priorities. For example, for the domain of attainment of life goals, actual attainment of life goals might not always reflect the quality of care, because not all goals are achievable or a result of efforts by the care team. However, high-quality care systems might ensure recognition of goals and discussion of life goals as part of care planning conversations.

    Once potential indicators were identified, we reviewed existing end-of-life care quality indicators within the group, with an eye toward using existing quality indicators when possible. We then delineated areas where new indicators were needed to speak to identified domains and worked within the advisory group to develop those indicators. The advisory group conducted iterative discussions, with drafting and revision of proposed quality indicators, until consensus was obtained, defined as agreement within the group that the proposed indicators were adequate and no new proposals of indicators were obtained.

    Results

    Interviews were conducted with 23 patients (mean [SD] age, 29.3 [7.3] years; 12 men [52%] and 11 women [48%]; 18 White participants [78%]), 28 family caregivers (23 women [82%] and 5 men [18%]; 14 White participants [50%]), and 29 clinicians (20 women [69%] and 9 men [31%]; 13 White participants [45%]). Caregivers included 22 parents (79%), 5 spouses or partners (18%), and 1 other family member (4%); the 29 clinicians included 15 physicians (52%), 6 nurses or nurse practitioners (21%), and 8 social workers or psychologists (28%) (Table 1).

    We identified 7 distinct end-of-life priority domains across all interviews: attention to physical symptoms, attention to quality of life, psychosocial and spiritual care, communication and decision-making, relationships with clinicians, care and treatment, and independence. No major differences were identified by participant type or across the AYA age range, although bereaved caregivers offered slightly greater focus on the days and hours before death across domains. Domains are shown in Table 2 along with illustrative quotations from participants.

    Each domain included relevant subdomains. For example, under attention to physical symptoms, participants prioritized attention to comfort and freedom from symptoms and suffering, but also the opportunity to maintain cognitive awareness when possible. The quality-of-life domain included global quality of life, but also attainment of life goals, legacy, and meaning; support of personal relationships; and the opportunity to maintain a sense of normalcy. Support of personal relationships included not just opportunity to be with loved ones and enjoy time together, but also support for the needs of those loved ones during and after the life of the AYA. Psychosocial and spiritual care included support in both domains, with acknowledgment that spiritual care includes support in dealing with existential challenges encountered by young people facing death.

    For the domain of communication and decision-making, relevant subdomains included communication about prognosis, communication about what to expect at the end of life, timely communication early in the disease course, and opportunity for the AYA to hold their desired role in decision-making. In this domain, emphasis was placed on ensuring that AYAs have a say in the information they receive and when, as well as opportunity for their values for care to be heard and respected. In the related domain of relationships with clinicians, participants emphasized the importance of a strong therapeutic alliance with clinicians as well as continuity and compassion.

    The domain of care and treatment included subdomains focused on use of cancer-directed therapy, use of life-sustaining therapy, and location of death. Although most participants emphasized a desire to avoid cancer therapy and intensive measures if they were unlikely to be effective, a minority of patients and family members expressed a desire to do everything possible to prolong life. Clinicians, too, noted that preferences are highly individual and that the best care for this population meets those individual preferences.

    Finally, participants emphasized a need for independence in medical care when possible as well as freedom from burdening others.

    Table 3 shows proposed quality indicators in each domain and subdomain and recommendations about whether new or existing indicators might best represent each.40-43 For example, attention to pain and symptoms is encompassed in a number of existing indicators for adults. However, we were unable to identify existing indicators that supported maintaining an experience of normalcy. We therefore proposed development of new indicators to support this priority.

    Discussion

    Interviews with AYAs, caregivers, and clinicians identified 7 distinct domains of end-of-life care quality for this population. In addition to existing domains, we identified new areas as well as unique manifestations of existing domains relevant to this young population. For example, we identified attainment of life goals and meaning as well as experiences of normalcy as key constructs of quality of life for AYAs. Adolescents and young adults also identified a need for independence as a core domain missed in existing quality indicators.

    We also found that some indicators, especially aspects of care delivery such as end-of-life cancer therapy or location of death, were dependent on the preferences of individual patients. Many participants, across all participant types and AYA ages, agreed that late-life chemotherapy is undesirable if there is little prospect of benefit. However, others felt that trying to prolong life, even at the cost of quality of life, can be significant for young people whose lives are being cut short, as well as to patients who may have children or other loved ones for whom any additional time is worthwhile. Thus, all participants generally agreed that optimal care and treatment at the end of life for AYAs is not universal, but rather what is right for the individual patient.

    The need for sensitivity to preferences was also emphasized in indicators related to communication and decision-making. Although many AYAs want to hear about prognosis, for example, some may prefer to defer these conversations or delegate them to other trusted individuals. For these indicators, we emphasize that patients need a say in how, when, and with whom these conversations take place.

    Because many indicators are highly individualized and focused on the patient experience, many will require patient and/or caregiver report. This need poses challenges to large-scale quality assessment. Where possible, we have proposed potential administrative or medical record–based indicators to facilitate more widespread data collection. However, sole reliance on administrative indicators will miss many of the constructs that are salient to this young population. We therefore advocate for use of a patient or caregiver report when needed and possible. These new indicators require further development and validation across a diverse population.

