1 INTRODUCTION

Cancer leads to the first or second cause of death in most countries. About 28.4 million cancer cases are estimated to occur by 2040, with an increase of 47% compared with 19.3 million in 2020, which has imposed a heavy burden on human health.¹ The morbidity and mortality of lung cancer,² gastric cancer,³ esophageal cancer,⁴ liver cancer,⁵ colorectal cancer,⁶ and breast cancer⁷ have remained at a high level in China, which Disability Adjusted of Life Years (DALYs) had reached more than 65% of all cancer DALYs by 2020.⁸ Studies have demonstrated that patients' prognosis is closely related to the time of initial diagnosis.^{9, 10} Unfortunately, symptoms tend to occur only in an advanced stage of the disease, when the prognosis is poor and treatment is expensive. Screening has been proven to be one of the most cost-effective health investments to improve the detection rate and the survival rate of cancer via moving the cancer diagnosis timing forward.^11-13 Multiple-site cancer screening (i.e. packaging cancer screening, PCS) can maximize efficiency, explicitly taking into account a limited budget and scarce resources, constraints acknowledged as significant obstacles for the provision of cancer screening in developing countries.^{14, 15}

Financing counts a great deal for the sustainability of screening. Different countries have applied specific screening financing measures. Japan stipulates that the cost of gastric cancer screening is subsidized by the government, with a 30% co-payment rate personally.¹⁶ South Korea's government and National Health Insurance (NHI) has been free of charge for Medicaid participants and those within the lower 50% of the NHI Corporation premium, although those within the upper 50% need to pay 10% co-payment.¹⁷ England's National Health Service provides free breast, cervical, and colorectal cancer screening for the common mass.¹⁸ China's central transfer payment fund that is only free to a few areas has been the main screening financing resource, whereas most people need to pay their expenses during screening. But Screening costs that are fully biased to one side would prove unaffordable form for whether individuals or the government. Currently, most developing countries have not yet formed reasonable financing strategies. It is unclear for the cost-sharing among financing resources including government, insurance, and individuals. Our study that ascertained the most appropriate proportion of individual payment takes the first step.

Willingness to pay (WTP) has been used extensively to obtain the amount of money that individuals will agree to pay for a product or service in many diverse settings.¹⁹ Specific methods to measure WTP include Contingent Valuation Method (CVM), Choice Experiments (CE), Conjoint Analysis (CA), etc. CVM, the most popular method to measure WTP, usually adopts a questionnaire survey to guide people's maximum WTP for goods or services by a hypothetical scenario, which can overcome the limitation of lacking market trading of public goods or new services to make researchers understand the public preference and then evaluate the value within various assumptions.²⁰ Therefore, CVM was selected as the methodological tool to guide the WTP in this study.

Recently, a few studies have assessed populations' acceptance and WTP toward single-site cancer screening such as colorectal, breast, upper gastrointestinal cancer, et al, which stated the high acceptance and WTP and relatively low WTP values for cancer screening. And factors including age, education level, income and cancer-screening experience were related to residents' willingness to accept and pay for screening.^21-23 However, there are limited studies focusing on individuals' acceptance and WTP toward multiple-cancer packaging screening.

This paper aimed to examine rural residents' acceptance and WTP for packaging screening of 6 high risk cancers (that is lung, breast, colorectal, liver, gastric, and esophageal cancer), which will help to improve the coverage and sustainability of screening, and provide a reference basis for other developing countries to formulate reasonable screening strategies.

2 METHODS

2.2 Questionnaire design

The guidance tools of CVM include open-ended, bidding game, payment card, single-bounded dichotomous choices, and double-bounded dichotomous choices. A combination of double-bounded dichotomous choices and open-ended questions was used to obtain accurate and realistic WTP values in the study.¹⁹ Scenario assumptions, as the core of CVM, were as follows in this study:

At present, the effective cancer-screening methods include low-dose spiral CT examination for lung cancer, endoscopy for gastric and esophageal cancer, serum alpha fetoprotein test combined with abdominal ultrasonography for liver cancer, fecal occult-blood test (FOBT) and colono-scopy for colorectal cancer, mammography, or ultrasonography-screening procedure for breast cancer. Assuming that the local hospital can provide residents with the service of “packaging” six kinds of cancer screening simultaneously with the price of about ¥3000, what is your willingness to accept and pay?

