Financial burden for caregivers of adolescents and young adults with cancer

Abstract Objective Adolescent and young adult (AYA) cancer survivors are vulnerable to cancer‐related financial burden, which is likely shared by their caregivers. This study aims to enhance an existing conceptual model of financial burden by conducting concept elicitation interviews with caregivers to generate knowledge that can be translated to inform instrumental and psychosocial support in cancer care. Methods Qualitative concept elicitation interviews were conducted with 24 caregivers of AYA cancer survivors (caregivers of adolescents, n = 12; caregivers of emerging adults, n = 12) recruited from four sites. Constant comparative methods were used to identify themes, and results were interpreted and organized into domains of the conceptual model. We also explored COVID‐19 related financial impacts among a subset (n = 12) of caregivers. Results Seven themes emerged, which varied by age group and strengthened the conceptualization of the model. Themes centered on: (1) direct and indirect costs of cancer; (2) impact of socioeconomic status on financial burden; (3) caregiver desire to shield AYAs from distress due to financial burden; (4) strategies to manage cancer‐related costs; (5) worries about AYAs' financial future; (6) seeking and receiving financial support; and (7) navigating the healthcare system. Findings also revealed that COVID‐19 exacerbates financial burden for some caregivers. Conclusions Building upon our prior work, we have adapted the conceptual model of financial burden to reflect perspectives of AYAs, oncology providers, and now, caregivers. An important next step is to develop a reliable and valid self‐report measure of financial burden among caregivers of AYA cancer survivors.


| BACKGROUND
Informal (unpaid/family) cancer caregivers are essential members of the health care team, providing an average of 33 h a week caring for their loved one. 1 Though caring for a loved one with cancer can be rewarding, the negative implications for the caregiver can be broad and substantial. [2][3][4] A scant, but growing body of literature describes cancer-related financial burden among informal (unpaid/family) caregivers with 25% of cancer caregivers reporting high levels of financial burden, often secondary to interruptions in employment (e.g., taking time off, taking a leave of absence) and costs associated with cancer care. 1,[5][6][7] External financial resources for cancer caregivers are limited, and caregivers report high unmet financial needs. 5,8 Increased financial burden in caregivers negatively impacts patients' health and care delivery as it is associated with poorer treatment adherence and quality of life. 9 Cancer-related financial burden may be exacerbated in those providing care to adolescent and young adult (AYA) survivors (i.e., those diagnosed with cancer from the ages of 15-39 years), a population at high risk for having inadequate insurance coverage and limited financial assets. [10][11][12][13] Compared to other age groups with cancer, AYAs self-report greater financial burden. [10][11][12] Caregivers often share or shoulder the responsibility for their loved one's cancer-related costs, and this burden may be greater and more persistent among those caring for younger AYAs (ages 15-25 years), as this group is beginning to navigate financial independence. 5,14 In addition to caregivers contributing to treatment-related costs, caregivers of young adults may also need to abruptly take responsibility for the young adult's living expenses, as they may be more likely to move in with their caregiver during treatment. 15 Further, unlike adult cancer patients who have an active role in managing the costs associated with their cancer care, adolescent cancer survivors report limited awareness of costs, which are instead managed by a family caregiver. 16 To our knowledge, no qualitative studies have explored the financial burden of cancer on caregivers of AYAs, though studies have documented substantial financial burden among parents of children with cancer. [17][18][19] Our team recently conducted a systematic review and found financial burden measures are sparse for cancer caregivers and non-existent for caregivers of AYAs. 20 A critical first step to understanding the multidimensional financial impact of cancer on caregivers of AYAs is to develop a psychometrically sound measure that specifically captures cancer-related financial burden among caregivers. Accordingly, we conducted qualitative concept elicitation interviews with stakeholders to refine a conceptual model for financial burden of cancer in AYAs as part of a standardized measurement approach (See Figure 1). 16,21 We previously reported on the conceptual model following AYA survivor and provider interviews, which reflects the multiple layers of impact and influence on financial burden including AYA patients/families, health care providers, the broader healthcare system, and the health policy environment. 16 Further, the model depicts critical interrelated material, psychosocial, and behavioral aspects of experience that characterize financial burden for patients and families and the "ripple effects" of indirect costs related to cancer. 16 This study aims to add to the existing model by capturing salient domains of financial burden focusing on the caregiver perspective to enhance instrumental and psychosocial support in cancer care. In this paper, we: (1) perform data source triangulation by presenting findings on caregivers' financial burden and how it relates to the conceptual model 22 ; (2) explore how financial burden varies for caregivers of adolescents (15-17 years old) and emerging adults (18-25 years old); and (3) explore financial burden associated with COVID-19 during or after cancer treatment.

