Financial costs of families in childhood cancer care: an integrative review

Abstract

Objective

To analyze evidence on the financial costs of families caring for children and adolescents during cancer treatment.

Methods

Integrative review with searches conducted in the PubMed, CINAHL, Scopus, Web of Science, and EMBASE databases. Articles published in Portuguese, Spanish and English, in the last ten years, with different study designs and that addressed the topic, were considered. For the searches, the controlled descriptors MeSH were used, for the titles CINAHL and EMTREE, their synonyms and keywords, linked by Boolean operators AND and OR.

Results

Twenty-two articles were included. The results were described in the following categories: Direct costs (medical and non-medical); and Indirect costs (parental employment status and family income). Medical costs are those associated with hospitalizations, treatment and diagnosis. The main elements responsible for non-medical expenses were travel, food and accommodation. The impact on families’ employment situation is a consequence of the high demand for care imposed on caregivers for childhood cancer treatment. As a result of the change in work situation, families need to deal with the risk and consequences of a decrease or loss of income.

Conclusion

The results indicate that families bear direct and indirect medical expenses as well as non-health-related costs resulting from the disease. Furthermore, they are affected by indirect costs, such as lost productivity, impact and changes in employment, which significantly reduces income.

Family; Child; Adolescent; Neoplasms; Health care costs; Cost of ilness

Resumo

Objetivo

Analisar evidências sobre os custos financeiros de famílias no cuidado de crianças e adolescentes durante o tratamento oncológico.

Métodos

Revisão integrativa. As buscas foram realizadas nas bases de dados: PubMed, CINAHL, Scopus, Web of Science e EMBASE. Foram considerados artigos publicados em português, espanhol e inglês, nos últimos dez anos, com diferentes desenhos de estudo e que abordassem a temática. Para as buscas, foram utilizados os descritores controlados MeSH, dos títulos CINAHL e EMTREE, seus sinônimos e palavras-chave, interligados pelos operadores booleanos AND e OR.

Resultados

Foram incluídos 22 artigos. Os resultados foram descritos em categorias: Custos diretos (médicos e não médicos) e indiretos (condição laboral parental e renda familiar). Os custos médicos são aqueles associados a hospitalizações, tratamento e diagnóstico. Os principais elementos responsáveis pelas despesas não médicas foram viagens, alimentação e acomodação. O impacto na situação laboral das famílias é consequência da alta demanda de cuidados imposta aos cuidadores para o tratamento do câncer infantojuvenil. Em decorrência da mudança na situação de trabalho, as famílias precisam lidar com o risco e desdobramentos da diminuição ou perda de renda.

Conclusão

Os resultados indicam que as famílias assumem despesas médicas diretas e indiretas, bem como custos não relacionados à saúde decorrentes da doença. Além disso, são afetadas por custos indiretos, como perda de produtividade, impacto e mudanças no emprego, o que reduz significativamente a renda.

Família; Criança; Adolescente; Neoplasias; Custos de cuidados de saúde; Custo da doença

Resumen

Objetivo

Analizar evidencias sobre los costos financieros de familias para el cuidado de infantes y adolescentes durante tratamientos oncológicos.

Métodos

Revisión integradora. Las búsquedas se realizaron en las siguientes bases de datos: PubMed, CINAHL, Scopus, Web of Science y EMBASE. Se consideraron artículos publicados en portugués, español e inglés, en los últimos diez años, con diferentes diseños de estudio, que trataran sobre el tema. Para las búsquedas se utilizaron los descriptores controlados MeSH, de los títulos CINAHL y EMTREE, sus sinónimos y palabras clave, conectados mediante los operadores booleanos AND y OR.

Resultados

Se incluyeron 22 artículos. Los resultados fueron separados en categorías: costos directos (médicos y no médicos) e indirectos (condición laboral parental e ingresos familiares). Los costos médicos son aquellos relacionados con hospitalizaciones, tratamiento y diagnóstico. Los principales elementos responsables de los gastos no médicos fueron viajes, alimentación y hospedaje. El impacto en la situación laboral de las familias es consecuencia de la alta demanda de cuidados impuesta a los cuidadores para el tratamiento del cáncer infantojuvenil. Como consecuencia de los cambios de la situación laboral, las familias tienen que lidiar con el riesgo y los efectos de la reducción o pérdida de ingresos.

Conclusión

Los resultados indican que las familias enfrentan gastos médicos directos e indirectos, así como también costos no relacionados con la salud resultantes de la enfermedad. Además, se ven afectadas por costos indirectos, como pérdida de productividad, impacto y cambios en el trabajo, lo que reduce significativamente los ingresos.

