Public opinions about participating in health research.

Teschke, Kay ; Marino, Suhail ; Chu, Rong 等

Over the last 20 years in Canada and elsewhere, the climate for research examining disease etiology has changed. Administrative databases have made it easier to identify individuals with diseases beyond those with research registries (typically cancer), opening the possibility of population-based study designs. However, this opportunity has raised concerns about releasing records to researchers for whom the data were not originally collected. In the late 1990s, some jurisdictions enacted legislation and ethics review boards implemented policies that restricted release of personal identifying information to enable subject contact. However, much etiological research requires subject contact to elicit details about lifestyle, occupations, residences, and other information not routinely recorded in administrative databases.

Academic and legal interest in the impact of research on subject privacy and, more recently, of privacy legislation on research, has resulted in ethics policies and policy commentaries by researchers, lawyers, and government agents. (1-11) A surprising aspect of the work to date is that little attention has been given to the opinions of those whose privacy is being protected, i.e., what do members of the public think? Some opinion research has examined the issue of consent prior to analyses of administrative data stripped of identifiers. (12-18) Surveys examining the public's desire to actively participate in research and the method of selection and contact are rare. (19-21)

To better understand public opinions, we conducted a survey of the willingness of British Columbia adults to participate in health research and how willingness was affected by the methods of selection, contact, and other factors.

METHODS

Households in the Vancouver region as far as Hope and Squamish, and on Vancouver Island except Victoria (identified using postal codes and representing about half the provincial population, including a wide range of community and socio-economic circumstances) were selected via simple random sampling from the residential listings of the Canpages directory. This method was chosen because it is population-based (though it misses approximately 10% whose numbers are unlisted, who have no telephone, or who have only mobile service; personal communication, Telus, August 2006) and because listed telephone numbers are in the public domain and therefore constitute the only sampling frame from which researchers currently can directly contact a sample. Because residences, not individuals, were the sampling frame (one reason that administrative databases are preferred for population sampling), the covering letter asked that the adult with the next upcoming birthday respond. Respondents were restricted to those well enough to participate and able to read English.

The study was conducted from fall 2006 to spring 2007. The self-administered questionnaire was mailed, followed as necessary by two mailed and two telephone reminders. It was pretested on a convenience sample of 25 adults from a range of educational backgrounds. It included four questions, listed in full in the tables and figures that follow.

Data analyses included summary statistics and confidence limits (95%) for the proportions (p) willing to participate, calculated as p [+ or -] 1.96 * [(p * (1 - p) / n).sup.1/2], where n is the sample size. Logistic regression was done to examine the joint impact on willingness to participate (dichotomized as yes or sometimes vs. never or almost never) of the age and sex of the respondents, and mailing. Multiple linear regressions examined the influence of the same independent variables on the answers to each of the 23 factors queried in the other questions.

The study methods were reviewed and approved by the University of British Columbia Behavioural Research Ethics Board.

RESULTS

The 1,477 participants were 54% female and the average age was 54.0 years (SD=15.7, range: 19 to 97). The participation rate of 58.3% (Table 1) is a conservative estimate, since the number of eligible participants listed includes those who were not contacted. Their eligibility and refusal status is unknown.

Willingness to participate in health research

Most respondents (85.3%) were willing to participate in health research at least some of the time (Table 2). Figure 1 illustrates the proportions willing to participate, stratified by the mailing to which they responded. Willingness declined with the number of mailings; however, even among those who needed several reminders, over 70% were willing.

Women were somewhat more reluctant to participate than men (never or almost never willing: 15.4% vs. 12.3%, respectively). Those who responded "sometimes willing" were younger than those who gave more definite responses in either direction (mean age 51.0 years vs. always or most of the time willing, 54.9 years, and never or almost never willing, 58.9). In logistic regression, sex was not significant (p>0.2), but both mailout and age were (p<0.0001) (odds ratios for willingness to participate: first mailout = 3.6, second mailout = 2.0, compared to third mailout; ages 19-29 = 0.77, 30-39 = 7.4, 40-49 = 4.3, 50-59 = 4.4, 60-69 = 4.0, 70-79 = 4.9, 80-89 = 2.3, compared to 90-97).

