Introduction

In China, the incidence of childhood cancer was 87.1 per million, and the mortality was 36.3 per million in 20 101. Acute lymphoblastic leukaemia (ALL) is the most common malignant disease among children, accounting for about 40% of all newly diagnosed childhood cancers.1 The 5-year survival rate in childhood ALL has greatly increased over time and is now about 70% in China.1 Regardless of better survival, life-saving therapy is costly and may result in a financial burden for these patients' families.2–7 On the one hand, the costs of treatment of ALL and illness-related expenses are immense; on the other hand, parents may have to reduce their work hours or give up paid work to care for their child resulting in loss of income.

Various studies have been conducted in developed countries to determine the costs associated with childhood cancer from a family perspective.8 In these studies, the economic and financial impact of childhood cancer on families was examined on two primary cost categories: direct costs including the actual monetary expenditure related to the illness such as those associated with transport, food, accommodation and so on,3 4 9 10 and indirect costs including the value of productivity loss such as cutting on work time, taking unpaid leave or quitting job.4–7 11–13 Although it is hard to make a precise comparison of the magnitude of the financial costs of families due to variation in study design, all studies reported substantial family financial burden associated with childhood cancer treatment. Specifically, two Canadian studies found that income loss due to work disruption and out-of-pocket expenses were estimated at over 30% of after-tax family income,2 6 and one American study reported that over 50% of the poorest families experienced annual income loss of more than 40%.11

While childhood cancer was shown to have an adverse economic consequence on families in developed countries, it is likely to have even more severe effects in developing countries. In China, average treatment costs for childhood ALL were estimated to be between US$15 128 and US$45 386,14 whereas per capita income was US$4270 in 2018. Although the Chinese government has made great efforts to provide universal health coverage by the year 2010, the coverage is typically shallow. Overall, 65.1% of childhood ALL patients' insurance covered less than 50% of overall medical costs.14

Unfortunately, medical costs are not the only financial burden faced by the Chinese families, and the families with a child with ALL may also incur substantial additional costs associated with the illness. Specifically, there are huge differences in the allocation of medical resources between rural and urban areas and among different provinces. High-quality medical resources are mainly distributed in large central cities such as Beijing, Shanghai, Guangzhou and so on. Therefore, families with a seriously ill child have to go to these cities to receive treatment for a better chance of survival. As a result, the corresponding non-medical out-of-pocket expenses may increase dramatically due to extra expenditures on transport and accommodation. In addition, it is also hard for the parents to keep their jobs while taking care of the sick child, therefore resulting in loss of income.

In addition to unequal distribution of medical resources, there are significant differences between urban and rural areas in terms of income and social security system in China. In 2018, urban per capita income was more than 2.5 times of rural per capita income (US$5938 vs US$2211). Regarding the social security system, rural and urban populations are entitled to enrol in different health insurance schemes with different coverage plans, and unemployment and retirement insurances are only available to the urban working population. As a result, the economic burden is very likely to be different between rural and urban families.

As can be seen, the economic burden on Chinese families with a child with ALL could be devastated. Lacking financial aids from various sources may cause these families to fall from above to below the poverty line or even give up treatment.14 However, research on determining the costs associated with a childhood ALL is rare, and the nature of these costs is poorly understood in developing countries.15–18 It makes policy planning in the context of essential medicines, national fiscal policy towards childhood ALL and donor policy difficult without any reliable estimates of costs. The purpose of this paper was to estimate the economic burden in families with a child with ALL in China. In addition, we also reported the total costs and its three components for rural and urban families separately.

Methods

Results

Medical expenses and parental contact information of a total of 171 patients were extracted from the SCMC database. We contacted the 171 parents using the telephone numbers provided in the database, and 161 parents gave the consent before we conducted the interview. The 10 failed calls were due to either loss of contact or refuse to participate.

