Deliberative Assessment of Surrogate Consent in Dementia Research

Abstract

Background

Research involving incapacitated persons with dementia involves complex scientific, legal, and ethical issues, making traditional surveys of layperson views regarding the ethics of such research challenging. We therefore assessed the impact of democratic deliberation (DD)—involving balanced, detailed education and peer deliberation—on the views of those responsible for persons with dementia.

Methods

178 community-recruited caregivers or primary decision-makers for persons with dementia were randomly assigned to either an all-day DD session group or a control group. Educational materials used for the DD session were vetted for balance and accuracy by an interdisciplinary advisory panel. We assessed the acceptability of family surrogate consent for dementia research (‘surrogate-based research’ or SBR) from a societal policy perspective as well as from the more personal perspectives of deciding for a loved one or for oneself (surrogate and self perspectives), assessed at baseline, immediately post-DD session, and a month after DD date, for 4 research scenarios of varying risk-benefit profiles.

Results

At baseline, a majority in both DD and control groups supported a policy of family consent for dementia research for all research scenarios. The support for a policy of family consent for SBR increased for the DD group, but not for the control group; the change in the DD group was maintained one month later. In the DD group, there were transient changes in attitudes from surrogate or self perspectives; in the control group, there were no changes from baseline in attitude toward surrogate consent from any perspective.

Conclusions

Intensive, balanced, and accurate education along with peer deliberation provided by democratic deliberation leads to a sustained increase in support for a societal policy of family consent for dementia research among those responsible for dementia patients.

1. Introduction

Alzheimer’s disease (AD) is an incurable and devastating illness and current treatments are of only modest benefit.[1] The number of persons with AD worldwide is expected to reach 80.1 million by 2040.[2] Research on AD can involve invasive procedures with unpredictable risks.[3;4] Although some persons with mild disease may be able to provide consent, the disease leads to early decisional incapacity [5;6] and surrogate consent for research is usually necessary. Yet the policies regarding the ethics of surrogate consent for dementia research remain unsettled.

In the US, regulations allow research with incapacitated adults based on consent by their legally authorized representatives (LAR) (45CFR46, 102c, 111.a4, and 116). However, the regulations defer to states for defining the LAR and few states have done so.[7;8] There is no consensus on how much special protection is needed when subjects are enrolled in surrogate-based research (SBR).[9–12] Three recent state laws (California, Virginia, and New Jersey) have diverged on how to balance the potential benefits with risks.[8] The US federal government is currently revisiting SBR oversight policy.[13] The UK is also currently focusing on ethical issues in dementia, including the ethics of SBR.[14]

Given the continuing uncertainty and variability in policies for SBR oversight, input from the public is especially important. There have been few attempts to understand the attitudes of the lay public or of stakeholder groups.[15–18] We recently conducted a national survey of older Americans[19] which showed fairly broad support for SBR. However, SBR policy issues involve considerable scientific, regulatory, and ethical complexities. How valid are these cross-sectional opinions of laypersons when they are based on sparse background knowledge regarding such complex issues? If people have an opportunity to learn directly from experts, deliberate with peers, and reflect more on the issues, their considered opinions may be different than their initial responses. It can be argued that these considered ethical opinions provide a more valid basis for policy-making. Therefore, some have advocated that citizen opinions should be obtained using methods based on the concept of deliberative democracy, [20] especially for certain bioethics questions such as research without subject consent.[21] In this study we employed democratic deliberation (DD) to assess the impact of democratic deliberation on the views of one key group—caregivers and primary decision-makers for persons with dementia—using a randomized experimental design.

2. Methods

2.1 Participants/study sample

This study was part of a larger DD project on SBR involving caregivers/decision-makers for persons with dementia (the study being reported here) and the general public (a study currently being conducted). For both subject groups, the primary aim is to elicit their attitudes toward societal policy for SBR.

The subjects for this study were recruited using mailing lists of the local Alzheimer Association (AA) and an Alzheimer disease research center and by advertisements in the University of Michigan research website and in a local AA chapter newsletter. Persons were eligible if they were either caregivers or primary decision-makers for persons with dementia, living or deceased. We conducted two waves of recruitment and two DD sessions to keep each session small enough for quality control.

2.2 Procedures

Surveys

The subjects were randomized to either a survey-only group (control group) or to the DD session group (DD group). Both groups completed the study survey three times: one month before the DD session date, at the end of the DD day (for DD group) or around that date (by mail, for the control group), and one month after the DD date.