Familia; Niño; Adolecente; Neoplasías; Costos de la Atención en Salud; Costo de enfermedad

Introduction

Childhood cancer accounts for 1 to 4% of all malignant neoplasms. Worldwide, it is estimated that around 300,000 children and adolescents, aged between zero and 19, are diagnosed with cancer each year.¹ In Brazil, the estimate for the number of new cases of childhood cancer, for each year of the 2023-2025 triennium, is 4,230 in males and 3,700 in females, with the median percentage of childhood tumors equivalent to 3% of total cases in the Brazilian population.² In Brazil, cancer accounts for the first cause of death (8%) due to disease in children and adolescents aged 1 to 19 years.²

Cancer diagnosis in a child or adolescent affects family dynamics systemically, which culminates in repercussions that weaken and make vulnerable both the child or adolescent who becomes ill and their parents.³-⁵ Parents are often involved in providing and monitoring treatment that includes long periods of inpatient and outpatient follow-ups, even years after the end of this treatment.⁶-⁸ Managing childhood cancer treatment is a challenge in parents’ lives and even has repercussions on their professional lives. Evidence from the literature shows an increase in the number of job abandonment and a decrease in family income during the acute care of a child with cancer.⁹,¹⁰ Furthermore, parents are faced with increasing medical and non-medical expenses.¹¹

With regard to costs for families, they can be divided into direct, indirect and psychosocial.⁹,¹² The former include out-of-pocket expenses related to diagnosis, treatment, supportive care, rehabilitation and general care. Indirect costs include loss of productivity due to work and impact the reduction of family income and allocation of other assets to treat the illness of sick children.⁹,¹² Despite the importance of the topic, only three reviews are currently available in the literaturem and these bring together studies published before 2018, predominantly in the decade 2000 to 2010.⁹,¹¹,¹³Additionally, such reviews used different methodological approaches, which often did not distinguish direct costs (medical and non-medical) and indirect costs (loss of productivity and income). The reviews in question concluded that included studies provided evidence of financial toxicity in pediatric oncology.⁹ Childhood cancer had a substantial impact on parents’ socioeconomic situation, as, after diagnosis, they experienced a wide range of adverse socioeconomic consequences.¹¹Families also bear direct costs for extra travel, food and childcare, with significant consequences for their lifestyle.⁹

In this context, the present review seeks to fill this gap in knowledge and provide updated evidence for healthcare professionals and decision makers about the economic burden imposed on families of children and adolescents by cancer treatment. Thus, it aims to analyze evidence on the financial costs of families caring for children and adolescents during cancer treatment.

Methods

This is an integrative review, whose development was based on the Whittemore and Knafl approach, which includes five steps: (1) problem identification; (2) literature search; (3) data quality assessment; (4) data analysis; and (5) presentation of results.¹⁴

The following research question guided the review: What knowledge is produced in the scientific literature about the financial costs of families caring for children and adolescents during cancer treatment? The PCO tool (Population/Population; Context/Context; Outcomes/Result) was used to outline the search strategy.¹⁵ The searches were carried out in the PubMed (US National Library of medicine), CINAHL (Cummulative Index of Nursing and Allied Health Literature), Scopus (Elsevier) Web of Science Core Collection (Clarivate Analytics) and EMBASE (Excerpta Médica Database) databases. Chart 1 presents the search matrix that was developed according to the PubMed model, which was adapted for each database, according to the specificities of each one. For the searches, the controlled descriptors MeSH were used, for the titles CINAHL and EMTREE, their synonyms and keywords, linked by Boolean operators AND and OR.

Chart 1
PubMed search strategy

Search strategy/PubMed
Population (Families of children and adolescents) #1	“Family”[Mesh] OR Families OR “Parents”[Mesh] OR Parent OR “Parents”[Mesh] OR Parent OR “Mothers”[Mesh] OR Mother OR “Fathers”[Mesh] OR Father
	“Child”[Mesh] OR children OR “Adolescent”[Mesh] OR Adolescents OR Adolescence OR Teens OR Teen OR Teenagers OR Teenager OR “Adolescents, Female” OR Adolescent, Female OR Adolescent OR “Female Adolescents” OR “Adolescents, Male” OR “Adolescent, Male” OR “Male Adolescent” OR “Male Adolescents”
Context (Childhood cancer) #2	“Neoplasms”[Mesh] OR Neoplasia OR Neoplasias OR Neoplasm OR Cancer OR Cancers OR Tumors OR Tumor OR Malignancy OR Malignancies OR “childhood cancer” OR “pediatric oncology” OR Cancer OR “pediatric cancer”
Outcome/result (Financial costs) #3	“Cost of Illness”[Mesh] OR “Illness Cost” OR “Illness Costs” OR “Sickness Cost” OR “Costs, Sickness” OR “Burden of Illness” OR “Illness Burden” OR “Illness Burdens” OR “Cost of Disease” OR “Economic Burden of Disease” OR “Disease Cost” OR “Cost, Disease” OR “Costs, Disease” OR “Disease Costs” OR “Cost of Sickness” OR “Sickness Costs” OR “Costs of Disease” OR “monetary costs” OR “Financial burden” OR “Economic effects” OR “economic impact” OR “travel expenses” OR “out-of-pocket expenses” OR “Hidden costs” OR “Economic hardship” OR “Non-Medical cost” OR “economic situation” OR “Employment”[Mesh] OR “family income” OR “financial toxicity”

