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. Author manuscript; available in PMC: 2025 Apr 1.
Published in final edited form as: Cancer. 2023 Dec 15;130(7):1125–1136. doi: 10.1002/cncr.35150

Financial hardship among siblings of long-term survivors of childhood cancer: a Childhood Cancer Survivor Study report

Timothy J D Ohlsen 1,2, Huiqi Wang 3, David Buchbinder 4, I-Chan Huang 3, Arti D Desai 1, Zhiyuan Zheng 5, Anne C Kirchhoff 6,7, Elyse R Park 8, Kevin Krull 3, Rena M Conti 9, Yutaka Yasui 3, Wendy Leisenring 2, Gregory T Armstrong 3, K Robin Yabroff 5, Paul C Nathan 10, Eric J Chow 1,2
PMCID: PMC10939878  NIHMSID: NIHMS1947538  PMID: 38100563

Abstract

Introduction:

Siblings of children with cancer may experience adverse household economic consequences, but their financial outcomes in adulthood are unknown.

Methods:

We surveyed 880 siblings (18-64 years) of adult-aged childhood cancer survivors to estimate the prevalence of financial hardship by three established domains (behavioral, material, psychological). For individual financial hardship items matching contemporaneous National Health Interview Survey or Behavioral Risk Factor Surveillance System, we compared siblings with the general population, calculating adjusted prevalence odds ratios (OR) to sample-weighted responses. Multivariable logistic regression models examined associations between sibling characteristics and each hardship domain, and between sibling hardship and survivors’ cancer/treatment characteristics.

Results:

Behavioral, material, and psychological hardship was reported by 24%, 35%, and 28%, respectively. Compared with national survey respondents, siblings were more likely to report worries about medical bills (OR 1.14, 95% CI 1.06–1.22), difficulty affording nutritious foods (OR 1.79, 95% CI 1.54–2.07), and foregoing needed medical care (OR 1.38, 95% CI 1.10–1.73), prescription medications (OR 2.52, 95% CI 1.99–3.20), and dental care (OR 1.34, 95% CI 1.15–1.57) due to cost. Sibling characteristics associated with reporting financial hardship in one or more domains included female sex, older age, chronic health conditions, lower income, not having health insurance, high out-of-pocket medical expenditures, and non-medical/non-home debt. No survivor cancer/treatment characteristics were associated with sibling financial hardship.

Conclusions:

Adult siblings of childhood cancer survivors were more likely to experience financial hardship compared with the general population. Childhood cancer may adversely affect entire households with potential lasting implications.

Keywords: Pediatric oncology, cancer survivorship, financial toxicity, financial hardship, financial worry, family members, siblings of children with cancer

Précis:

Adult siblings of long-term childhood cancer survivors may experience greater aspects of financial hardship compared with the general population. A childhood cancer diagnosis and treatment may adversely affect entire households, with potential lasting implications.

INTRODUCTION

As over 85% of children diagnosed with cancer will become five-year survivors, approximately 500,000 survivors of childhood cancer reside in the United States, many of whom are at elevated risk for physical, neurocognitive, and psychosocial comorbidities, as well as long-term financial toxicity related to their cancer and its treatment.1–7 Cancer diagnosis and treatment may also have a marked adverse impact on siblings, who play an integral role in a child’s cancer experience, given their proximity, emotional ties, and shared household resources.8–12 For example, emotional impairment among siblings is common during treatment and often persists following therapy completion.10,13,14 Psychosocial care for family members during cancer treatment is therefore a recommended standard,15 albeit applied inconsistently.16

A childhood cancer diagnosis and treatment can also have substantial adverse economic and psychosocial consequences for households. For example, with intensive treatment, many parents take paid and unpaid leave from work, reducing household income.17–20 Families typically also experience increased out-of-pocked expenses.21,22 Perhaps reflecting this, childhood cancer survivors as adults frequently experience various financial challenges, incurring higher medical expenditures, obtaining lower-paying employment, and experiencing greater productivity losses from missed work than their counterparts without a cancer history.23–25

Although adverse economic impacts during pediatric cancer treatment are experienced at the household level,26,27 little is known about the long-term economic experiences of siblings in adulthood. Given that financial impact on families may have consequences for other household members, or that some adult siblings may devote significant time and financial resources to caregiving or support for cancer survivors with ongoing medical requirements,28 there is a need to better understand siblings’ financial experiences as adults.