DD procedures

Two weeks prior to the DD date, the members of the DD group were provided with copies of the experts’ presentations to be given at the DD session. Participants were asked to read through the presentations before the meeting, and to prepare any questions they had for the experts. The controls were given only the surveys.

On the day of the DD session, DD group participants were randomly assigned to tables, in groups ranging from 6 to 8 persons per table. There were six groups per session, each with a trained facilitator. The schedule of the day was: plenary introduction; small group ice-breaker exercise; plenary interactive presentation on “Clinical Research in Alzheimer’s Disease,” followed by a plenary interactive presentation on “Ethical Issues in Surrogate-Based Research”; second small group discussion of the two plenary presentations regarding the reasons for and against SBR in general; a plenary interactive session on the four research scenarios (lumbar puncture study, new drug randomized clinical trial, vaccine study, gene transfer study—see below); the third, and final, small group discussion during which the participants were asked to reach a group decision, by consensus or majority, for the following question, “If patients cannot make their own decisions about being in studies like this one, should our society allow or not allow their families to make the decision in their place?” The groups were also asked to provide their rationale as to, “Why should surrogate consent be allowed or not allowed?” To maintain balanced expert responses to all questions, the two experts (AD clinical researcher and bioethicist) were available and traveled together from table to table to answer questions throughout the day.

2.3 Study materials

DD session slide presentations on AD clinical research and on ethics of SBR

There were two 45-minute Powerpoint^® presentations used during the DD day, one on AD clinical research and another on the ethics of SBR (available from corresponding author). These were developed using an iterative process involving experts in respective fields (DK for clinical research in AD; PSA for clinical ethics), the rest of the research team, and members of the project’s advisory panel (consisting of a political science expert in deliberative democracy methods, a senior AD researcher, a bioethicist-sociologist, a geriatrician, a director of human subject protections program at an academic medical center, a qualitative research expert, a gerontological nurse, and a caregiver of a person with AD). Numerous revisions were made on successive drafts for accuracy of the statements about the science of AD research and the ethics of SBR, as well as comprehensibility, clarity, and especially balance.

Attitudes toward surrogate-based research

The survey was a shortened version of an earlier instrument that had been validated and used in previous research.[15] The survey began with an introduction to AD and to the ethical dilemma of SBR, and presented 4 research scenarios of about 120 words each, about which the respondents answered five questions. The four scenarios depict a lumbar puncture (LP) study, a randomized clinical trial (RCT) for a medication, a vaccine trial, and an early phase gene transfer trial.

The first three questions elicit attitudes about the acceptability of SBR from three different perspectives: (1) “If patients cannot make their own decisions about being in studies like this one, should our society allow their families to make the decision in their place?” (societal perspective, Table 2)—this societal perspective question most directly assesses the subject’s attitude toward SBR as a matter of ethics policy; (2) “Suppose you wanted to give a close family member instructions for the future, in case you ever became unable to make decisions for yourself. Would you say you would want to participate in the study?” (self perspective, Table 3); and (3) “Suppose you have a loved one who has Alzheimer’s disease and cannot make decisions for him or herself. Would you give permission for your loved one to be part of this study?” (surrogate perspective, Table 4). The response options were: definitely yes, probably yes, probably no, and definitely no. The self perspective question (question 2) had another question associated with it: “How much freedom or leeway would you give the close family member to go against your preference and instead [opposite of answer to 2] enroll/not enroll you in the study?” with response options of no leeway, some leeway, or complete leeway (also in Table 3). The final question asks the respondents to categorize the risks involved in each scenario as minimal risk (described as “risk of everyday activities”), minor increase over minimal risk, moderate risk, or high risk (Table 5). The survey was written at an 8^th grade reading level (Flesch-Kincaid Grade Level 8.4).

Table 2.