Due to language limitations and budget restrictions for translation services, the search was carried out in English, Portuguese and Spanish. Furthermore, it was carried out by two independent reviewers and limited to studies published between January 1, 2011 and December 31, 2022 to ensure knowledge is updated, considering that previous literature reviews included studies predominantly from 2000 to 2010.⁹,¹¹,¹³

Original studies addressing financial costs (including direct and indirect costs) related to child and adolescent care, between zero and 19 years old, undergoing cancer treatment, from the family’s perspective, were included. By family, biological parents were considered those who took over parental roles through family reconstitution or any other person responsible for primary care in the family context. Original studies, regardless of the qualitative or quantitative methodological approach, were also included. Dissertations, theses, review, opinion and theoretical articles were excluded, as were studies that considered the perspective of healthcare professionals or those in conjunction with the family. Rayyan software was used to screen titles and abstracts found in database searches.¹⁶ Two independent reviewers (TFL; TGA) assessed the titles and abstracts. The level of agreement between the two was calculated according to Kappa, with a result of 0.83 (almost perfect agreement between the reviewers).¹⁷ Subsequently, eligible studies were read in full and separately by two reviewers (TFL; ML). In cases of disagreement, a third reviewer (RRN) was consulted. Finally, an unsystematic search was carried out in the list of references of articles already included in the review. The PRISMA flowchart was used to describe the search, identification and selection phases of studies, and the results are represented in Figure 1. Subsequently, the methodological quality of the primary studies included in the review was assessed using the checklists proposed by JBI¹⁸ and the Mixes Methods Appraisal Toll (MMAT) Version 2018 checklist.¹⁹ Qualitative, cross-sectional and cohort studies were assessed using specific instruments for each JBI method, and mixed studies were assessed using the MMAT instrument. Methodological quality assessment was carried out independently by two reviewers (TFL; TGA), and, in case of disagreements, a third reviewer (RRN) was consulted.

Figure 1
PRISMA(20) Flowchart representing the literature review process

Initially, two reviewers (TFL; RRN) extracted the following information from the included studies: name of authors, country and year of publication, objective, design, sample and main results. Subsequently, the extracted data was subjected to content analysis, which consisted of the following phases: (1) coding, when the data was read to identify significant words and categories and notes were written in the margins to describe all aspects of the content of interest contained in the data; (2) grouping, in which the codes previously collected were categorized and organized, considering category similarity and composition; (3) integration, phase in which the categories were integrated into larger topics, which allowed comparisons and observations on data that did not belong to the same group, in order to present relevant results for the study.²¹

Results

Characterization and quality assessment of studies included in the review

This integrative review included studies developed in 11 countries, such as Canada (n= 4), Sweden (n= 3), United States of America (USA) (n= 2), India (n= 4), China (n= 3), Japan (n= 1), Bangladesh (n= 1), Portugal (n= 1), Colombia (n = 1), Ghana (n= 1) and Denmark (n=1). As for the year of publication, one was published in 2012, three in 2013, two in 2014, two in 2015, one in 2016, two in 2017, two in 2018, three in 2019, five in 2020 and one in 2021. The full characterization of included studies and their quality assessment are presented in Chart 2.

