Keywords
financial strain - hematological neoplasm - health care cost - pediatric cancers -
health economics
Introduction
Pediatric hematological cancers (acute leukemia, lymphoma, and other blood-related
malignancies) are one of the main causes of cancer-related mortality in children worldwide.
The number of new cases of childhood cancer exceeds 200,000 annually, and more than
80% of these are from the developing world.[1] In 2017, childhood cancer was the fifth leading cause of death among 5- to 14-year-olds.[2] According to the Economic Survey 2022–23, the combined expenditure of India's central
and state governments on health care was 1.6% of the country's gross domestic product
(GDP) in the financial year 2020–21. However, this expenditure increased to 2.1% of
GDP in 2022–23, indicating a growing commitment to public health care funding. Emerging
therapies and diagnostics are extremely expensive, and these costs are borne by the
family, the health care sector, and society.[3] The economic burden of pediatric hematological malignancies extends beyond direct
medical expenditures, incorporating indirect costs such as loss of productivity and
impaired quality of life for patients and their families.
The various protocols for treating hematological malignancy in children entail intensive
multiagent chemotherapy to achieve and maintain complete remission. The chemotherapy
consists of induction, consolidation, central nervous system prophylaxis, delayed
intensification, and maintenance therapy for up to 2 to 3 years, depending on the
risk group. Nowadays, the cure rate with 5-year disease-free survival ranges from
50 to >90% at various centers in India and abroad, depending on risk stratification
and the expertise of the treating center.[4] However, intensive chemotherapy carries the potential for serious complications,
such as infections and bleeding requiring prolonged intensive care unit admission
and hospital stay, costing a lot to the family, with a lot of financial burden on
the family, society, and nation.
There is a paucity of Indian literature showing the cost implications for patients
with hematological malignancies. This study aimed to perform a cost-of-illness analysis
to determine the financial burden due to total direct and indirect costs incurred
in the treatment of hematological malignancy in children.
Study Design and Setting
This prospective observational study was conducted over a period of 6 months, from
February 2021 to July 2021. A consecutive sampling technique was used to recruit participants.
The study aimed to explore the economic impact of pediatric hematological cancer treatment
within a predefined time frame. Given the observational nature of the study and logistical
constraints, a prior sample size calculation was not performed.
Inclusion Criteria
Patients with hematological malignancy aged 1 month to 18 years presenting to the
hospital setting were enrolled.
Exclusion Criteria
-
Patients with hematological malignancy partially treated outside were excluded.
-
Families of patients who did not give consent were excluded.
Primary Outcome
The primary outcome was the total gross direct and indirect costs incurred in the
treatment of pediatric hematological malignancy.
Secondary Outcome
The secondary outcome included the assessment of the proportion of direct versus indirect
costs, variation in costs by diagnosis and treatment phase, catastrophic health expenditure
(CHE) borne by families, and out-of-pocket expenditure.
Statistical Analysis
Direct medical costs encompassed expenses related to diagnostics, laboratory investigations,
radiological imaging, outpatient and emergency consultations, supportive treatments
and chemotherapy, medications for managing complications, nursing services, operating
room charges, medical equipment, and care provided by nonphysician health care professionals.
Indirect costs included transportation costs, accommodation costs, and the cost of
care for the dependent families and caregivers. The average costs, median, and interquartile
range for direct and indirect categories were calculated to assess the overall financial
burden. Statistical analysis was done using Microsoft Excel.
Ethical Approval
The study was approved by the institutional ethics committee of All India Institute
of Medical Sciences (AIIMS), Jodhpur (vide letter no: AIIMS/IEC/2021/3591, dated April
24, 2021). The objectives of the study were explained to all the parents/caregivers
of the enrolled patients, and written consent was obtained from each. All of them
were also assured of confidentiality.
Results
[Table 1] depicts the sociodemographic distribution of the patients. A total of 60 children
were included in the study, with the maximum children falling in the age group of
0 to 5 years. Male pediatric patients were 34 (56.66%), and female pediatric patients
were 26 (43.33%). The majority of patients (98.3%) were diagnosed with acute lymphoblastic
leukemia (ALL). Hospital consultations were attended by the father, mother, siblings,
and grandparents of the patients. However, the majority of the children were attended
by their father. The geographic distribution of the patients is depicted by a map
of Rajasthan (India) ([Fig. 1]).