Willingness to allow societal policy of family consent for surrogate-based research (SBR) (societal perspective)

	DD Participants (n=80)						Controls (n=98)
Societal Perspective	Survey 1 (n=80)	Survey 2 (n=80)	Survey 3 (n=76)	Survey 1v2	Survey 2v3	Survey 1v3	Survey 1 (n=91)	Survey 2 (n=89)	Survey 3 (n=93)	Survey 1v2	Survey 2v3	Survey 1v3
Lumbar Puncture	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Definitely Not Allow	4	0	1	<0.0001	0.007	0.002	4	3	7	0.64	0.41	0.33
Probably Not Allow	6	1	7				10	11	8
Probably Allow	61	26	38				58	57	52
Definitely Allow	29	73	54				28	28	34
New Drug RCT	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Definitely Not Allow	1	0	0	0.0001	0.01	0.03	1	3	4	0.06	0.18	0.85
Probably Not Allow	5	3	5				7	2	7
Probably Allow	61	29	47				53	67	58
Definitely Allow	33	68	47				39	27	32
Vaccine	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Definitely Not Allow	6	5	3	0.0006	0.20	0.01	10	7	11	0.25	0.17	0.66
Probably Not Allow	29	9	17				20	19	20
Probably Allow	43	35	41				50	59	50
Definitely Allow	23	51	40				20	15	20
Gene Transfer	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Definitely Not Allow	13	13	8	0.0004	0.23	0.002	19	15	18	0.06	0.05	0.58
Probably Not Allow	34	13	17				20	18	22
Probably Allow	34	26	36				39	54	40
Definitely Allow	19	49	40				23	13	21

Table 3.

Willingness to participate in surrogate based research (SBR) and degree of leeway respondents would allow their future surrogates (self perspective)

	DD Participants (n=80)						Controls (n=98)
Self Perspective/Leeway	Survey 1 (n=80)	Survey 2 (n=80)	Survey 3 (n=76)	Survey 1v2	Survey 2v3	Survey 1v3	Survey 1 (n=91)	Survey 2 (n=89)	Survey 3 (n=93)	Survey 1v2	Survey 2v3	Survey 1v3
Lumbar Puncture	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Self - Definitely No	1	0	1	0.02	0.03		3	1	5	0.78	0.42	0.61
Self - Probably No	12	6	9			0.40	17	13	16
Self - Probably Yes	49	38	50				44	52	48
Self - Definitely Yes	38	56	40				36	35	30
Leeway - None	30	16	16	0.02	0.37		33	17	30	0.01	0.01	0.14
Leeway - Some	56	63	61			0.02	51	66	60
Leeway - Complete	14	21	24				17	17	10
New Drug RCT	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Self - Definitely No	1	0	1	0.02	0.07	0.70	2	1	1	0.19	0.90	0.62
Self - Probably No	9	4	7				11	8	9
Self - Probably Yes	46	38	51				48	54	51
Self - Definitely Yes	44	59	41				39	37	39
Leeway - None	30	19	18	0.10	0.66	0.12	33	23	27	0.10	0.39	0.03
Leeway - Some	55	61	59				52	64	63
Leeway - Complete	15	20	22				14	14	10
Vaccine	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Self - Definitely No	13	11	9	0.80	0.58	0.27	10	11	15	0.57	0.79	0.58
Self - Probably No	29	24	31				40	37	34
Self - Probably Yes	40	41	36				36	37	38
Self - Definitely Yes	19	24	24				14	15	13
Leeway - None	35	24	22	0.07	0.22	0.06	36	36	40	0.12	0.71	0.26
Leeway - Some	54	63	61				52	53	52
Leeway - Complete	11	14	17				12	11	8
Gene Transfer	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Self - Definitely No	20	27	24	0.61	0.31	0.63	26	21	19	0.49	0.78	0.17
Self - Probably No	32	19	32				30	33	34
Self - Probably Yes	32	40	28				28	33	35
Self - Definitely Yes	17	14	17				17	14	12
Leeway - None	39	34	36	0.21	0.80	0.39	52	38	41	0.03	0.57	0.01
Leeway - Some	51	49	49				36	51	51
Leeway - Complete	10	18	16				12	11	9

Table 4.

Willingness to allow participation of a loved one in SBR, when acting as a surrogate (surrogate perspective)