In this study, we examined financial hardship experienced by siblings as adults, using data from the Childhood Cancer Survivor Study (CCSS) cohort with publicly available national survey data to reflect the general US population without a cancer history. We also sought to assess survivor- and sibling-level associations with siblings’ reported financial hardship. We hypothesized that siblings of cancer survivors would report a greater prevalence of financial hardship compared with the general US population without a cancer history, and that sibling financial hardship would be associated with sibling demographic and survivor treatment characteristics. Results from this investigation may help inform the design of programs or policies to support the financial well-being of siblings of children with cancer and their families during treatment and in survivorship.

MATERIALS & METHODS

Study population and data sources

The CCSS is a retrospective, multi-institutional cohort study with longitudinal follow-up of 25,735 five-year survivors of cancer diagnosed from 1970 to 1999 before the age of 21 years and over 5,000 nearest-age siblings.6,29 The CCSS was approved by the 31 member institutions’ Institutional Review Boards and all participants provided informed consent for data collection. From 2017 to 2019, the CCSS surveyed a randomly selected sample of 1,590 siblings enrolled in the CCSS cohort on questions regarding financial hardship (Follow-Up 6; ccss.stude.org).6 The questionnaire was developed from existing evidence related to financial challenges associated with cancer care,3,30 and incorporated 20 items derived from the publicly available National Health Interview Survey (NHIS)31 and Behavioral Risk Factor Surveillance System (BRFSS)32 (Supplemental Table 1). Items were grouped into three previously defined financial hardship domains: material [difficulty in affording basic necessities such as food, housing, utilities, or medical care], psychological [increased or excessive degree of worry regarding finances], and/or behavioral [intentionally delaying, forgoing, or rationing recommended medical care to save money].6 We excluded siblings whose survey was completed by a proxy respondent and those with non-US health insurance (including Canadian participants). We also excluded siblings 65 years or older, due to universal age-eligibility for coverage for the US federal Medicare program, as well as those with a history of cancer. Data from the NHIS and BRFSS are publicly available and de-identified, so were exempt from Institutional Review Board review.

To allow for comparisons between siblings and the general US population, we also included 21,271 respondents to the NHIS and 259,901 respondents to the BRFSS who answered questions corresponding to matched CCSS financial hardship items (7 NHIS questions, 1 BRFSS question). We selected the 2019 NHIS for comparison as it was conducted synchronously with the CCSS follow-up survey and corresponding items between these surveys were identical or near-identical (Supplemental Table 2). Furthermore, because the 2019 NHIS was redesigned and differed from prior NHIS years,33 we did not include respondent data from 2017-2018. Similarly, we selected the 2017 BRFSS for comparison, given its concurrent administration and overlapping elements to the CCSS follow-up survey. We excluded NHIS/BRFSS respondents who were 65 years or older to align with the CCSS sample, as well as those with a history of cancer (Supplemental Figure 1).

Financial hardship outcomes

Responses to each financial hardship item in the CCSS and corresponding items in the NHIS and BRFSS were categorized as yes, no, refused, or don’t know (Supplemental Table 2). For comparisons between siblings and the general population, the proportions of positive responses to each individual survey item were the main outcomes of interest. For analyses of sibling- and survivor-level correlates of financial hardship in siblings, outcomes were defined for each individual financial hardship domain as whether the sibling reported any financial hardship item in that domain. Additionally, survivors and sibling participants were asked in two separate items whether they had ever declared bankruptcy or were sent to debt collections. Responses to these two questions were each categorized as separate outcomes of interest, as they did not fit clearly into one of the hardship domains based on prior research.6

Statistical analysis

We used descriptive statistics to calculate and compare the sociodemographic characteristics of CCSS siblings and national survey participants. For the CCSS items of financial hardship with a corresponding item on the national surveys, we estimated adjusted odds ratios (OR) of the prevalence of the financial hardship item with 95% confidence intervals (CI) to compare siblings to the US general population using a multivariable logistic regression model, adjusting for age, sex, race and ethnicity, household income, educational attainment, and marital status at the time of the survey. Because both the NHIS and BRFSS use complex survey designs and oversample certain populations,34,35 we incorporated sampling weights in analyses of these data to ensure nationally representative comparisons.