Chart 2
Characterization of included studies

Author/year/country	Study objective	Study method	Sample characterization	Results	Quality assessment
Öhman, et al (2021).(22) Sweden	Investigate annual income adjusted for inflation, the employment rate of fathers and mothers and the number of fathers and mothers who received an inpatient diagnosis in the area of behavioral, emotional and/or mental disorders.	Cohort study. Data were obtained from the Swedish Childhood Cancer Registry, Intergenerational Registry and Inpatient Registry.	The sample consisted of 3865 fathers and 3865 fathers of 3865 children diagnosed with cancer. The five most common diagnoses were ALL, malignant cerebellar tumor, malignant brain tumor with unspecified location, Hodgkin’s lymphoma, and malignant kidney tumor.	The study showed that childhood cancer had short-term negative impacts on the income of fathers and mothers and long-term impacts on fathers’ income as well as short- and long-term impacts on their employment. It also had a long-term positive impact on mothers’ income and had no effect on the number of hospitalizations for mental and behavioral disorders for fathers or mothers.	10 of 11 Checklist for Cohort Studies JBI.
Mader, et al (2020).(23) Denmark	Assess the impact of childhood cancer on parents’ employment and income status and identify the determinants of adverse changes following a child’s cancer diagnosis.	National population-based cohort study based on Danish registry data. For statistical data analysis, logistic regression models were applied.	The study cohort included 12,418 parents of children with cancer and 125,014 comparison parents. The children’s cancer types were: lymphoid leukemia, other leukemias, lymphoma, central nervous system tumor, bone tumor and other solid tumor.	Mothers of children diagnosed in 1982-1999 were more likely to be not working or have low income than comparison mothers up to 10 years after diagnosis. This risk of not working or having low income was lower in mothers of children diagnosed in 2000-2014 compared to the previous period. Low parental education, lymphoblastic leukemia diagnosis, and younger age of children at diagnosis were the main determinants of changes in work or income status after diagnosis.	9 of 11 Checklist for Cohort Studies JBI
Tsimicalis, et al (2020).(24) Canada	Explore the financial impact of a new childhood cancer diagnosis on families and understand the strategies they adopt to manage these impacts.	Qualitative and descriptive study. Data were collected through semi-structured interviews and analysis was carried out through content analysis.	The sample consisted of 78 parents of children with cancer. The diagnoses were divided into leukemia, lymphoma, kidney tumors and central nervous system tumors.	To absorb expenses, families needed to increase their debts, at the same time as they looked for ways to take advantage of available resources, relying on their support networks and seeking financial help from philanthropy and government agencies.	8 of 10 Checklist for Qualitative Research JBI.
Jiang, et al (2020).(25) China	Assess the economic burden of treating neuroblastoma in children for the family, aiming to reduce patient-related expenses and promote the establishment of a disease management policy.	Quantitative and cross-sectional study. Data collected through a structured questionnaire consisting of 33 items. Statistical data analysis used the Statistical Package for the Social Sciences (SPSS) version 22.	The sample consisted of 122 families of children diagnosed with neuroblastoma.	The median direct medical costs of low risk (LR), medium risk (MR) and high risk (HR) during treatment were 180, 200 and 650 thousand Chinese yuan, respectively. Direct non-medical costs, including transportation, food and accommodation, were 60, 80 and 100.0 thousand Chinese yuan, considering the LR, MR and HR groups, respectively. Furthermore, the parents missed work to attend treatment and lost a total of 100,000 Chinese yuan in indirect costs.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Zhou, et al (2020).(26) China	Estimate the economic burden of pediatric patients with retinoblastoma (RB).	Quantitative and retrospective study. A telephone questionnaire was administered. Statistical analysis was performed using SPSS version 20.	The sample consisted of 50 parents of children diagnosed with RB.	The total direct cost was US$27,814.621 ± 15,137.73, and the mean medical cost was US$15,034.48 ± 8,224.19. The mean total non-medical expenses was US$12,252.93 ± 9,872.64. The mean time of reduced work was 11.50 ± 8.06 months, and the mean lost income reached US$13,512.23 ± 11,545.83. Families from other provinces (outside the capital) had higher expenses on transportation and accommodation.	4 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Dawson, et al (2020).(27) Ghana	Determine the economic costs and quality of life of primary caregivers of children with lymphoma.	Quantitative and cross-sectional study. The estimated costs were incurred by caregivers in the period of one month prior to data collection. Zarit Burden Interview was used to determine caregiver burden, and the EUROHIS-QoL tool to determine primary caregivers’ quality of life.	The sample consisted of 156 primary informal caregivers of children with lymphoma treated at the pediatric cancer unit at Komfo Anokye University Hospital.	The mean cost of treating lymphoma in children was estimated at US$440.32, 97% of which were direct costs. On average, caregiver burden was 26.3 on a scale of 0 to 48. About 94% of caregivers reported high burden (higher in men). Overall, the mean quality of life among caregivers was 2.20 in the range of 1 to 5. Approximately 85% of respondents reported low quality of life, with women reporting a lower quality of life than men.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Ren; Li, (2019).(28) China	Estimate direct and indirect costs in families with a child diagnosed with acute lymphoblastic leukemia (ALL).	Quantitative and cross-sectional study. Data were collected through a structured questionnaire. Data analysis was carried out using descriptive statistics.	The sample consisted of 161 parents of children diagnosed with ALL between 2010 and 2012.	Direct medical costs accounted for 51.7% of total costs, with the remaining 48.3% being direct non-medical and indirect costs. Urban families incurred more direct costs than rural families.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Mardakis, et al (2019).(29) India	Explore the cost experiences of caregivers of children undergoing pediatric cancer treatment.	Qualitative and descriptive study. Data were collected through a semi-structured interview consisting of six open questions. A thematic data analysis approach was carried out.	The sample consisted of 25 caregivers of 24 children, with variable diagnoses, with ALL being the most common.	The main sources of direct costs were: hospital admissions, medications, food and travel. The indirect ones involved managing the child’s treatment, affecting caregivers’ sleep, working hours and time spent with other family members. Psychosocial costs included dealing with the uncertainty caused by the diagnosis, feelings of guilt, and sadness.	8 of 10 Checklist for Qualitative Research JBI.