Table 1 presents the child, parent and family characteristics for the whole sample and for the urban and rural subsamples. The mean patient age at diagnosis was 4.9 years (SD=3.3 years; range: 0–14 years), and the majority were men (58.4%). The average length of therapy (induction, consolidation and intensification) was 11.6 months (SD=9.6 months). Fifty-two patients (33.1%) did not have any insurance at the time of diagnosis. The mean age of parents at diagnosis was 33.2 years (SD=4.3 years), and the majority of the parents' highest education level was high/vocational school or below (61.9%). In terms of household characteristics, the average family size was 4.1 (SD=1.1), 71 households (44.1%) had rural registration and only 33 households (20.6%) were local residents (Shanghai). The average household monthly income at diagnosis was US$1232.25. Regarding urban and rural families, the highest education for parents in an urban area was significantly higher than that of parents from a rural area (p<0.001). In addition, urban families had smaller family size (p<0.001), higher monthly income (p=0.02) and were more likely to be local residents (p<0.001) than their rural counterparts.

Table 2 describes the parents’ employment statuses at the time of diagnosis and during the treatment period. On diagnosis, 35 (22%) fathers worked in government, state-owned enterprise (SOE) or public sector, 109 (68.6%) worked in private sector or self-employed, 11 (6.9%) were farmers, and 4 (2.5%) were unemployed. During the treatment, 47 working fathers managed to keep their employment status unchanged, 13 completely stopped working, and 97 reported to take extended absences from work. The average length of absences was 14.4 months (SD=11.1 months). Regarding mothers, on diagnosis, 32 (20.1%) worked in government, SOE or public sector, 73 (45.9) worked in private sector or self-employed, 16 (10.1%) were farmers, and 38 (23.9%) were unemployed or doing housework. Among those who had a job, 14.8% did not change their employment status, 6.6% stopped working, and the majority of working mothers (78.7%) took extended absences from work. The average length of absences was 18.1 months (SD=10.8 months).

The three categories of the total costs and their components for the whole sample are given in table 3. Panel A reported that the average total medical costs during the treatment were US$16 307 (SD=14 488; IQR 9441–18 120). Medical costs were then divided into two subcategories: outpatient and inpatient costs. The inpatient costs accounted for the majority of the total medical costs (66.9%). Panel B of table 3 presents the direct non-medical costs incurred during the treatment. The average direct non-medical costs were US$6441 (SD=5038; IQR 3013–8543) with the largest expenditure on accommodation. The average indirect costs incurred during the treatment (Panel C of table 3) were estimated to be USD 8733 (SD=24 321; IQR 0–6727). On average, the total costs for the whole sample were US$31 480 (SD=31 847; IQR 15 518–33 177). The direct medical costs accounted for more than half of the total costs (51.8%), followed by indirect costs (27.7%) and direct non-medical costs (20.5%).

Table 4 reports the total costs and their components for rural and urban families, respectively. The total costs were significantly different between the two groups (mean: US$36 125 vs US$25 592; p=0.021). Regarding the three cost categories, the urban families incurred significantly larger indirect costs than the rural families (mean: US$12 343 vs US$4157; p=0.018). Although the direct non-medical costs were not significantly different, the urban families spent more money on hygiene cleaning products and auxiliary treatment equipment (p=0.041) and gifts and treats (p=0.034) than the rural families.

Discussion

A cancer diagnosis in childhood can substantially affect the physical, psychosocial, and socioeconomic well-being of patients and their families. Yet, research on determining the costs associated with a childhood ALL is rare, and the nature of these costs is poorly understood, especially in developing countries. The present study provides a breakdown of families' costs and resource use and an in-depth understanding of families' financial burden. We found that the financial burden faced by Chinese families with a child with ALL was tremendous. Among the three cost categories, direct medical costs accounted for about 51.8% of the overall costs, and the rest of 48.2% of the total costs were attributed to direct non-medical costs and indirect costs. Regarding families with different household registration type (rural vs urban), the distributions of costs among the three categories were different. Productivity loss contributed a much higher weight in total costs for urban families than for rural families. In addition, rural families spent most of their money on the treatment of ALL.