	DD Participants (n=80)						Controls (n=98)
Surrogate Perspective	Survey 1 (n=80)	Survey 2 (n=80)	Survey 3 (n=76)	Survey 1v2	Survey 2v3	Survey 1v3	Survey 1 (n=91)	Survey 2 (n=89)	Survey 3 (n=93)	Survey 1v2	Survey 2v3	Survey 1v3
Lumbar Puncture	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Definitely Not Allow	6	4	5	0.002	0.003	0.30	5	7	9	0.59	0.24	0.12
Probably Not Allow	17	9	20				28	22	20
Probably Allow	57	49	51				48	54	52
Definitely Allow	20	39	24				19	17	20
New Drug RCT	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Definitely Not Allow	6	3	4	0.02	0.04	0.65	2	3	5	0.23	0.51	0.33
Probably Not Allow	6	9	11				13	6	7
Probably Allow	63	49	61				57	70	63
Definitely Allow	24	40	25				28	21	25
Vaccine	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Definitely Not Allow	16	20	20	0.03	0.60	0.31	21	17	22	0.60	0.77	0.85
Probably Not Allow	40	26	32				36	34	33
Probably Allow	40	39	40				34	41	38
Definitely Allow	4	15	8				9	8	8
Gene Transfer	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Definitely Not Allow	24	35	26	0.12	0.17	0.49	34	31	30	0.37	0.45	0.13
Probably Not Allow	43	24	38				29	34	36
Probably Allow	29	33	28				28	31	26
Definitely Allow	4	8	8				10	5	8

Table 5.

Perception of risk level of four research scenarios

	DD Participants (n=80)						Controls (n=98)
Level of Risk	Survey 1 (n=80)	Survey 2 (n=80)	Survey 3 (n=76)	Survey 1v2	Survey 2v3	Survey 1v3	Survey 1 (n=91)	Survey 2 (n=89)	Survey 3 (n=93)	Survey 1v2	Survey 2v3	Survey 1v3
Lumbar Puncture	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Minimal Risk or Less	20	46	30	0.002	0.05	0.04	23	30	25	0.41	0.15	0.33
Minor Increase Over Minimal Risk	42	29	29				40	30	44
Moderate Risk	34	24	40				36	38	28
High Risk	4	1	1				1	2	3
New Drug RCT	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Minimal Risk or Less	28	26	18	0.08	0.63	0.26	30	32	32	0.54	0.04	0.26
Minor Increase Over Minimal Risk	38	41	51				41	35	50
Moderate Risk	32	31	29				29	34	19
High Risk	3	1	1				0	0	0
Vaccine	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Minimal Risk or Less	3	1	0	0.26	0.25	0.84	2	5	5	0.68	0.82	0.86
Minor Increase Over Minimal Risk	9	17	9				18	16	17
Moderate Risk	58	63	62				54	51	52
High Risk	31	19	29				25	29	25
Gene Transfer	%	%	%	p-value	p-value	p-value	%	%	%	p-value	p-value	p-value
Minimal Risk or Less	3	1	0	0.15	0.57	0.52	2	1	3	0.59	0.19	0.57
Minor Increase Over Minimal Risk	8	1	1				9	12	7
Moderate Risk	32	39	40				37	31	39
High Risk	58	58	59				52	56	51

Demographics and subject characteristics

Table 1 describes the subjects’ characteristics.

Table 1.

Comparison of characteristics of subjects in democratic deliberation (DD) group and control group

	DD Group (n=80) n (%)a	Control Group (n=98) n (%)a	Pearson Chi-Square or T-test (p value)
What is your gender?
Female	61 (76)	69 (70)	0.95
What is your current marital status?
Single	11 (14)	6 (6)
Married	51 (64)	59 (60)
Divorced	7 (9)	14 (14)
Widowed	9 (11)	12 (12)
Other	2 (3)	0 (0)	0.19
What is your age? (in years)
Mean	58 (SD13)	59 (SD14)	0.56
Are you Hispanic or Latino/a?
Yes	0 (0)	1 (1)	0.35
What is your race?
White or Caucasian	76 (95)	89 (91)
Black or African-American	3 (4)	2 (2)
Other	0 (0)	0 (0)	0.88
What is the highest level of education you have completed?
High school or less	4 (5)	4 (4)
Some college	29 (36)	29 (30)
Bachelor’s degree	22 (28)	35 (36)
More than Bachelor’s degree	27 (34)	19 (19)	0.68
In general, how do your finances work out at the end of a typical month?
Some money left over	54 (68)	67 (68)
Just enough to make ends meet	18 (23)	16 (16)
Not enough to make ends meet	8 (10)	7 (7)	0.61
Is the person with dementia for whom you are/were a caregiver and/or decision-maker living or deceased?
Living	49 (61)	53 (54)
Deceased	29 (36)	37 (38)	0.69

^aThe percentages of some variables do not total 100% because not all of the respondents chose to answer or because of rounding.

Research attitudes questionnaire (RAQ)

The RAQ is an 11-item scale that assesses participants’ general attitudes towards medical research.[22] The five response options range from Strongly Agree to Strongly Disagree, with total score range of 11 to 55, with higher scores indicating more favorable attitude toward biomedical research.