Table 1
Details of the participants
|
Variables
|
N
|
%
|
|
Age group (y)
|
0–5
|
26
|
43.3
|
|
6–10
|
14
|
23.3
|
|
11–15
|
17
|
28.3
|
|
16–18
|
3
|
5
|
|
Sex
|
Male
|
34
|
56.6
|
|
Female
|
26
|
43.3
|
|
Malignancy
|
ALL
|
59
|
98.3
|
|
AML
|
1
|
1.6
|
|
Average number of visits to hospital (d)
|
1–5
|
55
|
91.6
|
|
6–10
|
3
|
5
|
|
11–15
|
2
|
3.3
|
Abbreviations: ALL, acute lymphoblastic leukemia; AML, acute myeloid leukemia.
Fig. 1 Geographic distribution of patients attending our hospital located in the Jodhpur
district (each dot represents a patient).
The average duration of treatment in these children was 6 months. The average expenditure
on medical investigations is considerable, while costs for procedures and medications
further exacerbate the financial strain on patients and their families. These factors
collectively represent the direct costs associated with pediatric hematological cancers
([Table 2]). Transportation costs per visit and expenses related to attendants during hospital
admissions further add to the overall costs ([Table 3]).
Table 2
Expenditure for the patients in various domains (direct cost)
|
Cost component
|
Average (INR)
|
Median (INR)
|
IQR
|
|
1
|
Cost incurred for investigations by patients in the last 6 months
|
260,922.8
|
250,000
|
335,500–153,000 = 182,500
|
|
2
|
Cost incurred for investigations in AIIMS by patients in the last 6 months
|
101,290.1
|
100,000
|
105,000–57,500 = 47,500
|
|
3
|
Cost incurred for any procedures during hospital admissions in the last 6 months
|
48,137.9
|
38,000
|
53,725–35,000 = 18,725
|
|
4
|
Medication cost incurred in the last 6 months
|
31,818.1
|
30,000
|
35,000–20,000 = 15,000
|
Abbreviations: AIIMS, All India Institute of Medical Sciences; IQR, interquartile
range.
Table 3
Expenditure for the patients in various domains (indirect cost)
|
Cost component
|
Average (INR)
|
Median (INR)
|
IQR
|
|
1
|
Cost incurred for transportation per visit in the last 6 months
|
2,324.1
|
500
|
525–475 = 50
|
|
2
|
Cost incurred for transport, food, and stay of one attendant in a single admission
|
5,593.3
|
500
|
1,000–500 = 500
|
|
3
|
Cost incurred per attendant during hospital stay
|
11,281.6
|
500
|
15,750–500 = 15,250
|
Abbreviations: INR, Indian rupee; IQR, interquartile range.
On average, attendants lost a substantial number of workdays during a single visit
for patients. Specifically, 26 patients reported losing between 31 and 60 days of
work, and 3 patients reported losing >90 days of work. These data underscore the considerable
impact of medical visits on the working lives of attendants ([Table 4]).
Table 4
Average number of days of work lost in a single visit for one attendant
|
Number of days
|
Number of patients
|
|
1–30
|
24
|
|
31–60
|
26
|
|
61–90
|
7
|
|
>90
|
3
|
Among the 60 patients analyzed, 19 required blood transfusions. The average cost of
illness for these patients amounted to ₹ 74,771.27. Additionally, it was observed
that 21% of households experienced catastrophic financial impacts due to hematological
cancers. CHE is typically defined as health care spending that significantly affects
a household's ability to maintain a basic standard of living. Specific to India, a
household is said to suffer from CHE if out-of-pocket expenses exceed a certain percentage
(10–25%) of annual household income or total consumption expenditure. We can observe
that blood transfusions substantially contribute to the overall cost of illness, and
a notable percentage of households are financially devastated by the expenses associated
with hematological cancers. [Fig. 2] depicts different types of costs for patients who underwent blood transfusions.
Fig. 2 Graph depicting different types of costs for patients who underwent blood transfusion.