After introducing the scenario assumptions to the respondents, the first step was to ask the respondents whether they were willing to accept it in the case of the free PCS. The second step was to guide the WTP of the respondents who were willing to accept PCS by the method of double-bounded dichotomous choices. According to our previous survey on the price of 6 high-risk cancer screening, ¥3000 was finally determined as the total PCS price under the hypothetical scenario. The respondents were required to randomly select one from the price card (¥50, 150, 250, 350, 450, 600, 800, 950, 1200, 1800, 2200, and 2800, see Figure S1 for details) and then to be asked whether they were willing to pay the price on the card (intermediate value). If the answer was “YES”, the investigators inquired for a higher price, conversely, for a lower price (values on both sides). After two rounds, the double-bounded dichotomous choices were completed. In the third step, an open-ended question was used to obtain the respondents' MAXWTP (Figure 1).

3 RESULTS

3.2 WTP for packaging cancer screening

Table 2 shows 89.36% of participants were willing to accept PCS, comprising 57.66% of positive WTP and 31.70% of 0-WTP. And 10.64% were unwilling to accept it, which the dominant reason being “attending clinics after feeling uncomfortable” (74.51%), followed by “other reasons” and “worrying about the pain or danger of examination” (17.65% versus 15.69%, respectively).

TABLE 2. WTP for packaging cancer screening

Variable	Frequency	Percent (%)
WTP values
Protest-0a	102	10.64
True-0b	304	31.70
WTP >0	553	57.66
Reason for “Prorest-0”
Worry about the pain or danger of examination	16	15.69
Attend clinics after feeling uncomfortable	76	74.51
Physical examination organized by the work unit	2	1.96
Othersc	18	17.65

• Abbreviation: WTP, willingness to pay.
• ^a Unwilling to accept PCS.
• ^b Willing to accept PCS at zero money.
• ^c Others include unbelieving, unwillingness due to being far away and trouble, being sick, and only wanting to do an endoscopy.

Figure 2 lists the distribution of participants' WTP values after excluding protest zeros. The values ranged from ¥50 to ¥3000. The respondents' average WTP for screening was ¥622, accounting for 20.73% of the total cost (¥3000). The WTP values went down as the bid prices increased. More than half of the residents (50.64%) were willing to pay less than ¥350, about one fifth (17.15%) were willing to pay less than ¥1200, and only 5.13% were willing to pay all ¥3000.

4 DISCUSSION

This study, as far as we know, is the first to examine residents' acceptance and WTP for packaging cancer screening. Rural residents have a high acceptance (89.36%), which reflects the possibility of popularizing PCS to a certain extent. Despite the provision of free cancer screening, 10.64% of respondents indicated reluctance to accept it. The main reason was that they did not attend clinics until feeling uncomfortable, but it was too late by then, which stated the necessity to strengthen rural residents' awareness and enthusiasm for precancerous screening. Besides, respondents placed more worry on the pain or danger of examination, as with the results of colonoscopy-related packaging screening.²⁵ It appeared that standardized training, operation skill, and comfortable processes should be emphasized and strengthened.

Region, age, annual income, and cancer-screening experiences were the significant influencing factors of residents' acceptance for PCS. Residents in Dongchangfu and Linqu, with higher age (60–69), with lower income (<10,000) and without cancer-screening experiences had lower acceptance of PCS, which may be related to their weak health awareness, poor cognition and understanding of health information, slender family income, and limited previous cancer-screening experience. It suggested that these kinds of population may be the key intervention population, and efforts should be made to promote cancer screening-related health education to the public on the severity of cancer, the importance of screening, and provide screening costs subsidies to rural residents to a certain extent, so as to improve the population-screening participation rate.^{22, 26}