We constructed multivariable logistic regression models to evaluate associations between sibling-level characteristics and outcomes of interest, estimating ORs for each covariate with 95% CI. Multivariable models were adjusted for siblings’ age, sex, race and ethnicity, household income, educational attainment, marital status, chronic health conditions (limited to Grade 3+ [i.e., severe or disabling];36 none, one, or multiple), health insurance coverage type, out-of-pocket medical expenses, and non-home/non-medical debt at the time of the survey.

To evaluate associations between outcomes of interest among siblings and characteristics of cancer survivors themselves, we constructed separate multivariable logistic regression models. Each model included the following characteristics of cancer survivors as covariates of each sibling financial hardship domain: age at cancer diagnosis, time from diagnosis, sex, diagnosis type, number of Grade 3+ chronic health conditions (none, one, or multiple), survivors’ marital status, receipt of radiation, hematopoietic stem cell transplant, surgery-only cancer treatment, history of a late relapse (relapse occurring 5 or more years from diagnosis), and late death (also occurring 5 or more years from diagnosis). All statistical analyses were conducted in SAS version 9.4 (SAS Institute, Cary, NC).

RESULTS

Participant demographics

Of the 1,590 siblings of five-year childhood cancer survivors who were sent the survey, 1,037 (65.2%) completed it and 880 (55.3%) were included for analysis (Figure 1). Sibling responders, compared with non-responders, were significantly older and more often female, non-Hispanic White, and college educated (Supplemental Table 3). Responders were also less likely than non-responders to be the sibling of a CCSS survivor participant who were initially enrolled while the survivor participant was living but died prior to the financial hardship survey administration. We included 21,271 NHIS respondents and 259,901 BRFSS respondents who completed the corresponding questions on the respective surveys (Supplemental Figure 1). Response rates to the NHIS and BRFSS were 59.1% and 44.9%, respectively. Siblings had a median age of 46 years (IQR 38–52 years; Table 1) at the time of the survey, and a median time from cancer diagnosis of 37 years (IQR 33–41 years). Compared with the national survey respondents, siblings were more likely to be older, female, non-Hispanic White, college educated, and married or living with a partner.

Figure 1.

Figure 1

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CONSORT diagram depicting selection of sibling participants from the Childhood Cancer Survivor Study (CCSS) who completed the Follow-up 6 survey, 2017-2019.

Table 1:

Characteristics of siblings of survivors of childhood cancer versus national survey respondents

Characteristic, n (%) Siblings
N=880		 NHISa
N=21,271		 BRFSSa
N=259,901
Age at questionnaire (years)
 21-29 55 (6.3) 4,427 (27.7) 47,798 (27.7)
 30-39 224 (25.5) 5,044 (22.3) 49,212 (23.1)
 40-64 601 (68.3) 11,800 (50.0) 162,891 (49.2)
Sex
 Male 388 (44.1) 10,278 (49.8) 123,565 (50.6)
 Female 492 (55.9) 10,990 (50.2) 136,223 (49.3)
 Other/Refused 113 (0.0)
Race and Ethnicity
 White, Non-Hispanic 781 (88.8) 13,311 (58.8) 180,484 (56.7)
 Black, Non-Hispanic 13 (1.5) 2,582 (12.8) 24,252 (12.7)
 Hispanic/Latino 30 (3.4) 3,463 (19.1) 29,476 (19.8)
 Otherb 56 (6.4) 1,915 (9.3) 25,689 (10.8)
Chronic Health Conditions (Grade 3+)
 None 783 (89.0) c c
 1 or more 97 (11.0)
Annual household Income ($)
 0-35,000 79 (9.0) 5,420 (22.9) 76,270 (36.9)
 35,000-49,999 59 (6.7) 2,568 (11.9) 28,984 (12.6)
 50,000-74,999 128 (14.6) 3,914 (18.9) 35,678 (14.5)
 ≥75,000 609 (69.6) 9,369 (46.3) 83,219 (35.9)
Educational attainment
 <High school 9 (1.0) 1,721 (11.3) 18,463 (13.4)
 High school - some college 264 (30.1) 11,667 (59.4) 142,757 (59.1)
 College graduate and or greater 605 (68.9) 7,771 (29.3) 98,041 (27.5)
Marital Status
 Married 628 (72.8) 11,769 (61.2)d 148,321 (54.6)
 Single 146 (16.9) 62,171 (30.9)
 Divorced/Separated 84 (9.7) 9,002 (38.8)d 40,385 (12.6)
 Widowed 5 (0.6) 7,636 (2.0)
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a