Ahuja, et al (2019).(30) India	Determine the feasibility of assessing direct costs incurred by families of children undergoing cancer treatment in public hospitals in India during two weeks before diagnosis and (b) 12 weeks after diagnosis.	Quantitative and prospective study. Data were collected from a basic questionnaire and a fortnightly cost record, which was completed by participants. Data analysis was carried out using descriptive statistics.	11 families of children with cancer who were undergoing treatment in the hospitals selected for the study participated. Cancer types included leukemia, neuroblastoma, non-Hodgkin lymphoma, bone sarcoma, and Wilms tumor.	Mean costs for the 14-week period were $764, with 47% in medical categories and 53% in non-medical categories. A third of these costs were borne by the NGO CanKids. Mean costs two weeks before diagnosis were $113, with 73% of costs in medical categories and 27% in nonmedical categories. Mean costs for 12 weeks after diagnosis were $651, with 42% costs in medical categories and 58% in non-medical categories.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Rativa Velandia, et al (2018).(31) Colombia	Determine the family economic burden associated with caring for a child with cancer.	Quantitative and descriptive study. Data were collected from a questionnaire. Analysis was carried out using the methodology “Economic burden attributable to the care of a person with a chronic illness in Colombia” (CARACOL, for the Spanish term).	The sample consisted of 50 families of children with cancer. The study did not specify the type of cancer diagnosed in the children.	The main expenses were transportation (28.5°),2 communications (26.3°),2 health (20.8°),2 housing (19.7°)2 and food (17.4°).2 The financial strain associated with caring for a child with cancer was a cause for concern for all caregivers (high 58%, mean 38% and low 4%). The costs of childcare are borne by several family members (36%), which is why they are forced to resort to debt or the sale of goods.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Marques, (2018).(32) Portugal	Identify the socioeconomic variables that influence families of children with cancer.	Quantitative, descriptive and correlational study. Data were collected using four instruments: “Questionnaire to Assess the Impact of the Children’s Cancer on the Family”, “Social Support Satisfaction Scale”, “Graffar Scale” and “Apgar Scale”.	128 families of children with cancer participated. The most frequent diagnosis was leukemia.	Economic costs increased due to the disease due to travel and medication, in addition to the loss of income of one of the parents. Families with greater support needs and less social support have a greater economic impact.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Lindahl Norberg, et al (2017).(33) Sweden	Investigate the short- and long-term effects of childhood cancer on the income and employment status of mothers and fathers.	National cohort study. Data related to employment income and employment status were obtained from the Longitudinal Integration Database of Health Insurance and Labor Market Studies. Using generalized linear models, comparison was made with a matched control cohort of 34,874 parents sampled from the general population.	The sample consisted of 1,899 families with a child diagnosed with cancer. Central nervous system tumor was the most common.	Parents’ income decreased after the child’s diagnosis. The effect was more pronounced for mothers, whose income was reduced for 6 years after diagnosis, whereas fathers’ income was similar to that of control parents for 3 years after diagnosis. Mothers were more likely to stop working after a cancer diagnosis compared to controls. Younger parental age, lower education level and, among mothers, being born outside Sweden were factors associated with more adverse effects on income.	8 of 11 Checklist for Cohort Studies JBI.
Sneha, et al (2017).(34) India	Know the details of the expenses paid by families during the treatment of children with cancer and their implications for quality of life.	Quantitative and cross-sectional study. A structured questionnaire developed from a literature review was applied.	The sample consisted of 70 families whose children were diagnosed with ALL.	Non-medical expenses accounted for 46% of the family income of parents in rural areas and 22% of that of parents in urban areas. A family in a rural area spends four times the monthly amount spent at home on food expenses. Furthermore, 38% of families had to borrow money from loan sharks.	3 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Ghatak, et al (2016).(35) India	Analyze the financial burden for families with a child with cancer.	Quantitative and cross-sectional study. Participants completed a cost diary for one month. Socioeconomic status was assessed using the Kuppuswamy scale. For data analysis, statistical analysis was performed.	50 families with children diagnosed with ALL, in the disease induction phase, participated in the first month of therapy.	Medical and non-medical expenses totaled US$524 and US$207, respectively. Monthly expenses were seven times India’s per capita monthly income. 72% of families suffered loss of income during treatment.	4 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Okada, et al (2015).(36) Japan	Investigate the relevant changes that working mothers of children diagnosed with cancer must deal with. Such changes include (1) job change, (2) stress, (3) social support, and (4) work motivation.	