    Assessment of AYA end-of-life care quality will require careful consideration of implementation methods. Because patient report is of value, prospective identification of appropriate populations will be required, and we currently lack a definition of which patients might be most appropriate for such assessment and when. We included patients with stage IV or recurrent disease in this study, and although some had the prospect of a cure, they were able to discuss end-of-life issues because they were already thinking about these issues. Thus, a relatively inclusive population of patients with a limited prognosis from the time of diagnosis may be appropriate, despite our own use of the term end of life; in fact, some areas may be salient to all AYAs with cancer. In addition, processes around data collection and reporting, including whether findings should be presented in the aggregate or available at the individual level for review by clinicians, patients, and caregivers, will need to be defined. These questions offer the opportunity to define health systems that are responsive to the needs of AYAs with advanced cancer and their families.

    Limitations

    This study has some limitations. Racial and ethnic diversity was limited, and no Asian or Spanish-speaking patients or caregivers participated. The preferences of a diverse population need continued focus. In addition, by requesting permission from clinicians and screening with the readiness assessment, we may have identified patients who were more open to conversations about end-of-life care and whose values reflected those preferences. Notably, many patients declined participation. However, inclusion of family caregivers and clinicians was used to ensure a diversity of experiences, and clinicians were specifically probed to consider recent patients without respect to the choices they made for care. Our next step, a Delphi process (ie, a structured communication technique to help a group arrive at a decision) with patients, caregivers, and clinicians to prioritize identified indicators, will allow us to obtain broader input on our findings with renewed efforts to include a diverse population. We interviewed few patients in the youngest age range, in part due to challenges of in-person interviews during the COVID-19 pandemic. We dealt with this by enrolling more caregivers of adolescents and ensuring inclusion of pediatric clinicians. The content from interviews with younger patients was consistent with that from older patients. However, the needs of younger AYAs require ongoing attention and emphasis. Although no interviewers were known to the participants, 1 interviewer was a pediatric oncologist, raising concerns about reflexivity. We worked to mitigate this concern by involving a patient advocate at each step from interview development to analysis.

    Conclusions

    Despite recommendations from the National Academy of Medicine for inclusion of stakeholder voices in the development of quality indicators, inclusion of patient and caregiver perspectives remains an underused aspect of quality indicator development.32 In addition to revealing important quality domains for AYAs, some of the domains we identified in this qualitative study may have relevance to older adults as well. These domains may have not been previously identified owing to more limited use of patient perspectives in quality measure development. Although these topics were difficult for young people and their family members to discuss, many strongly desired to express what was most important to them and sought care that would support those personal values. Responsive health care professionals and systems can honor these values by systematically measuring and reporting salient areas to promote high-quality care for young people at the end of life.

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    Article Information

    Accepted for Publication: June 9, 2021.

    Published: August 23, 2021. doi:10.1001/jamanetworkopen.2021.21888

    Open Access: This is an open access article distributed under the terms of the CC-BY License. © 2021 Mack JW et al. JAMA Network Open.

    Corresponding Author: Jennifer W. Mack, MD, MPH, Department of Pediatric Oncology, Dana-Farber Cancer Institute, 450 Brookline Ave, Boston, MA 02215 (Jennifer_mack@dfci.harvard.edu).

    Author Contributions: Dr Mack had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.

    Concept and design: Mack, Chao, Josephs, Buchanan, Fasciano, Kolevska, Lakin, Wall.

    Acquisition, analysis, or interpretation of data: Mack, Fisher, Kushi, Chao, Vega, Rodrigues, Josephs, Brock, Buchanan, Casperson, Cooper, Fasciano, Lakin, Lefebvre, Schwartz, Shalman, Wall, Wiener, Altschuler.

    Drafting of the manuscript: Mack, Josephs, Brock, Buchanan, Schwartz.

    Critical revision of the manuscript for important intellectual content: Mack, Fisher, Kushi, Chao, Vega, Rodrigues, Josephs, Brock, Casperson, Cooper, Fasciano, Kolevska, Lakin, Lefebvre, Schwartz, Shalman, Wall, Wiener, Altschuler.

    Statistical analysis: Brock.

    Obtained funding: Mack, Kushi.

    Administrative, technical, or material support: Fisher, Kushi, Vega, Rodrigues, Josephs, Buchanan, Casperson, Lakin.

    Supervision: Kushi, Chao.

    Qualitative analysis: Altschuler.

    Conflict of Interest Disclosures: Dr Mack reported receiving grants from the National Institutes of Health during the conduct of the study. Dr Kushi reported receiving grants from the National Cancer Institute as a subcontract from Dana-Farber Cancer Institute during the conduct of the study. Dr Fasciano reported receiving consultation fees from Oncology Analytics for a virtual oncology supportive care product outside the submitted work. No other disclosures were reported.

    Funding/Support: This study was supported by grant 5 U01 CA218651 from the National Cancer Institute (Dr Mack).

    Role of the Funder/Sponsor: The National Institutes of Health/National Cancer Institute as study funder had no role in design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication.

    Additional Information: Advisory group members include Katharine E. Brock, MD, MS; Susan Buchanan, MS, PA-C; Mallory Casperson, MS; Robert M. Cooper, MD; Karen M. Fasciano, PsyD; Tatjana Kolevska, MD; Joshua R. Lakin, MD; Anna Lefebvre, NP; Corey M. Schwartz, MD; Dov M. Shalman, MD; Catherine B. Wall, NP; and Lori Wiener, PhD.

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