Discussion
The findings from this study underscore the substantial economic impact of pediatric
hematological cancers on families in Western Rajasthan. The direct medical costs,
including diagnostic tests, medications, and supportive care, constitute a considerable
portion of the financial burden. These expenses are exacerbated by indirect costs
such as transportation and accommodation for caregivers. The cumulative effect of
these costs leads to considerable financial strain on families, many of whom are already
economically vulnerable.
The geographic distribution of patients suggests that families from various parts
of Rajasthan travel substantial distances to access specialized care, further increasing
the nonmedical costs. Families living in rural or remote areas face considerable barriers
in accessing these facilities, including long travel times, high transportation costs,
and logistical challenges of arranging travel and accommodation. This delay can occur
at initial consultation, referral to a specialist, and the actual diagnostic process
involving specialized tests and procedures.[5]
[6] Overall, the geographical distance from advanced health care services is a critical
factor in timely diagnosis and treatment for pediatric hematological cancers, where
early detection and intervention are crucial for better health outcomes. This geographic
factor emphasizes the importance of regional health care infrastructure improvements
and telemedicine to reduce travel-related expenses.[7]
[8]
The expenditure on medical investigations alone demonstrates the exorbitant expense
of health care services for treating pediatric hematological cancers. Our findings
are coordinated with some Indian studies, which indicate that the highest expenditure
is on medical expenses.[3]
[9] This financial burden is compounded by the indirect costs associated with travel,
stay, loss of productivity and income, as caregivers often need to take extended leave
from work to support their children during treatment. Our findings are consistent
with research that found that a cancer diagnosis in children had a significant influence
on the productivity and duration of work of their parents.[10]
[11]
Several factors contribute to the rising costs associated with pediatric hematological
cancers, primarily centered around the issues of late presentation and diagnosis.
When patients seek medical attention at advanced stages of their illness, they often
face a higher likelihood of complications. These complications can significantly worsen
the patient's condition, leading to disease progression that may require more intensive
and expensive treatments. As the severity of the disease increases, the need for hospital
admissions becomes more frequent. Extended hospital stays, emergency interventions,
and additional diagnostic tests add to the financial burden on families, ultimately
escalating overall expenses.
A substantial portion of the study population required blood transfusions, which added
to the overall cost of illness. The study found that these transfusions significantly
contribute to the financial burden, with the average cost of illness amounting to
₹ 74,771.27. The economic impact is profound, with 21% of households experiencing
catastrophic financial consequences, indicating that the cost of treating pediatric
hematological cancers can lead to severe economic distress and potentially push families
into poverty. Prior studies conducted in eight Southeast Asian nations found that
the prevalence of CHE associated with cancer was 48%.[12] This statistic reflects the severe economic disruption caused by the costs associated
with treating pediatric hematological cancers, which can lead to long-term financial
instability for affected families.
The high cost of illness emphasizes the necessity for robust health insurance schemes
and financial support programs specifically tailored for pediatric cancer patients.
The cost of cancer care can be exceedingly burdensome, with patients often facing
substantial out-of-pocket expenses that can be financially crippling. Recognizing
this challenge, the Government of India has introduced the National Health Protection
Scheme under the Ayushman Bharat Program. This initiative aims to provide much-needed
financial relief and ensure that vulnerable populations have access to essential health
care services without the burden of excessive costs. By covering a substantial portion
of medical expenses, the scheme seeks to alleviate the financial strain on families
affected by cancer and other serious illnesses, thereby promoting more equitable health
care access across the country.[13]
Strengths
-
Prospective Design: The prospective nature of the study ensures real-time and accurate data collection,
reducing recall bias.
-
Focused Population: Exclusive focus on pediatric hematological malignancies provides a specific, high-burden,
and under-researched subgroup.
-
Comprehensive Cost Analysis: Inclusion of both direct and indirect costs offers a holistic view of the economic
burden.
-
Real-World Data: Conducted in a tertiary care government center, it reflects real-world treatment
patterns and expenditure in a resource-constrained setting.
-
Contextual Relevance: Provides crucial insights for policymakers and health care planners in low- and
middle-income settings, particularly rural and semiurban India.