Quantitative, exploratory and cross-sectional study. Data were collected through self-report questionnaires.	62 mothers of children diagnosed with cancer up to the age of 15 participated. The most common diagnosis among children was leukemia.	Of the 32 mothers who worked at the time of diagnosis, 10 continued working, 12 took extended leave, 10 stopped working and 70% lost motivation to work. Half of the mothers who continued working during treatment reported financial reasons.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Islam, et al (2015).(37) Bangladesh	Identify the mean costs of childhood cancer treatment for families.	Quantitative and cross-sectional study. The costs were assessed based on the receipts presented by the families.	50 families of children diagnosed with ALL participated, who were treated using the same modified UK Medical Research Council protocol.	The mean cost of all treatments for each family was 4,443 USD, distributed across costs of medications, investigations, procedures, blood and blood products, transportation, food and parental accommodation.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Fluchel, et al (2014).(38) United States	Assess the impact of residence and travel time on care burden for families of childhood cancer patients.	Quantitative and cross-sectional study. A 48-item self-administered questionnaire was used.	354 caregivers of pediatric cancer patients participated, who had been with them for at least three months after diagnosis. The types of diagnosis were ALL, acute myeloid leukemia, brain tumors and solid tumors.	Of the 29% of caregivers who reported changing residence with their child’s diagnosis, 33% said the change was due to cancer. Rural and remote caregivers (>1 hour travel time) missed more workdays, had greater travel expenses, and reported a greater perceived financial burden. Rural patients missed more school days and were at greater risk of having to repeat a grade. One third of caregivers reported leaving or changing jobs after their child’s diagnosis.	8 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Bona, et al (2014).(39) United States	Describe perceived financial difficulties, work interruptions, income losses, and associated economic impact in families of children with advanced cancer, stratified by poverty level.	Quantitative and cross-sectional study. Data for the analysis come from the cross-sectional Survey about Caring for Children with Cancer (SCCC).	104 children and 71 families participated. The children were diagnosed with hematological, solid or brain cancer.	Work interruption was prevalent across all income levels. At least one parent left their job because of illness in 29 (42%) families. Nineteen (27%) families described the disease as a major economic hardship. Six (50%) of the poorest families lost more than 40% of their annual income compared to two (5%) of the richest families. As a result of the loss of income, nine (15%) previously non-poor families fell into poverty.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Tsimicalis, et al (2013a).(40) Canada	Identify and determine independent predictors of the direct and time costs incurred by the family support network and the impact of these costs.	Prospective mixed methods study. A questionnaire was prepared to construct the participant sociodemographic profile. Costs were recorded in diaries. The AHCR was used to record and assess costs.	28 fathers and 71 mothers of children with different cancer diagnoses participated.	The mean total direct and time costs for the 3 months were CAN$154 and $2,776, respectively. The direct and time costs were travel and lost leisure. Direct and time costs were higher among parents who identified a support network at baseline (with financial help, support with household chores, and cancer-related care).	7 of 7 Checklist for Mixed methods MMAT.
Tsimicalis, et al (2013b).(41) Canada	Disaggregate and describe the direct costs borne by families of children with cancer.	Prospective mixed method study. For data collection, diaries, in which parents recorded costs, “The Ambulatory and Home Care Record” (AHCR) instrument and interviews with participants to collect and verify data and discuss costs, were used.	99 parents participated, who completed cost diaries, and 78 participated in the qualitative interview. Children were diagnosed with different types of cancer, with a predominance of ALL.	Total costs incurred by families were US$539,185, with almost three-quarters of these attributed to travel (56%) and food (18%). Parents described bearing out-of-pocket expenses for their children’s treatment, in addition to the side effects of treatment and maintaining the house.	7 of 7 Checklist for Mixed methods MMAT
Hovén, et al (2013).(42) Sweden	Increase knowledge about the socioeconomic conditions of parents of children with cancer.	Quantitative and longitudinal study. Data were collected through telephone interviews, from diagnosis to one year after the end of treatment.	The sample consisted of 139 mothers and 138 fathers of children with cancer. The children had different cancer diagnoses, most with leukemia.	Parents’ work situation was most impacted during treatment. Compared to the time since diagnosis, fewer mothers worked within three months of ending treatment and more mothers were on sick leave one year after ending treatment. Fewer parents worked a year after ending treatment. Family income was reduced after diagnosis and up to three months after the end of treatment.	6 of 8 Checklist for Analytical Cross Sectional Studies JBI.
Tsimicalis, et al (2012).(43) Canada	Identify and determine independent predictors of direct and time costs incurred by families of children with cancer.	Quantitative, prospective and observational study. Descriptive statistics and multiple regression were performed to analyze the data. Participants recorded costs and resources consumed.	99 fathers and mothers of children with cancer participated. Most of the children were female and diagnosed with leukemia.	The mean total direct and time costs over three months were $CAD3503 and $CAD23,130, respectively. The largest component of direct costs was travel and, of time costs, time allocated to unpaid activities.	8 of 8 Checklist for Analytical Cross Sectional Studies JBI.