| Study design
Semi-structured qualitative concept elicitation interviews were conducted with 24 caregivers of AYAs between 13 November 2018 and 12 March 2021.

| Recruitment
Caregivers of AYA cancer survivors were eligible if they: (1) were ≥18 years of age; (2) were able to read and understand English; (3) had a loved one for whom they were providing care and at least partly financially responsible; and (4) had a loved one who met criteria as an eligible AYA cancer survivor participant in the larger study. 16

| Data collection
Semi-structured interviews (Mean = 46 min) were conducted with 24 caregivers of AYA patients (adolescents, n = 12; emerging adults, n = 12). Interviews were conducted in-person (n = 8; WFBCCC only), by video conference (n = 12), or by phone (n = 4). All interviews were conducted through the Qualitative and Patient Reported Outcomes (Q-PRO) Shared Resource at the WFBCCC by a trained research associate who has expertise in qualitative methods. The interview guide followed a funnel approach with open-ended questions eliciting participants' understanding and experience of financial aspects of cancer with subsequent follow-up questions exploring material (e.g., financial resources and deficits), psychosocial (e.g., distress related to financial hardship), and behavioral (e.g., cost-cutting strategies) factors that shape and/or characterize their perception of financial burden.
Data collection was halted briefly in the spring of 2020 due to COVID-19 restrictions. During that time, the study team revised the interview guide to include questions specific to the financial burden of COVID-19. Therefore Wave 1 (n = 12) did not include COVID-19related discussion but Wave 2 (n = 12) did. All interviews were recorded.

| Data analysis
All interviews were outsourced for transcription. Each transcript was reviewed by the interviewer for accuracy. Two Q-PRO research associates reviewed all transcripts to devise and test conceptual codes.
Transcripts were coded in tandem; coding discrepancies were resolved with discussion. Final primary analyses were stratified by participant group (caregivers of adolescents; caregivers of emerging adults). Data saturation was evaluated at the theme level within each strata. Interviews within each strata were divided into sets of four based on chronological order. Themes were considered saturated if they were present in at least one interview within each set. 23 We reviewed the thematic categories and associated data to identify if and how themes fit within domains previously identified in the conceptual model. Domain affiliations were discussed and confirmed by study authors. Differences were resolved by consensus. 16 Data were stored and managed using ATLAS. ti (Version 7.5).   Table 2 provides a summary of caregiver financial burden themes and related conceptual model domains. 16 See Supplemental Table 3 for themes, theme properties, exemplar quotes, and theme variation by age group.

| Theme 1: Socioeconomic status at the time of diagnosis and treatment can amplify or mitigate financial burden
Caregivers reported financial burden was mitigated by: 1) steady income, especially multiple stable salaries from two-income households; and, 2) health insurance (public, private, Medicaid) that secured the ability to pay for direct and indirect costs of cancer.

| Theme 3: Caregivers shield AYAs from financial burden
Caregivers consistently described the belief that financial burden is a stressor an AYA with cancer "does not need." They fear AYAs will become "overwhelmed," "worried," "anxious," or "crushed." Consequently, caregivers employed strategies to "shield" or "shelter" AYAs.
Caregivers reported hiding medical bills from AYAs, omitting information about the extent of the impact on the caregiver's finances, minimizing depth and specificity of information disclosed (e.g., "we're in a tight spot"), or admitting financial challenges but assuring AYAs they can be managed. This theme mapped onto all three domains of the conceptual model.

| Theme 4: Caregivers seek and/or receive financial support from social networks and healthcare institutions/organizations
Caregivers sought and received financial support from organizations, family and friends, and community (school, employers, Church) event-based, in-person, and social media fundraisers. Insurance companies, hospitals, and cancer centers also assigned case workers/navigators who connected caregivers with financial resources for treatment costs, lodging, travel expenses, and food.
This theme mapped onto all three domains of the conceptual model.