Weighted samples responding to the NHIS or BRFSS

b

Due to small sample size (<10) of some groups, this category was aggregated from participants who self-reported their race and ethnicity as American Indian/Alaska Native, Asian/Pacific Islander, “Other”, or did not provide race and ethnicity information

c

Chronic health conditions were not collected similarly to the CCSS survey and therefore not included

d

The NHIS response option “living with a partner as an unmarried couple” was included as married; the NHIS also grouped single, divorced/separated, and widowed into a single category

Prevalence of financial hardship.

Siblings frequently reported behavioral, material, and psychological hardship—24.1%, 35.3%, and 28.2%, respectively (Figure 2). In addition, 22.3% of siblings reported having been sent to debt collection, and 7.7% reported having filed for bankruptcy. Some siblings also reported skipping needed medical care specifically due to cost, including medical care (8.3%), foregoing prescription medications (8.4%), mental health services (5.5%), and dental care (16.5%). Worries regarding ability to pay for food, housing, and utilities were reported by 16.4%, 24.6%, and 17.8%, respectively. Over half (51.6%) reported worries regarding ability to pay for medical bills in the event of sickness or an accident.

Figure 2.

Figure 2

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Prevalence of financial hardship by individual item, reported by adult siblings of long-term childhood cancer survivors enrolled in the Childhood Cancer Survivor Study. Bar colors correspond to financial hardship domain categories. Error bars denote standard errors. A summary of sibling responses to all survey items is available in Supplemental Table 5.

Comparison of siblings to the general population.

Compared with the US general population (Table 2) and adjusting for available sociodemographic covariates, siblings had greater odds of reporting financial hardship related to worries about paying bills (OR 1.14, 95% CI 1.06–1.22) and difficulty affording nutritious foods (OR 1.79, 95% CI 1.54–2.07). Additionally, siblings were more likely to forego aspects of medical care due to cost: any needed medical care (OR 1.38, 95% CI 1.10–1.73), prescription medications (OR 2.52, 95% CI 1.99–3.20), and dental care (OR 1.34, 95% CI 1.15–1.57).

Table 2:

Financial hardship for select common financial hardship items comparing siblings of survivors of childhood cancer with the US general population (NHIS, BRFSS)

Adjusted
ORc (95% CI)		 Adjusted
P-value
Items related to material hardship
 Problems paying medical billsb 1.19 (1.00 – 1.43) 0.074
 Unable to pay medical billsb 1.25 (0.96 – 1.61) 0.13
Items related to behavioral hardship
 Foregoing medical care due to costb 1.38 (1.10 – 1.73) 0.016
 Foregoing prescription medications due to costb 2.52 (1.99 – 3.20) <0.001
 Foregoing mental health care due to costb 1.11 (0.83 – 1.48) 0.51
 Foregoing dental care due to costb 1.34 (1.15 – 1.57) <0.001
Item related to psychological hardship
 Worried about ability to pay bills if getting sickb 1.14 (1.06 – 1.22) <0.001
 Worried about ability to afford nutritious foodsc 1.79 (1.54 – 2.07) <0.001
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a

Adjusted for age at questionnaire, race and ethnicity, sex, income category, education level, and marital status

b

Compared to those responding to the NHIS, using recommended sample weights

c

Compared to those responding to the BRFSS, using recommended sample weights

Correlates of sibling financial hardship.

Sibling characteristics associated with significantly greater frequency of reporting any behavioral financial hardship item included female sex, lower household income, greater non-home/non-medical debt, lack of health insurance coverage, and out-of-pocket medical expenses ≥10% of household income (Table 3). These factors, in addition to current age 30–39 years, were also associated with greater reporting of any material hardship item. Female sex, lower household income, and greater non-home/non-medical debt, and “Other” race category also were associated with greater reporting of any psychological hardship item. Factors associated with being sent to debt collection included lower income, Hispanic ethnicity, and non-medical/non-home debt (Table 4). Lacking a college degree was associated with having filed for bankruptcy. In multivariate logistic regression models with survivors’ disease- and treatment-related factors, cancer survivor-specific factors were not associated with financial hardship among siblings (Supplemental Table 4).