And 57.66% of respondents were willing to pay for PCS, lower than 76.7% in urban China,¹⁴ 88% in England,²⁷ 72% for breast cancer screening in America,²¹ implying that rural residents' WTP seemed to be relatively low. A study on the attitude of rural residents toward cancer screening in China concluded that most rural residents were only willing to pay 10%~30% of the total screening cost, whether single-site cancer screening or multiple-site packaging cancer screening.²⁸ Our study used CVM to derive the specific WTP values of rural residents, which may be closer to the actual situation. The results show that the average WTP of respondents was ¥622, accounting for 20.73% of the total cost of ¥3000, suggesting that about 20% may be the optimal payment proportion to maximize rural residents' participation in packaging cancer screening. And more than half of respondents were willing to pay only ¥350, which put far from meeting the actual charge of ¥2000–3000 in local hospitals. In other words, rural residents expressed a relatively small amount of WTP in addition to the low WTP. Combined with various studies on cancer screening, price is an important obstacle to WTP.^{29, 30} Whereas cancer-screening costs are usually fully paid by the demander in the case that the current government and health insurance have not yet taken action on mass cancer screening.³¹ Because the USA has incorporated cancer screening into Medicare, the cancer-screening rate of those with Medicare was reported significantly higher than those without.³² Again, a study in the USA pointed out that Medicaid reimbursement was significantly correlated with the screening participation rate.³³ Studies in Japan found that reducing out-of-pocket costs can arouse breast and cervical cancer screening attendance.^{34, 35} Thus, we encourage the government and health insurance to participate in the payment of PCS and manage to bear the remaining 80% except about 20% of the screening fees that individuals were willing to pay themselves. In the future, multi-party financing for cancer screening (e.g. individuals, insurance, and government) may become a promoted policy for rural residents to undergo PCS.

Male, cancer-screening experiences and higher income affected WTP values positively. It may be related that residents with the above characteristics can afford cancer-screening costs based on meeting basic needs, coupled with their high acceptance of screening forms and technology. These findings were roughly consistent with the study in China,^{23, 31} England on colorectal cancer,³⁶ and Ethiopia on cervical cancer.²⁹ Besides, the region was one of the factors affecting WTP, of which Feicheng residents were willing to pay more for PCS than Linqu residents. It may be because Feicheng has just organized endoscopic screening before our survey, which further confirmed the importance of cancer-screening experience to residents' WTP. Moreover, it was interesting that self-employed or freelancers would pay more than the participants from government, enterprises, or factories, with the reverse being the case for Zhou's study.²³ The possible explanation was that these two occupations accounted for a small proportion of our study, so this result should be interpreted with caution. The above findings suggest that it is necessary to strengthen publicity and raise public awareness of cancer screening. More importantly, the economic level greatly limits WTP, which also certifies the importance to broaden financing channels to improve cancer-screening utilization.

The present study has some limitations. First, the respondents were from Shandong and interviewed during a specific period, thus the results need to be carefully applied to the whole rural residents in China. Second, given the hypothetical nature, the WTP values answered by respondents may differ from the corresponding actual behavior. To improve the quality and effectiveness of the findings, the connection between willingness and actual behaviors should be examined in future studies. Third, this study only discussed the influencing factors of WTP on the side of demographic characteristics. Future research should involve participants' knowledge of cancer screening and their health behavior.

5 CONCLUSION

Rural residents were willing to accept and pay for PCS, but the WTP values were low. Region, age, annual income, and cancer-screening experiences were the significant influencing factors of residents' acceptance for PCS. And Feicheng residents, male gender, self-employed or freelancers, residents with higher income and cancer-screening experience had higher willingness to pay for PCS. On the one hand, to improve public awareness and participation in precancerous screening, it is necessary to carry out packaging cancer-screening publicity activities. On the other hand, for improving the coverage and sustainability of cancer screening, residents' out-of-pocket proportion should be controlled within 20% and the rest could be borne by the government, insurance, and other sources. Additionally, should the next research discuss the providers' WTP for PCS in combination with our findings, it will be more conducive to format a reasonable financing mechanism.

AUTHOR CONTRIBUTIONS

Zhang Q: Data curation, Formal analysis, Methodology, Software, Writing-original draft; Ren D: Data curation, Investigation, Software; Chang X: Data curation, Investigation; Sun C: Data curation, Investigation; Liu R: Data curation, Investigation; Wang J: Funding acquisition, Project administration, Resources, Supervision, Writing-review & editing; Zhang N: Conceptualization, Funding acquisition, Investigation, Project administration, Resources, Supervision, Writing-review & editing.

FUNDING INFORMATION

This study was supported by the National Natural Science Foundation of China (71904109) and the Natural Science Foundation of Shandong Province (ZR2019PG006).

CONFLICT OF INTEREST

None.

ETHICS APPROVAL STATEMENT

This study was ethically approved by the Institutional Ethical Review Board of Shandong Cancer Hospital and Institute (SDTHEC201909001).

INFORMED CONSENT STATEMENT

All participants gave written informed consent prior to engaging in this study.