Our results showed that the average medical costs were approximately US$16 307, which were comparable to the findings from previous studies using data of developing countries.14 15 20 Unlike most developed countries where costs of treatment are borne mainly by the public sector and health insurance,4 13 patients in developing countries have to bear a significant portion of direct medical costs.16 17 According to the health insurance regulations of China, if patient chooses to receive treatment in other province or city or in non-designated hospital, the reimbursement rate could be very low or none at all. Although we were unable to determine this from our study directly, one report did have shown that the actual reimbursement rate was less than 50% for most of the rural families with a child with leukaemia, of which around 27% of children only got 30% of reimbursement.14 In addition, in the questionnaire we asked ‘any comments or suggestions on current insurance reimbursement policy?’, more than half of the non-local parents (57.8%; data not shown) mentioned that the reimbursement rate was too low. Specifically, they identified the low reimbursement mainly due to the following reasons: there existed a big gap on reimbursement rate between local and non-local residents; outpatient and imported medicines were not covered by the insurance, and the reimbursement procedure across provinces was tedious and time consuming, and the actual reimbursement rate was low, so some parents chose to forgo reimbursement.

In contrast to the previous literature which found that transport took a significant portion of family financial cost,9 10 21 our results showed that transport only contributed to 2.7% of the total direct non-medical costs, whereas around 45% of the total direct non-medical expenses were spent on accommodation. That was because most of the non-local families chose to rent near the hospital, which saved travel costs. Although the very poor families can receive 30 days of accommodation at the centre at very low price,15 space is limited, and the 30 day rental period is far from enough. Most non-local families had to rent a room or an apartment near the hospital for about US$146.02–US$730.09 per month for an average of 12 months. Food and nutritional supplements accounted for about 35% of the total direct non-medical cost. As described by Tsimicalis et al10, increased expenses on food were to accommodate the child’s fluctuating weight, satisfy food cravings, and taste alterations.

Following diagnosis, 85% of working mothers and 70% of working fathers gave up all paid employment or took unpaid extended leaves in our sample. These numbers were much higher than those reported in the previous studies.4 6 12 13 The possible explanation was that in our sample, the majority families were from other provinces (79%), and it was hard for only one parent to handle all the issues related to treatment, accommodation, and food; therefore, both parents had to quit their jobs or took unpaid leaves during the treatment.

Our data indicated that families with high socioeconomic status were more likely to receive treatment in high-quality medical facilities. Specifically, according to data from the National Bureau of Statistics of China, in 2010, the annual urban per capita income was US$2822.87, and the amount was RMB874.36 in the rural areas.22 However, our data indicated that the sample urban per capita income (was calculated as urban household monthly income χ 12/family size, using the data from table 1) was 1.9 times that of the national urban average, and the ratio became 2.4 times for the rural per capita income. In addition, our data showed that the total costs were 1.79 times of the sample urban family’s annual income and were 2.72 times of the sample rural family’s annual income. This finding indicated that even for these high socioeconomic families, the economic burden of childhood ALL was huge, especially for rural families.

Our findings have very important policy implications. First, policymakers should make effort on simplifying the reimbursement procedure across provinces and eliminating the huge disparities in reimbursement ratio across regions; second, our sample indicated 33% of patients did not have any insurance at diagnosis, although we did not have direct data on why these parents chose not to purchase insurance for their child, previous study has shown that lack of knowledge or the concept of insurance could be a major barrier for people from participating the insurance program;23 therefore, the government should work hard on educating people regarding the different programmes; third, patients with cancer and their families may need ongoing financial management with a designated financial advisor well beyond the initial treatment phase to help them manage debt, access resources to cope with direct and indirect costs of cancer treatment and maintain patients’ and families’ financial capacity later in life.

There are limitations to this study. First, majority measures were based on parents' self-report, and there may exist recall bias for some measures. However, to minimise recall bias, before conducting a formal interview, we contacted them 1 week in advance and asked parents to recall and list out the details of all the expenses during the treatment. After the interview, we double checked data. If there was inconsistency in the data, we called back to clarify. Second, while the generalisability of this study may be somewhat limited as we focused on one hospital, SCMC, as one of the primary paediatric tertiary care centres in China, it provides treatment of severe disease in children around China (our data showed that 79% of patients were non-local residents). Therefore, our results are likely applicable to other geographic areas. Third, our sample included the families who were relatively rich compare with the national average, which limited our ability to assess the financial impact among the families with low socioeconomic status.

Families of children with ALL experience a wide range of costs. An ongoing investigation of families' costs will yield a rich understanding of the disease costs, formulate the basis of cost assessments, and lend insight into practice and policy changes aimed at lessening the economic impact of this burden.