Table 3:

Associations of sibling-level characteristics and sibling report of any financial hardship, by financial hardship domains

Sibling characteristic Behavioral hardship
Odds ratioa (95% CI)		 Material hardship
Odds ratioa (95% CI)		 Psychological hardship
Odds ratioa (95% CI)
Age at questionnaire (years)
  18-29 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
  30-39 2.97 (0.99 – 8.93) 2.77 (1.06 – 7.25)* 0.99 (0.39 – 2.50)
  40+ 2.20 (0.73 – 6.66) 1.61 (0.61 – 4.23) 1.07 (0.42 – 2.72)
Sex
  Male 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
  Female 1.83 (1.21 – 2.77)* 1.80 (1.27 – 2.55)* 1.92 (1.28 – 2.86)*
Race and ethnicity
  White, Non-Hispanic 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
  Black, Non-Hispanic 0.15 (0.02 – 1.37) 1.17 (0.33 – 4.16) 0.31 (0.07 – 1.50)
  Hispanic/Latino 0.75 (0.23 – 2.48) 0.74 (0.28 – 1.95) 0.35 (0.08 – 1.47)
  Other 1.32 (0.61 – 2.87) 0.82 (0.40 – 1.65) 2.34 (1.14 – 4.81)*
Education
  <High school 0.32 (0.05 – 2.17) 5.82 (0.87 – 39.16) 1.09 (0.20 – 5.94)
  High school – Some college 0.66 (0.41 – 1.07) 1.02 (0.68 – 1.52) 0.96 (0.61 – 1.51)
  College graduate and or greater 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
Marital status
  Married 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
  Singled 0.95 (0.52 – 1.73) 0.60 (0.34 – 1.05) 1.71 (0.98 – 3.01)
  Divorced/Separated 1.37 (0.74 – 2.56) 0.77 (0.42 – 1.38) 1.48 (0.79 – 2.75)
  Widowed 1.72 (0.21 – 13.89) 0.47 (0.04 – 5.79) 0.40 (0.03 – 4.65)
Annual household income ($)
  $0-35,000 9.86 (4.53 – 21.43)* 1.87 (0.89 – 3.92) 11.86 (5.54 – 25.37)*
  $35,000-50,000 4.11 (1.93 – 8.74)* 3.07 (1.49 – 6.30)* 7.10 (3.42 – 14.74)*
  $50,000-75,000 4.75 (2.83 – 7.97)* 2.04 (1.26 – 3.32)* 6.61 (4.00 – 10.91)*
  ≥$75,000 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
Chronic health conditions (Grade 3+)b
  None 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
  ≥1 0.96 (0.50 – 1.85) 1.64 (0.94 – 2.88) 0.98 (0.52 – 1.85)
Insurance coverage
  None 4.73 (2.12 – 10.55)* 3.02 (1.35 – 6.72)* 1.36 (0.61 – 3.02)
  Private 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
  Medicare 0.41 (0.07 – 2.55) 0.68 (0.13 – 3.52) 1.00 (0.16 – 6.23)
  Medicaid or Indian Health Service 0.75 (0.26 – 2.20) 0.40 (0.13 – 1.23) 1.29 (0.43 – 3.87)
  Other 1.08 (0.21 – 5.51) 0.95 (0.19 – 4.66) 1.75 (0.37 – 8.22)
Out-of-pocket expenses
  <10% of annual income 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
  ≥10% of annual income 2.74 (1.03 – 7.29)* 5.23 (1.90 – 14.37)* 1.06 (0.41 – 2.76)
Non-home/non-medical debt
  None 1.00 (Ref) 1.00 (Ref) 1.00 (Ref)
  $1 – $25,000 2.52 (1.45 – 4.39)* 3.09 (1.93 – 4.94)* 2.21 (1.31 – 3.72)*
  $25,001 – $50,000 5.81 (3.14 – 10.75)* 5.89 (3.45 – 10.06)* 4.01 (2.20 – 7.28)*
  $50,001 – $ 100,000 3.50 (1.68 – 7.26) 4.47 (2.43 – 8.22)* 3.68 (1.83 – 7.42)*
  $100,001 – $250,000 2.23 (0.80 – 6.21) 8.04 (3.51 – 18.42)* 6.83 (2.80 – 16.64)*
  $250,001 – $500,000 4.43 (0.86 – 22.79) 8.96 (1.87 – 42.89)* 7.92 (1.54 – 40.61)*
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*