Limitations
-
Short Study Duration: The 6-month duration of the study may limit the ability to capture long-term economic
impacts and variations in treatment costs over a more extended period, especially
considering the prolonged nature of pediatric cancer treatments.
-
Small Sample Size: The study sample of 60 subjects may not be large enough to draw generalized conclusions
about the economic impact of pediatric hematological cancer treatment across diverse
populations. A larger sample would increase the statistical power and external validity
of the findings.
-
Regional Limitation: The study focuses on a tertiary care center in Western Rajasthan, which may not
be representative of other regions in India or other countries. The findings may be
influenced by the regional health care infrastructure, economic conditions, and accessibility
to resources, limiting the generalizability of the results.
-
Single-Center Study: Being based at a single tertiary care center, the study may not account for variations
in treatment practices, costs, and patient experiences across different hospitals
or health care settings, leading to potential bias in the results.
-
Disease-Related limitation: ALL was the predominant malignancy in this cohort, and it included only a few lymphoma
cases.
Future Research Directions
Future Research Directions
-
Longitudinal Multicenter Studies: To capture variations over time and across different health care settings.
-
Cost-Effectiveness Analyses: Evaluating different treatment protocols or support interventions for economic efficiency.
-
Impact of Financial Support Schemes: Analyzing the effectiveness of government or NGO-based funding mechanisms.
Generalizability
Our findings are broadly generalizable to:
-
Government-run tertiary health care facilities in rural or semiurban India.
-
Populations with similar socioeconomic and health care access profiles in developing
countries.
Less applicable to:
Gray Areas
-
Economic Impact on Education of the Child and Siblings: School dropout or absenteeism due to treatment demands.
-
Long-Term Financial Toxicity: Extended impact on household savings, debt, and social mobility.
-
Comparison of Cost Across Cancer Types or Risk Groups: Not explored in the current study.
-
Psychosocial Coping Mechanisms and Costs: Largely missing from the analysis.
-
Delayed Diagnosis and Its Economic Implications: Not addressed but potentially significant in rural setups.
Suggestions
-
To reduce the catastrophic financial impact of costs associated with hematological
cancers and blood transfusions, several strategies can be considered:
-
✓ Insurance Coverage Improvements and Awareness: These patients should be automatically enrolled in the Ayushman Bharat program,
with compulsory enrollment facilitated through panchayats, particularly in rural areas.
This initiative is essential at the tertiary level, as complex cases often arise during
diagnosis.
-
✓ Cost Transparency: Improving transparency around the costs of treatments and procedures can help patients
make informed decisions and plan their finances better.
-
✓ Community Support Services: Enhancing access to community support services, including counseling and financial
planning, can help families navigate the financial complexities of treatment.
-
Shared-Care Options: It will support oncology specialty services and help reduce transportation expenses.
Additionally, increasing support for blood transfusions and ensuring that doctors
are well-educated on the latest treatment protocols will contribute to more effective
and affordable care for patients.
-
InHouse Facilities: If all tests and investigations, such as flow cytometry, minimal residual disease
assessment, molecular cytogenetics, and karyotyping, are made available in-house,
overall costs could be reduced. Additionally, the Ayushman Bharat could cover these
services, further alleviating financial burdens on patients.
-
Good Nutrition and Hygiene: Effective treatment for pediatric hematological cancers relies on good nutrition
and hygiene. However, the health system often focuses on arranging finances and logistics,
which can compromise the quality of primary care. This highlights the need for a more
integrated approach that addresses both financial and foundational health needs.
Conclusion
This study highlights the substantial financial burden faced by families of pediatric
patients undergoing treatment for hematological cancers in western Rajasthan. The
direct and indirect costs associated with care not only strain household finances
but also contribute to significant emotional and psychological stress. Implementing
targeted strategies, such as enhancing insurance coverage through the Ayushman Bharat
program, improving cost transparency, and expanding community support services, is
crucial. Additionally, developing in-house diagnostic facilities and prioritizing
nutrition and hygiene can improve health outcomes while alleviating financial pressures.
A multifaceted approach is essential to ensure families can afford treatment and maintain
their dignity during these challenging times.
