Product information and disease description Place of product in therapy Disease description Burden of RAF-altered pediatric low-grade glioma requiring first-line systemic therapy

Economic impact of RAF-altered pediatric low-grade glioma requiring first-line systemic therapy on families

23 evidence topics · 7 sources

Summary: family financial burden and parental work loss

No study was identified that measures family financial burden specifically in pLGG. Evidence from pediatric brain tumors and pediatric cancer overall indicates the following. At a U.S. cancer center, 16% of 202 patients younger than 21 years who underwent neurosurgery for a cancer-related diagnosis had financial toxicity before surgery, and the 2-year cumulative incidence after surgery was 19%; among 86 patients who survived at least 2 years, 34% experienced financial toxicity, and most affected patients had commercial insurance. In a U.S. single-site survey of 254 caregivers of children with cancer, approximately one third reported that a parent quit or changed work because of the child's cancer, and this was associated with a 13.4-point higher financial burden score (0 to 100 scale) at 1 to 5 years after diagnosis. A systematic review of 35 studies reported frequent parental job loss or job quitting, particularly among mothers, with effects persisting into early survivorship. In a Canadian cohort of 28 families, median first-year indirect costs were $9,668 and median income loss was 55 days. In the Childhood Cancer Survivor Study (3,023 adult survivors; 16% with CNS tumors), survivors with 4 impaired neurocognitive domains had odds ratios of 3.12 for debt collection and 3.77 for bankruptcy compared with survivors with no impaired domains.

Financial toxicity in pediatric and adolescent neurosurgical oncology (United States)

“A retrospective review of medical and financial services data was conducted at Memorial Sloan Kettering Cancer Center of patients under 21 years of age who underwent neurosurgery for a cancer-related diagnosis from 2016 to 2020.” (opens the source at this quote in a new tab)
“294 surgical procedures across 202 patients were analyzed. 16% (33/202) of patients experienced FT in the two years prior to the first neurosurgical procedure, and the most common toxicity identifier was utilization of the financial assistance program (45%). For all 202 patients, the 2-year cumulative incidence of FT following first neurosurgical procedure was 19% (CI:14%,24%). Among patients who survived for at least 2 years after surgery, 34% (29/86) experienced FT with the most common toxicity being having bills sent to collections (34%). Most patients who experienced FT had at least one commercial insurance policy.” (opens the source at this quote in a new tab)

Parental employment disruption and perceived financial burden in pediatric cancer (United States)

“Eligible patients were treated at a pediatric cancer hospital, diagnosed at age ≤ 21 years and were ≤ 5 years from diagnosis (N = 254). Financial burden was rated on a visual analog scale of 0 to 100.” (opens the source at this quote in a new tab)
“Approximately one third of caregivers in both groups reported a parent quitting or changing work as a result of their child's cancer diagnosis.” (opens the source at this quote in a new tab)
“When adjusted for sex, age at diagnosis, insurance status, and rural residence, caregivers whose child was 1 to 5 years from diagnosis with ≥ 5 unexpected hospitalizations experienced 24.9 (95% CI, 9.1 to 40.7; P < .01) points higher financial burden than those with no unexpected hospitalizations. In addition, when compared with families without employment disruptions, families of children 1 to 5 years from diagnosis in which a caregiver had quit or changed jobs reported 13.4 (95% CI, 3.2 to 23.6; P = .01) points higher financial burden.” (opens the source at this quote in a new tab)

Systematic review: impact of childhood cancer on parents' socio-economic situation

“Our systematic review included 35 eligible articles. Childhood cancer had a substantial impact on parents' socio-economic situation across all studies. This impact varied largely by geographical region. We observed a high prevalence of disruptions in parental employment such as job quitting or job loss, particularly among mothers. The associated income losses further contributed to families' perceived financial burden in addition to increased cancer-related expenses. Adverse socio-economic consequences were most pronounced shortly after diagnosis, however, persisted into early survivorship for certain groups of parents.” (opens the source at this quote in a new tab)

Parents' employment after completion of childhood cancer treatment

“We interviewed 56 parents of childhood cancer survivors (M = 2.13 years after treatment completion; 89% mothers) using the vocational and financial impact section of the Psychosocial Adjustment to Illness Scale-Carer Interview Form.” (opens the source at this quote in a new tab)
“Parents reported multiple sources of financial toxicity including travel to and from the hospital and needing to reduce their working hours during their child's cancer treatment. Workplace flexibility was an important factor to protect against unwanted vocational changes. After treatment completion, families living in low socioeconomic areas commonly reported ongoing financial difficulties.” (opens the source at this quote in a new tab)

Indirect costs and income loss in the first year of childhood cancer treatment (Canada)

“Thirty families participated in the study, with 28 providing financial information and 29 participating in interviews. Financial information was collected during the first year of treatment.” (opens the source at this quote in a new tab)
“Families spent a median of $9668 (IQR $7944-12958) on indirect costs and had a median of 55 (IQR 0-125) days of income lost. Most families (79%) in our study experienced a psychosocial burden using terms like stress, worry, or frustration directly as a result of the financial burdens from cancer treatment, in addition to the already significant anxiety around having a child with cancer.” (opens the source at this quote in a new tab)