Statistically significant with p<0.05.

a

Models were adjusted for the variables included in the table.

b

Defined as “severe or disabling”

Table 4:

Associations between sibling-level characteristics and sibling report of being sent to debt collection or filing for bankruptcy

Sibling characteristic Sent to debt collection
Odds ratioa (95% CI)		 Filed for bankruptcy
Odds ratioa (95% CI)
Age at questionnaire
  18-30 1.00 (Ref) 0.00 (0.00 – 1.16)
  30-40 3.52 (0.89 – 13.96) 1.00 (Ref)
  40+ 3.38 (0.86 – 13.35) 1.87 (0.86 – 4.10)
Sex
  Male 1.00 (Ref) 1.00 (Ref)
  Female 1.36 (0.88 – 2.08) 1.56 (0.83 – 2.93)
Race and ethnicity
  White, Non-Hispanic 1.00 (Ref) 1.00 (Ref)
  Black, Non-Hispanic 2.90 (0.77 – 10.95) 1.98 (0.37 – 10.51)
  Hispanic/Latino 4.85 (1.78 – 13.19)* 1.57 (0.41 – 6.04)
  Other 1.06 (0.47 – 2.40) 0.66 (0.18 – 2.46)
Education
  <High school 4.84 (0.39 – 59.70) 0.00 (0.00 – 3.86)
  High school – Some college 1.42 (0.89 – 2.29) 2.87 (1.52 – 5.42)*
  College graduate and or greater 1.00 (Ref) 1.00 (Ref)
Marital status
  Married 1.00 (Ref) 1.00 (Ref)
  Singled 0.55 (0.26 – 1.13) 0.66 (0.20 – 2.25)
  Divorced/Separated 1.12 (0.58 – 2.16) 0.77 (0.30 – 1.94)
  Widowed 0.00 (0.00 – 1.42) 0.00 (0.00 – 14.15)
Annual household income ($)
  0-35,000 12.24 (4.88 – 30.71)* 1.51 (0.48 – 4.79)
  35,000-50,000 3.56 (1.57 – 8.05)* 0.64 (0.16 – 2.54)
  50,000-75,000 2.45 (1.40 – 4.29)* 1.77 (0.81 – 3.84)
  >=75,000 1.00 (Ref) 1.00 (Ref)
Chronic health conditions (Grade 3+)b
  None 1.00 (Ref) 1.00 (Ref)
  1 or more chronic conditions 0.87 (0.43 – 1.74) 0.93 (0.37 – 2.31)
Insurance coverage
  None 1.44 (0.58 – 3.60) 2.17 (0.68 – 6.99)
  Private 1.00 (Ref) 1.00 (Ref)
  Medicare 0.30 (0.05 – 1.87) 0.77 (0.07 – 8.74)
  Medicaid or Indian Health Service 1.59 (0.45 – 5.63) 1.08 (0.20 – 6.00)
  Other 2.08 (0.25 – 17.50) 3.52 (0.49 – 25.46)
Out-of-pocket expenses
  <10% of annual income 1.00 (Ref) 1.00 (Ref)
  ≥10% of annual income 1.14 (0.37 – 3.49) 2.77 (0.78 – 9.81)
Non-home/non-medical debt
  None 0.00 (0.00 – 0.01)* 0.00 (0.00 – 0.09)*
  $1 – $25,000 1.00 (Ref) 1.00 (Ref)
  $25,001 – $50,000 1.90 (1.16 – 3.11)* 1.79 (0.90 – 3.53)
  $50,001 – $ 100,000 1.58 (0.86 – 2.91) 0.73 (0.25 – 2.09)
  $100,001 – $250,000 1.39 (0.57 – 3.38) 1.09 (0.29 – 4.15)
  $250,001 – $500,000 1.30 (0.23 – 7.30) 0.00 (0.00 – 7.03)
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*

Statistically significant with p<0.05.

a

Models were adjusted for the variables included in the table.

b

Defined as “severe or disabling”

DISCUSSION

In this cross-sectional analysis of almost 1,000 adult siblings of long-term childhood cancer survivors, we observed that siblings reported financial hardship across multiple domains and measures, and had greater odds of reporting several aspects of financial hardship compared with the US general population. Several sibling-level characteristics were associated with reporting financial hardship, including female sex, older age, lower income, lack of health insurance coverage, chronic medical conditions, and greater non-medical/non-home debt. Characteristics of the survivors themselves related to cancer and treatment exposures were not associated with siblings’ report of financial hardship as adults.