| Theme 5: Caregivers desire help navigating the healthcare system and locating financial resources
Caregivers reported they would like assistance navigating financial aspects of the healthcare system and locating financial resources.
They described receiving information about procedures, costs, and internal/external resources in "bits and pieces" at "random times" via intermittent phone calls from different people across the cancer trajectory. They described a desire for face-to-face meetings with one dedicated financial case worker/navigator, clear written documentation of costs and resource contact information, and email summaries of meetings. At times, financial assistance/resources were delivered after significant debt had accrued. They reported they would like assistance managing deadlines to ensure payments are made on schedule. This theme mapped onto all three domains of the conceptual model.

| Theme 6: Caregivers use strategies to prepare for and adapt to cancer-related costs
Caregivers suggested they would do whatever is needed to pay cancer-related costs and "resolve" financial consequences; they employed multiple strategies to prepare for/adapt to those costs.
Caregivers reported delaying unnecessary travel and vacations and reducing non-essential expenses for themselves and their children.
They took steps to maximize their financial resources including applying for loans, deferring existing debt payments, using savings, working higher-paying (and often less-enjoyable) jobs, longer hours, or multiple jobs. They reported researching cancer treatments, hospital procedures, and insurance systems to plan ahead financially. They delayed their own medical appointments or delayed filling AYA prescriptions to wait for generic drugs or meet deductibles. Caregivers reported that managing the AYA's cancer prompted them to reevaluate their spending philosophy as a parent/caregiver and renewed their intent to save money. This theme mapped onto the psychosocial and behavioral domains of the conceptual model.

| Theme 7: Caregivers worry about AYAs' medical and financial futures
Caregivers discussed concerns about future financial burden. They  had not begun to make career decisions. Caregivers of adolescents also reported concern that cancer would delay or interfere with developmental milestones that could impact their future finances.

| COVID-19-related financial impacts
Analyses revealed the COVID-19 pandemic affected financial burden (see Supplemental Table 4). While some caregivers' income remained consistent (e.g., essential workers) or increased (suppliers of critical materials or resources), many caregivers, families, and AYAs experi-

| DISCUSSION
This study is the first to explore experiences of financial burden among caregivers of AYA survivors. These findings deepen our understanding of material, psychosocial, and behavioral domains of the adapted patient-and provider-informed conceptual model of cancerrelated financial burden, in particular, illuminating nuanced experiences of people caring for cancer patients in different phases of the AYA lifespan. 16,21 Further, this study uncovered insights about financial burden and AYA cancer caregiving, including the impact of the evolving COVID-19 pandemic, which can be used to enhance instrumental and psychosocial support in cancer care and the timing of that support.

| Conceptual model of financial burden and lifespan developmental insights
Results reinforce and extend the original material-psychosocialbehavioral conceptual model that we adapted based on AYA patient and provider perspectives. 16,21 Insurance coverage and financial assistance (material domain) shaped participants' psychosocial and behavioral experiences in both studies. In our prior work, emerging adult AYA participants reported the ability to assist with cancer costs, like traveling to treatment centers and paying medical bills, but they also reported varied, less consistent sources of insurance. 16 In this study, caregivers of emerging adults who were approaching 26 years of age discussed concerns about future insurance coverage. They suggested emerging adults may not be ageeligible for future coverage through caregiver's insurance, which magnifies financial burden. Further, we previously found though adolescents and older AYAs report financial burden, adolescents described extensive financial assistance through social networks and special assistance programs as compared to older AYAs. 16 Providers in our prior study posited that young patients receive more assistance than older AYAs because people are more motivated to donate to younger cancer patients. 16

| Clinical implications
This study highlights the need for healthcare professionals to be aware that age, employment status, and level of caregiver sup-

| Strengths
This study is strengthened by the rigorous qualitative methods that were used to collect and analyze data. 23 In addition, we compared our findings for caregivers of adolescents and caregivers of emerging adults and found important differences for caregivers of these two age groups. Further, while studies have been conducted that highlight the validity of the material-psychosocial-behavioral conceptual model of cancer-related financial burden, we have adapted this model to reflect perspectives of AYA cancer survivors, oncology providers, and informal caregivers of AYAs. 16,21,[35][36][37]

| Future directions
This study is part of a larger initiative to develop self-report measures of financial burden for AYAs and their caregivers and builds upon our team's recent systematic review of financial burden measures and concept elicitation interviews with AYA cancer survivors and oncology providers. 16

ACKNOWLEDGMENTS
The authors would like to thank the caregivers for their participation in this study; we are grateful to have had this opportunity to learn about their experiences with cancer-related financial burden.
We also want to acknowledge Dr. Sharon Castellino of the Aflac