• ^* The values described refer to the US dollar; **The level of economic burden and financial cost are in sexagesimal degrees

Repercussions of childhood cancer on parental financial conditions

Direct costs

Direct medical costs

Among the studies included, 11 assessed and discussed the impact of direct medical costs.²⁵-²⁷,²⁹-³²,³⁵-³⁷,⁴⁰Medical expenses, associated with hospitalizations, treatment and diagnosis, involved costs related to hospitalizations for treatment and emergency hospitalizations, chemotherapy, diagnostic exams/investigations, surgeries, chemotherapy and radiotherapy cycles and medications in general.²⁶,²⁹,³²,³⁵,³⁷ Parents mentioned paying the expenses for their children to have access to treatment and considered medical costs to be unexpected, burdensome, difficult to deal with and of a cumulative and overwhelming nature, as they consumed a large part of families’ resources. Even so, they continued to pay for them, as it was a matter of survival for their children.²⁶,²⁹ Total medical costs varied between studies as they assessed and tracked different phases and periods of treatment. Furthermore, variations in total cost values can also be explained by changes in children’s conditions and the occurrence of complications or injuries throughout treatment.³⁵,³⁷ The studies described a predominance of hospitalization for episodes of fever and neutropenia, mainly during induction, which required antibiotics or modifications in therapy, due to failure to respond to the first line of antibiotic therapy and/or the need to add treatment antifungal, during treatment.²⁹,³⁷

Direct non-medical costs

The components of families’ non-medical costs for care during childhood cancer treatment were described and assessed by 14 studies included in the review.²⁵-³²,³⁴,³⁵,³⁷,³⁸,⁴¹,⁴³ The main elements responsible for expenses were travel, food and accommodation.²⁵-²⁸,³¹,³⁵,³⁷,⁴¹ Non-medical expenses were responsible for generating a serious financial effect on the family, as they accounted for around two thirds of the total expenses incurred during each hospital stay.³⁴ These costs, which cause substantial, cumulative expenses, are considered a central component of financial distress for parents.²⁸

Travel and accommodation costs are related to the need for families of children with cancer to travel to the hospital so that their children have access to treatment and attend outpatient appointments, which generates expenses with fuel, rent, car purchases, tickets flights or buses, hotel stays, among others.²⁹,³²,³⁴,³⁷,³⁸,⁴¹,⁴³ Furthermore, parents reported the need to change house or even city to stay close to treatment location or family support.²⁹,³¹,³⁸ Regarding food costs, they were associated with recommended nutritional changes and adaptations in the diet of children undergoing treatment, in addition to those for purchasing food during travel and hospital stays, both for the sick children and for other family members.²⁹,³¹Furthermore, costs related to communication were described, since children undergoing treatment often stop attending school and parents incur expenses for purchasing electronic devices and internet access, which become sources of recreation and education for their children.²⁸,³¹ Costs attributed to general child care included expenses for hygiene products, clothes, wigs, wheelchairs and gifts in general.²⁸,⁴³

Indirect costs

Indirect costs: parental employment status

In relation to parents’ work situation, 15 studies analyzed the consequences and adaptations they faced during the care and treatment of their sick child.²²,²⁴-²⁹,³²,³³,³⁵,³⁶,³⁸,³⁹,⁴²,⁴³ Due to intense care demands, parents accumulated absences from work, had to reduce their working hours, changed jobs, took long leaves of absence or, in some cases, abandoned work.²⁵,²⁶,²⁸,²⁹,³²,³⁶,³⁸,³⁹,⁴²,⁴³ Regarding parents who managed their own business, it is observed that they had to ask for support from other family members to manage their establishment or even close the company, which meant loss of income for the family.³⁵Furthermore, among family members, mothers were more likely to have their work situation affected by their children’s diagnosis, giving up their job and other unpaid activities so that they could dedicate time to caring for their sick child.²²,²⁴,³³,⁴³ The impact on families’ employment situation is a consequence of the high demand for care imposed on parents by childhood cancer treatment, as it requires hours dedicated to housework, general child care, adverse effect management, monitoring and waiting in hospital appointments and long trips to children’s treatment center.²⁷,²⁹Family members spent up to 12 hours a day caring for and accompanying their sick children.²⁵Trips to the hospital where the children were undergoing treatment lasted around 110 minutes.³⁸

Indirect costs: family income

As a consequence of the change in work situation, families had to deal with the risk and reduction or loss of income, an impact analyzed by eight studies included in this review.²²,²³,²⁶,³²,³³,³⁵,³⁹,⁴² Family income from work decreased significantly after children’s cancer diagnosis, with a relative reduction in mothers’ income of 21% and fathers’ income of 10%.³³ Parents described constant deprivation of income during the period of their children’s illness.³² The greatest risk for decreased or lost income was observed in the first year of diagnosis and lasted up to three months after the end of treatment.²²,²³,⁴² In a study carried out in India, total treatment costs in the first month of induction therapy accounted for 6.9 times the monthly income of lower-middle class families and 1.7 times the income of upper middle class families.³⁵Although the financial impact affects families at different socioeconomic levels, those poorest were more likely to suffer it and lost up to 40% of their annual income.²⁶