The mechanisms contributing to siblings’ increased financial hardship compared with the general population remain unclear. One possibility could be the lingering adverse financial effects experienced by families and households following cancer treatment such as loss of educational opportunities, reduced transfer of financial resources from parents to children, and/or sequelae from other adverse household-level effects like material hardship. Alternatively, survivors’ ongoing caregiving/support needs,37 or siblings’ heightened sensitivity to financial challenges after witnessing intensive treatment for a chronic medical condition, could potentially contribute to the observed associations. Although we did not observe any clear associations between survivors’ cancer/treatment characteristics and their siblings’ report of financial hardship, it could be possible that the general process of cancer diagnosis and treatment itself, rather than specific individual treatment exposures, are associated with family economic impact. There may also be other contributing factors unaccounted for by this analysis. Thus, our findings underscore a critical need for a more in-depth exploration of contributors to long-term sibling and familial hardship, including longitudinal research to better examine trajectories of financial hardship among families following a childhood cancer diagnosis.

Our examination of financial hardship among siblings is novel. Although financial hardship is common among long-term survivors of childhood cancer,6,23–25,38 these findings of higher odds of hardship among adult siblings versus the general adult population adds to accumulating research examining the economic effects of a cancer diagnosis on family members. Several studies have reported declines in parental income and employment during treatment and survivorship.17,18,39,40 Additionally, families typically experience increases in non-healthcare out-of-pocket spending that exceed what is reported among adults with cancer,21,22 and report increased rates of material hardship after starting therapy that concerningly continue after treatment is complete.41,42 Siblings of childhood cancer survivors may experience important psychosocial stressors that can affect their own education, career choices, and future finances, and thus, greater attention must be given to supporting entire families during treatment and into survivorship. Prior CCSS evaluations of financial hardship among childhood cancer survivors utilized a comparison to nearest-age siblings as controls.6,23,38 However, it is also conceivable if siblings of children with cancer also experience long-term financial effects, that prior comparisons between survivors and siblings underestimate differences between cancer survivors and the general population.

Our results highlight a need for psychosocial and financial support for families across the trajectory of cancer care, and may suggest that interventions to address financial hardship should not only target individual patients, but their family units.16 Importantly, the negative financial consequences from cancer experienced by patients themselves may be modifiable,43 suggesting that mitigation strategies may have positive effects on family members, including siblings. Regular screening and measurement of financial hardship for survivors and their families, along with the development of the practical clinical tools necessary to do so, will be critical to better characterize the degree of family burdens longitudinally and develop strategic solutions, both during treatment and survivorship.3,44 Although patient-reported instruments measuring the negative consequences of cancer care have been developed and validated for adults with cancer,45,46 adaptations of these instruments or the development of outcome measures more specific to the pediatric oncology context may more effectively evaluate childhood cancer survivors and their family members.27 Potential interventions could be implemented during treatment and/or survivorship, at the patient-, family-, organizational-, or policy-level. Patient- and family-level interventions could include the more aggressive provision of financial resources to families experiencing high degrees of hardship.47 Organizational-level interventions could include strategies to better connect patients and families with existing resources or assist in navigating complex system and insurance needs.48 Policy-level interventions would likely require state or national legislation, and could include increasing support for workplace accommodations to maintain income during treatment and survivorship, offering more substantial extended sick leave for caregivers to promote the care of siblings and other children in the household, and providing resources to assist childhood cancer survivors with financial hardship and support whole family well-being.19,20

This study has important limitations. First, our comparison population was drawn from US national surveys that were intended to be representative of the adult general population and thus incorporate purposeful oversampling and sample weighting techniques to account for demographics and non-response characteristics. The CCSS sibling cohort differed significantly from the US general population by several key sociodemographic factors, including race and ethnicity, sex, education, and income. While we adjusted for these sociodemographic factors to compensate for these differences in our analysis, there may still be unmeasured confounding that biases the results. Similarly, our aggregation of some race categories in our models, due to relatively small sample sizes within these groups, is another limitation to our analysis that may limit generalizability. Next, differences in survey administration between the CCSS and NHIS/BRFSS could contribute to differences in responses, even to items that were identical or near-identical. Finally, this analysis is cross-sectional and cannot make inferences regarding causality, describe temporal trends, nor explain the mechanisms by which siblings of childhood cancer survivors may be experience financial hardship.