Factors influencing willingness to participate

Figure 2 lists the five methods of selection queried. Respondents felt most comfortable being selected from a government list of the population, but the level of comfort for each of these methods was not high, and there was relatively little differentiation between methods. A method designed by the Ministry of Health to help maintain confidentiality of a patient's medical status (i.e., including in the sample both those selected as patients and those selected from the population as a whole so that no one knows which until they are contacted and agree to participate) did not help respondents feel more comfortable.

Figure 3 shows the question about organizations contacting subjects. Respondents felt most comfortable participating in research when contacted by universities or hospitals (68% and 65% comfortable, 10% and 12% uncomfortable, respectively). A higher proportion (26%) felt uncomfortable participating in research when contacted by governments alone, though this was tempered somewhat if universities or hospitals were their research partners. Most respondents (55%) felt uncomfortable participating in research when contacted by private research firms.

Figure 4 lists a series of other factors that might influence whether respondents felt better or worse about participating in health research. Respondents were most positively influenced by the potential to provide future health benefits to society or to themselves (87%). The next strongest positive influence was receiving a copy of study results (81%). Knowing how much time the study would take was a more positive influence than compensation for that time (77% vs. 55%, respectively). Information about the study was a positive influence for most respondents, though information about study funding and design were more important than information about method of selection (71% and 72% respectively, vs. 63%). Most respondents felt positively about their doctor deciding whether they should be contacted (60%), but this was not the case for the pharmacist making that decision (35%). Having intermediaries make the decision about contact was one of the most negative factors, with 15% and 29% feeling negative about their doctor or their pharmacist deciding, respectively. Ethics review by a university was a positive influence for most (65%), but privacy impact assessment by the government was not (46% positive influence, 13% negative). Ethics review and privacy impact assessment may not have been understood by respondents, since these questions had the largest proportions of missing responses (7.9% and 12.5%, respectively, vs. 4.2 to 6.9% for all other questions). Some respondents asked the meaning of a privacy impact assessment.

In multiple linear regressions to examine the influence of mailing, age and sex on the answers to each factor, the only variable that had a consistent association with mean scores was mailing, with those answering on the second or third mailing giving lower scores to all methods of selection and all contact organizations. None of the variables was associated with responses to the fourth question.

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DISCUSSION

Most respondents (85%) were willing to participate in health research in at least some circumstances. The decreasing proportions willing to participate among those answering after the second and third mailings suggest that the non-respondents would have a lower willingness to participate. However, even among those who replied to the later mailings, the majority were willing to participate in health research, suggesting that non-response was not solely indicative of lack of interest.

Respondents felt most comfortable being selected from a government list of the population, though there were few differences by mode of selection. One contradictory result suggests that this may be an area of inquiry that needs discussion with participants: the selection method meant to maintain confidentiality of medical status received less support than direct selection from medical records.

The organization contacting participants was important to comfort about participating in health research. Most respondents felt uncomfortable being contacted by a private research firm. In Canada, such research has the least stringent oversight: it does not require peer review, nor review by a human subjects ethics board. Respondents felt most comfortable being contacted by university or hospital researchers and less comfortable about government contact. This suggests that requiring government data holders to contact study subjects prior to contact by university or hospital researchers may reduce response rates and therefore study quality. (13,22,23) Because our study was conducted by a university, potential participants favouring university-based contact may have been more willing to complete our survey. This might explain some of the comfort gap between university vs. hospital contact, but seems unlikely to explain the much greater difference in comfort with contact by government or private research firms.

[FIGURE 3 OMITTED]

Among other factors postulated to motivate or detract from participation in health research, the strongest motivators were future health benefits to the individual or to society. Many respondents wrote comments about their illnesses or those of family members and the importance of health research to society. Information about the study was also a strong motivator, and the most important information was study results. This has influenced us to send brochures summarizing study results to every participant of our research. Having a doctor as a gatekeeper for participant contact was positive for more than half the participants, but negative for 15%. Comments on the surveys suggested this may have been due to negative feelings about the medical system and the influence of pharmaceutical companies. Pharmacists as gatekeepers elicited more negative responses than all other factors.

Our study measured self-reported preferences, not behaviour. Potential gaps between stated preferences in a hypothetical context and behaviour (24) limit our ability to estimate the true proportion who would participate in research, however, opinion surveys remain important in a policy-making context. Without population-based survey data, ethics boards and government policy-makers are left to be influenced by negative feelings of small numbers of complainants.