Neurocognitive late effects and financial hardship in adult survivors of childhood cancer (Childhood Cancer Survivor Study)

“The analytic cohort comprised 3023 adult (median age at financial hardship survey 40.5 [IQR 35.0-47.8] years) survivors of childhood cancer with the most common diagnoses being leukemia (31%), central nervous system tumor (16%), and Hodgkin lymphoma (13%).” (opens the source at this quote in a new tab)
“Individuals with 4 impaired neurocognitive domains had over 3-fold increased odds of debt collection (OR=3.12, 95% CI=1.91-5.09) and bankruptcy (OR=3.77, 95% CI=2.01-7.07) compared to individuals with no impaired neurocognitive domains.” (opens the source at this quote in a new tab)

Out-of-pocket costs and parental work loss specific to pLGG or to oral MAPK inhibitor versus intravenous chemotherapy

No evidence found.

Definition of financial toxicity and insurance coverage in the neurosurgical oncology cohort

“Financial toxicity was defined as meeting one or more of the following criteria: bankruptcy, bills sent to collections, settlement on medical bills, participation in a timed payment plan, copay assistance program use, financial assistance program use, social work philanthropy use and expression of financial concerns to social work.” (opens the source at this quote in a new tab)
“The median ZIP-code income, which is the median income living within a zip code, for these patients’ families was $88,636 (range $ 65,101, $116,191). 94% of patients had some type of insurance coverage with 60% of these insured patients being covered by private insurance and 33% by public insurance.” (opens the source at this quote in a new tab)

Types of financial toxicity and predictors of post-operative financial toxicity

“The most common toxicities were utilization of a financial assistance program (45%), use of social work philanthropy fund (30%), and having bills sent to collections (24%)” (opens the source at this quote in a new tab)
“The most common toxicities for patients surviving at least 2 years after last neurosurgical procedure were having bills sent to collections (34%), utilization of the financial assistance programs (31%), and use of the social work philanthropy fund (28%) (Table 2). Among these patients experiencing FT, 69% had at least one commercial insurance policy.” (opens the source at this quote in a new tab)
“In a univariate regression model, only living within the Tri-state area was significantly predictive of developing post-operative FT (p = 0.037) compared to living outside the Tri-state area (Table 3). Age at first procedure, sex, living in New York versus outside of NY, race, type of insurance, and diagnosis (neuroblastoma vs. diffuse midline glioma (DMG) vs. other) were not significantly associated with developing post-operative FT.” (opens the source at this quote in a new tab)

Proportion of parents who reduced or left employment during their child’s treatment

“Thirty-eight parents (67.9%) reported experiencing disruption to their employment while their child was on treatment, which is encapsulated in two subcategories.” (opens the source at this quote in a new tab)
“Twenty-nine parents (51.8%) reported that they and/or their partner reduced their hours or resigned from their employment after their child’s diagnosis. Participants and their partners reduced working hours by taking unpaid leave (n = 13, 23.2%), taking extended paid leave, including sick and long-service leave (n = 5, 8.9%), changing from full-time to part-time or casual employment (n = 6, 10.7%), changing their self-employed working hours (n = 3, 5.4%), resigning from their employment (n = 12, 21.4%), or a combination of these.” (opens the source at this quote in a new tab)

Medical appointments as the leading cause of ongoing employment disruption after treatment

“Two-thirds of parents (n = 38, 67.9%) reported that their child’s ongoing cancer-related needs caused disruptions to their employment, barriers to reentering the workforce, or barriers to increasing their working hours to full-time employment. The most frequent disruptions were medical appointments (n = 30, 53.6%) and their child’s ongoing health needs (n = 21, 37.5%; e.g., child being too unwell to attend day-care or school).” (opens the source at this quote in a new tab)

Persistence of family financial toxicity after treatment completion

“Only five parents (8.9%; of whom four had an SES decile greater than six and one was unknown) reported that at the time of the study, their financial toxicity related to their child’s cancer treatment had resolved.” (opens the source at this quote in a new tab)

Family costs as a proportion of family income in a critical review of cost of illness studies

“The major components of family’s direct costs were travel, food, and child care.” (opens the source at this quote in a new tab)
“Families’ costs, in relation to family income, ranged from 0.2% to 283.1% in the US” (opens the source at this quote in a new tab)
“During the first hospitalization of insured Mexican children diagnosed with leukemia, parents’ out-of-pocket costs and lost wages were US$ 679 (range US$ 127–1,882) exceeding the total household income for nearly half the families” (opens the source at this quote in a new tab)

Methodological limits of the family cost of illness evidence base

“Existing research indicates that families incur significant variable costs throughout cancer treatment. However, problems with the published studies related to various PQAQ domains (e.g., time horizon, sensitivity analysis) rendered the magnitude of families’ childhood cancer costs somewhat uncertain.” (opens the source at this quote in a new tab)
“All data in the reviewed studies were collected within a 1-year time frame.” (opens the source at this quote in a new tab)
“None of the researchers examined how sensitive their results were in relation to the estimates used for particular variables or underlying assumptions” (opens the source at this quote in a new tab)