Deliberative democracy session day evaluation (Table 6)

Table 6.

Perception of the DD^a session by participants (n=79^b)

Question	Score Range 1–10 (SD)
1. Do you feel that your opinions were respected by your group?	9.4 (1.0)
2. Do you feel that the process that led to your group’s responses was fair?	9.7 (0.7)
3. How willing are you to abide by the group’s final position, even if you personally have a different view?	8.4 (2.0)*
4. How helpful did you find each of the following?
Question and answer interaction with experts.	9.5 (0.9)
The formal presentations given by the experts.	9.5 (1.1)
Discussing the issues with other people in similar situation as yours.	9.4 (1.2)

^aDD= Deliberative democracy

^bOf the 79 respondents, 70 (89%) were willing, 9 (11%) were not.

The feedback on the DD Session evaluation form contained six questions on a ten-point (1–10) scale and an open ended question asking, “Is there anything about today’s session that changed how you think about surrogate based research?”

2.4 Analyses

Personal characteristics between the two study groups were compared using the chi-square test for categorical variables, and the two-sample t-test for interval data variable. Within subject categorical responses were analyzed using a test of symmetry.[23] Interval data were analyzed using paired t-tests for within-subject comparison. Analyses were conducted using Stata 9.2 (Stata Corp LP, College Station, TX).

2.5 Human subjects

This study was approved by the University of Michigan’s Institutional Review Board and subjects provided verbal informed consent after the procedures had been fully explained.

3. Results

We randomized 212 volunteers (103 controls; 109 DD). Of the 109 assigned to attend the all-day DD session, 80 (73%) attended. Of the 103 assigned to the control group, 5 dropped out without completing any of the surveys, leaving 98 (97%) who completed at least one survey. All DD group analyses included only those who attended the DD session, and we found no meaningful or statistically significant differences in baseline characteristics between the attendees and non-attendees among those assigned to the DD group. Further, the personal characteristics of the DD attendees and the final control group were similar (Table 1).

3.1 Attitudes toward surrogate based research

Tables 2 through 5 present the responses of the DD group and control group participants, regarding their attitudes toward SBR. At baseline (Survey 1), there were no significant differences between the control group and the DD group for any of the five survey questions regarding attitudes towards the four SBR scenarios, indicating successful randomization.

The baseline support for the societal policy of family consent for SBR (combining probably and definitely allow) (Table 2) was high, ranging from 53% (for the DD group) and 62% support (for the control group) for the gene transfer protocol to 92% to 94% support for the new drug RCT study. For the DD group, there was an increase in support for allowing family consent for SBR when measured just after the DD session (Survey 1 vs. Survey 2) and this change remained significant a month after the DD session (Survey 1 vs. Survey 3), for all four research scenarios. For the controls, there were no sustained changes from baseline to a month after the DD session in their attitudes toward family consent for SBR as a social policy.

In terms of whether one would participate in SBR (Table 3), the attitude of the DD group became more favorable just after the DD session for the lower risk scenarios (LP and new drug RCT scenarios) but not for the higher risk vaccine and gene transfer scenarios. The changes were not sustained a month later. The DD group became more favorable toward allowing leeway to their surrogates for the LP and possibly also for the vaccine scenarios that were sustained at one month. For the controls, there were no changes from the self perspective, and there were small sustained changes in leeway responses for the RCT and gene transfer scenarios.

With regard to whether one would enroll a loved one in SBR (i.e., responding from the surrogate’s perspective, Table 4), the DD group became more favorable toward enrolling a loved one in SBR for the LP, RCT, and vaccine scenarios (but not for gene transfer) just after the DD session, but none of the changes were sustained a month later. For the control group, there were no changes in attitudes across the three surveys on this question.

In terms of perception of risk (Table 5), there were no significant changes in the DD session group’s perception of level of risk for the drug RCT, vaccine, and gene transfer scenarios across the three surveys; for the LP scenario, the post-DD session survey showed decrease in perception of risk, and by one month post session, there appeared to be a polarization in the group, such that there was a decrease in the middle level (minor increase over minimal risk) but an increase in the minimal risk and the moderate risk responses.

The general attitude toward biomedical research (RAQ) scores (scale range 11 to 55) changed from 41.1 (SD 4.0) at baseline to 42.1 (SD 3.9) to 41.8 (SD 4.0) for the DD group. Although, the change was statistically significant (paired t-test for Survey 1 and 3, p=.03), the magnitude was very small. For the control group, the scores were 41.8 (SD 4.1), 41.7 (SD 4.0), and 42.1 (SD 4.4); these were not statistically different.