CONCLUSION

In summary, our study found that siblings of adult survivors of childhood cancer had a greater likelihood of reporting financial hardship compared with US adults in the general population. Several sibling-level factors, though not survivor characteristics, were associated with greater likelihood of reporting financial hardship. Increased screening and support for family members across the trajectory of cancer treatment may be a potential first step to help prevent or reduce financial hardship. To accomplish this, reliable screening and targeted interventions will be needed.

Supplementary Material

Fig S1

Supplemental Figure 1

CONSORT diagram depicting selection of general population respondents included for analysis. (A) National Health Interview Survey (NHIS) participants. (B) Behavioral Risk Factor Surveillance System (BRFSS) participants.

Tab S1
Tab S2
Tab S3
Tab S4
Tab S5

Acknowledgements:

This work was supported by the National Cancer Institute (CA55727, G.T. Armstrong, Principal Investigator). Support to St. Jude Children’s Research Hospital was also provided by the Cancer Center Support (CORE) grant (CA21765, C. Roberts, Principal Investigator) and the American Lebanese-Syrian Associated Charities (ALSAC). This work was also supported by a CCSS Trainee Career Development Award awarded to T. Ohlsen.

Conflict of interest statement:

K. Robin Yabroff reports consulting fees from Flatiron Health unrelated to this work. Rena Conti reports research support from Arnold Ventures and consulting fees from Greylock McKinnon Associates unrelated to this work. Eric Chow has received grant funding from Abbott Laboratories unrelated to this work. Zhiyuan Zheng and K. Robin Yabroff are employed by the American Cancer Society, which receives grants from private and corporate foundations, including foundations associated with companies in the health sector, for research outside the submitted work. All other authors declare no conflicts of interest.

Data sharing statement:

The Childhood Cancer Survivor Study (CCSS) is an NCI-funded resource to promote and facilitate research among long-term survivors of cancer diagnosed during childhood and adolescence. CCSS data are publicly available on dbGaP at https://www.ncbi.nlm.nih.gov/gap/ through its accession number phs001327.v2.p1. In addition, utilization of the CCSS data that leverages the expertise of CCSS Statistical and Survivorship research and resources will be considered on a case-by case basis. Details on data access may be found at: https://ccss.stjude.org/.

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Associated Data

This section collects any data citations, data availability statements, or supplementary materials included in this article.

Supplementary Materials

Fig S1

Supplemental Figure 1

CONSORT diagram depicting selection of general population respondents included for analysis. (A) National Health Interview Survey (NHIS) participants. (B) Behavioral Risk Factor Surveillance System (BRFSS) participants.

NIHMS1947538-supplement-Fig_S1.pdf (56.1KB, pdf)
Tab S1
NIHMS1947538-supplement-Tab_S1.docx (18.9KB, docx)
Tab S2
NIHMS1947538-supplement-Tab_S2.docx (15KB, docx)
Tab S3
NIHMS1947538-supplement-Tab_S3.docx (17.8KB, docx)
Tab S4
NIHMS1947538-supplement-Tab_S4.docx (18.9KB, docx)
Tab S5
NIHMS1947538-supplement-Tab_S5.docx (19.1KB, docx)

Data Availability Statement

The Childhood Cancer Survivor Study (CCSS) is an NCI-funded resource to promote and facilitate research among long-term survivors of cancer diagnosed during childhood and adolescence. CCSS data are publicly available on dbGaP at https://www.ncbi.nlm.nih.gov/gap/ through its accession number phs001327.v2.p1. In addition, utilization of the CCSS data that leverages the expertise of CCSS Statistical and Survivorship research and resources will be considered on a case-by case basis. Details on data access may be found at: https://ccss.stjude.org/.

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