Scope of the systematic review and timing of employment and income disruption

“Thirteen (37%) studies were conducted in Europe, 16 (46%) in North America/Australia, and 6 (17%) in Asia/Africa (Table 1). Eight (23%) studies included comparison parents. The majority of studies (85%) included different cancer types.” (opens the source at this quote in a new tab)
“Twenty-six (74%) studies reported on employment, 20 (57%) on income, 21 (60%) on financial situation, and 20 (57%) addressed financial assistance (Table 2).” (opens the source at this quote in a new tab)
“Most work disruptions occurred shortly after diagnosis and attenuated within one year” (opens the source at this quote in a new tab)

Financial toxicity before surgery as a predictor of financial toxicity afterwards

“Patients experiencing preoperative FT within 2 years of their first neurosurgical procedure had a statistically significantly higher cumulative incidence of post-operative FT (2-year estimate: 61% (95% CI: [41%,75%]) compared to their counterparts who did not experience pre-operative FT (2-year estimate: 11% (95% CI: [7%,16%]) (p < 0.001).” (opens the source at this quote in a new tab)
“This establishes the critical need for assessment and intervention for those at risk of FT prior to meeting criteria and longitudinally throughout treatment, to help decrease the initial and cumulative incidence of FT.” (opens the source at this quote in a new tab)

Caregiver work loss and cost-related medication nonadherence in adolescents and young adults

“In addition, working caregivers often need to take significant time away from work, utilizing unpaid leave, sick leave, a leave of absence, or quitting their job. This caregiver inability to work leads to reduced income and halted career progression” (opens the source at this quote in a new tab)
“As the patients age into adolescence and young adulthood, they become more likely to report cost-related nonadherence to medication, inability to afford medication, ask physicians for a lower cost medication, and use alternative therapies compared to a non-cancer control group of similar ages” (opens the source at this quote in a new tab)

Why the measured financial toxicity rate is likely an underestimate

“A notable limitation to this manuscript is that the data are derived from a quaternary cancer care center without a public emergency department. The rates of FT reported are likely lower than in the community due to the inherent barriers involved with navigating the health care system to receive care at a quaternary cancer care center.” (opens the source at this quote in a new tab)
“Indirect costs are often not captured by hospital financial service departments, and prospectively partnering with patient families is crucial to understanding the impact of indirect costs associated with pediatric neurosurgical oncology.” (opens the source at this quote in a new tab)

Out-of-pocket cost categories and the supports families relied on

“Thirteen parents (23.2%) discussed the factors that directly contributed to their financial toxicity after their child’s diagnosis. Major contributors during their child’s treatment included parking fees, petrol (fuel), and other expenses related to regularly traveling to and from the treating hospital (e.g., road tolls).” (opens the source at this quote in a new tab)
“Twenty-one parents (37.5%) discussed factors that helped protect their family from financial toxicity after their child’s diagnosis and throughout treatment. These included loans, donations, or gifts from friends and family, charitable support (e.g., free accommodation near their child’s treating hospital and petrol/fuel vouchers), government payments (i.e., welfare payments), and receiving paid leave from their employment.” (opens the source at this quote in a new tab)

Employment effects persisting years after treatment completion

“Nine mothers (16.1%) reported that their child’s ongoing cancer-related needs continued to affect their employment, an average 3.4 years after treatment completion (SD = 3.78). Mothers reported that they accepted lower-paid roles compared with roles prior to their child’s diagnosis, chose part-time or casual employment when they had previously been employed full-time, or chose to leave their employment altogether to become stay-at-home mothers despite expressing their desire to work.” (opens the source at this quote in a new tab)

Thresholds for judging family cost burden

“Lansky and coworkers indicated that the direct costs plus loss of pay were, on average, considerably more than 15%. For half of their sample, the figure was over 25%.” (opens the source at this quote in a new tab)
“According to the studies reviewed, families of children with cancer are experiencing an unfair burden regardless of whether the threshold is 5% or 15%.” (opens the source at this quote in a new tab)

Factors that drive family costs and resource use

“Presently, socioeconomic status, distance from hospital, cancer type, and length of hospitalization have been reported to significantly influence families’ costs and resource use.” (opens the source at this quote in a new tab)
“Despite the high likelihood of cure, the costs families incur over the course of treatment may be unsustainable and lead to considerable stress and financial hardship” (opens the source at this quote in a new tab)

Caregiving time required and families at particular risk

“A Swiss study estimated that parents need on average 240 working days for caretaking during the child´s treatment” (opens the source at this quote in a new tab)
“Also in the US, a study showed that 15% of families fell below the poverty level due to cancer-related financial strains” (opens the source at this quote in a new tab)
“We identified families with lower socioeconomic position, parents of children diagnosed with haematological cancers and diagnosed at younger age to be at particular risk for adverse socio-economic consequences.” (opens the source at this quote in a new tab)