Discussion

The results of this review allow understanding and perception about the different components of direct costs incurred by families from the moment their children are diagnosed with cancer. It was evident that the treatment of this disease requires rapid organization and adaptation of families to the needs and financial demands imposed during children’s therapy trajectory. A previously published study concluded that the financial impact experienced by families in caring for children diagnosed with chronic diseases, such as cerebral palsy, diabetes and cancer, is significantly greater, due to more frequent visits to hospitals and higher costs of medications used in treatment.⁴⁴ Additionally, medical and non-medical expenses incurred by parents during cancer treatment represent a central component of the financial suffering experienced by families and contribute to the financial toxicity present in pediatric oncology.⁹

The current study highlighted that, despite the increase in treatment expenses and the financial difficulties that these additional expenses caused for families, child care and survival were a priority. Furthermore, this study made it possible to detail in which categories family financial costs are concentrated. Medical costs were described in this review similarly to the results presented in a previous study, in which it was identified that medical expenses for childhood cancer begin before diagnosis, last throughout treatment and are related to diagnostic investigations, tests, hospitalizations for routine or emergency, chemotherapy, radiotherapy, medications, among others.⁴⁵ Spending on travel, meals, accommodation, clothing and gifts, for instance, are the most common non-medical costs experienced and reported by families when caring for a sick child, as shown in the scientific literature.¹³,⁴⁶

This review also demonstrated that childhood cancer diagnosis affects the lives of families in different areas, including parents’ professional sphere. Parents experience the challenge of reconciling the hours dedicated to caring for their children with work demands and responsibilities. Similar results were found in a systematic review, which described a high prevalence of interruption in parental employment, in addition to unemployment and prolonged leaves.¹¹ The results of this review also allow to understand significant differences in relation to the impact on the employment situation of fathers and mothers, as presented in a previous study, despite families considering that caring for a child with cancer is a shared responsibility between family members.⁴⁷ However, the role of primary caregiver for children is generally played by mothers, who are therefore more likely to leave their jobs or suffer work interruptions to dedicate themselves exclusively to caring for a child with cancer.⁴⁸ Furthermore, some predictors, such as younger children and low educational level, are more susceptible to the risk of not remaining in employment after this diagnosis.⁴

Finally, the results of this review provide important information for all healthcare professionals involved in caring for families of children and adolescents with cancer. By recognizing the socioeconomic and risk profile of families, it is possible to develop financial guidance plans and implement measures to assess and alleviate the economic burden of cancer treatment. Additionally, it allows these families to be offered information about existing rights, services and social resources. It is necessary to empower them with knowledge about their rights and available resources as a way of helping in times of greater financial instability. Constant efforts to raise awareness about the financial impact experienced by families can ensure that they receive the support they need and have access to all essential health services.

Although this review adds important results to care for children and adolescents with cancer and their families, the presence of limitations is recognized, as it may not completely reflect the diversity of socioeconomic and health contexts, since included studies were conducted in specific geographic regions with different health systems and social policies, which can make it difficult to interpret the results and generalize the conclusions. Furthermore, as financial costs can change over time due to medical advances and changes in health policies, the review does not address the possible evolution of costs over the years, leaving a gap that can be explored by future research.

Conclusion

This integrative review brought together evidence from the scientific literature about the financial costs of families caring for children and adolescents during cancer treatment. The results showed the need for these families to take on a high economic burden throughout their children’s treatment. Direct medical costs were associated with hospitalizations, treatment, and diagnosis. In relation to direct non-medical costs, the main elements responsible for expenses were travel, food and accommodation. The impact on families’ employment situation is a consequence of the high demand for care imposed on caregivers for childhood cancer treatment. As a result of the change in their work situation, they had to deal with the risk and consequences of a decrease or loss of income.

Acknowledgements

This work was carried out with the support of the Coordination for the Improvement of Higher Education Personnel - Brazil (CAPES - Coordenação de Aperfeiçoamento de Pessoal de Nível Superior) - Financing Code 001 and the Brazilian National Council for Scientific and Technological Development (CNPq - Conselho Nacional de Desenvolvimento Científico e Tecnológico), Process 309528/2021-6.

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Edited by

Associate Editor

Meiry Fenrnada Pinto Okuno (https://orcid.org/0000-0003-4200-1186) Escola Paulista de Enfermagem, Universidade Federal de São Paulo, São Paulo, SP, Brasil

Publication Dates

• Publication in this collection
  04 Oct 2024
• Date of issue
  2024

History

• Received
  19 July 2023
• Accepted
  6 May 2024