Our study and the few others that have addressed the use of administrative databases for identifying subjects for contact-based research are beginning to elucidate public attitudes. In a US study of 735 patients using antihypertensive medication, almost none objected to the use of medication records to identify them. (19) Two studies asked directly about the potential chain of events from a medical record to study contact: an Australian study with 301 respondents; (20) and a British study with 2,872 respondents. (21) The majorities were willing to have records released to researchers for subject contact. Surveys to date suggest that people recognize the value of research to public health, and point to features that positively influence participation. Remaining complexities related to sample selection and the path to subject contact likely require research methods other than a self-administered survey. Willison suggested a deliberative forum such as a citizen's jury as a means to present and consider competing interests related to personal privacy and health research. (25)

Balancing privacy and research in the public interest is not easy, even for experts. Recent studies in Europe and Canada have shown that those responsible for ethics reviews and for policies related to individual consent have defined many different standards of practice. (26,27) Given the difficulty of these decisions, we hope that others will continue to investigate public opinions. They are vital to researchers who need to approach study respondents appropriately, to policy-makers who must set rules that protect privacy while allowing legitimate access to data for the public good, and to the public whose privacy and health must be the joint foundations for the path followed.

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Acknowledgements: We appreciate the kind participation of all the study respondents. Hasina Jamal and Saleema Dhalla were the research assistants who conducted the mailings and telephone follow-up. Malcolm Maclure, British Columbia Ministry of Health, had the initial idea to question the subjects of our Parkinson's disease study about their willingness to participate in health research and the method of identification. This study was funded by the British Columbia Medical Services Foundation.

Received: June 18, 2009

Accepted: November 19, 2009

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Kay Teschke, PhD, [1] Suhail Marino, BA, [1] Rong Chu, MSc, [2] Joseph K.C. Tsui, MD, [3] M. Anne Harris, MSc, [1] Stephen A. Marion, MD, MS, MHSc [1]

Author Affiliations

[1.] School of Population and Public Health, University of British Columbia, Vancouver, BC

[2.] Department of Clinical Epidemiology and Biostatistics, McMaster University, Hamilton, ON

[3.] Department of Neurology, University of British Columbia, Vancouver, BC

Correspondence: Kay Teschke, School of Population and Public Health, University of British Columbia, Vancouver, BC V6T 1Z3, Tel: 604-822-2041, Fax: 604-822-4994, E-mail: kay.teschke@ubc.ca

Conflict of Interest: None to declare.

Table 1. Participation in the Survey

                                                         n       (%)

Households mailed surveys                               3000
  Ineligible, name not at this address (% of mailed)     309   (10.3%)
  Ineligible, unable to complete survey in English
    (% of mailed)                                        110   (3.7%)
  Ineligible, deceased or too ill (% of mailed)           47   (1.6%)
Eligible (% of mailed) *                                2534   (84.5%)
  Refused ([dagger]) (% of eligible)                     183   (7.2%)
  No contact, eligibility & refusal status unknown
    (% of eligible)                                      874   (34.5%)
  Surveys completed (% of eligible)                     1477   (58.3%)

* This is a maximum estimate of the number eligible. It does not
take into account those who may be ineligible but who were not
contacted and whose eligibility status could not be identified.

([dagger]) This is a minimum estimate of the number of refusals.
It does not take into account those who were not contacted and
whose non-response may have indicated refusal to participate.

Table 2. Answers to the Question, "Are You Willing to
Participate in Health Research?"

                                  Percent Giving Each Response
                                             N=1435 *

                                         %

Yes, always or most of the time        41.7%    (39.2-44.3)
Sometimes                              43.6%    (41.0-46.2)
No, never or almost never              14.8%    (13.0-16.6)

* N is smaller than reported in Table 1, because some respondents
did not answer this question.

Figure 1. Answers to the question, "Are you willing to participate
in health research?", stratified by mailing to which participant
responded

1st mailing, n = 839

No, never or almost never         10%
Yes, always or most of the time   47%
Sometimes                         43%

2nd mailing, n = 399

No, never or almost never         18%
Yes, always or most of the time   35%
Sometimes                         47%

3rd mailing, n = 197

No, never or almost never         28%
Yes, always or most of the time   35%
Sometimes                         38%

Note: Areas of circles proportional to numbers answering that mailing.

Note: Table made from pie chart.