3.2 Perception of DD session

The participants of the DD session had a very positive view of their experience, as indicated by their responses to the questions in Table 6.

Of note, nearly 90% of the respondents were willing, to varying degrees, to abide by the policy decision put forward by their small group. When asked “Is there anything about today’s session that changed how you think about surrogate based research?” 72 of 80 attendees provided comments. Of these 58% (42/72) were general comments about the session (e.g., “Everything was great!” “Make it longer,” etc.), and not specific answers to the question. The remaining responses commented on increase in knowledge (“By receiving an explanation of the rationale behind research, I became better informed to make a decision. It was helpful to have ‘the experts’ answering questions and providing background information.”), the benefits of deliberation (“Listening/discussing several viewpoints helped clarify my opinions rather than specifically changing them…”), the broadening of frameworks (“Thinking about the issues from a societal instead of personal perspective was a different way to think of research benefits/detriments.”), and a greater appreciation for the complexity of the issues (“I came in with a mindset that it could be easily handled by a POA [power of attorney]… and realized the issues are much more complex.”).

4. Discussion

4.1 Principal findings

Surrogate-based research entails enrolling persons in research who are incapable of providing their own informed consent. Given that this is a particularly vulnerable group, policies should be based on solid data. Because of the scientific, legal, and ethical complexities of the topic, we provided our respondents balanced, detailed information and a chance to deliberate the issues with lay peers. The DD group became more supportive of SBR from a societal perspective, and this change was sustained even after one month. Because the baseline support was already high, this change resulted in very high support (ranging from 76% for gene transfer scenario to 94% for drug RCT) for a societal policy for allowing SBR. Although for the lower risk studies (LP and RCT scenarios) there was a transient increase in acceptance of SBR from surrogate and self perspectives as well, they were not sustained. Further, the change in attitude toward SBR policy was not generally accompanied by a change in risk level perceived by the subjects, nor was there an increase in favorable attitude toward biomedical research in general. Thus, the sustained change was quite specific for allowing SBR from a societal perspective.

4.2 Strengths and weaknesses of the study

This study had specific strengths. Our experimental design was successful, as the control group and the DD group were not different in personal characteristics and did not differ on their initial attitudes toward SBR. Second, the quality of deliberative sessions was high, as reflected in the level of engagement and the comments reflected in the self report feedback. We believe there is good reason to believe that post-DD session opinions of our DD group are more informed, thoughtful, and considered than opinions solicited via traditional surveys. It is also notable that over 90% of the DD participants were willing to abide by policy positions resulting from small group deliberations.

Among the limitations of our study is that although the internal validity is high, external validity is limited by a self-selected sample. However, we note that AD research tends to rely on similarly self-selected samples [24] and that the caregiver/decision-maker samples recruited for this study are highly relevant to the overall debate, since these are the persons likely to function as research surrogates. Second, we did not examine the mechanism of effect, i.e., whether education or deliberation caused the changes. Our sense from the self-report feedback is that peer group discussions and interactive learning from expert presentations (as well as their availability throughout the day) played a greater role than simply having the written materials available. Finally, our conclusions cannot be generalized to other settings, such as research in intensive care units.

4.3 Comparison with prior studies

Although DD has been used for other research ethics questions, [25;26] there have not been any studies using DD methods to assess laypersons’ perspectives on SBR ethics. However, the results of our baseline survey showing that there is broad support for SBR are consistent with several prior surveys showing generally favorable views of SBR in persons at high risk for AD enrolled in a prevention trial,[15] a national survey of American older adults [19], relatives of dementia patients,[17] and elderly clinic patients and senior center patrons.[18] Our results suggest that this generally favorable view of SBR (at least from a societal policy perspective) will increase even further as respondents receive balanced and detailed education and are given an opportunity to deliberate with peers.

4.4 Meaning and implications of the study

Our subjects’ support for SBR increased from a policy (i.e., societal) perspective but not from a personal or a surrogate perspective. It is important to keep in mind the significance of each perspective in interpreting the findings.[19] The responses from the societal policy perspective and the more personal perspectives (surrogate and self perspectives) are closely related, but they should not be treated as equivalent. Personal willingness to participate or willingness to enroll a loved one in research is not a proxy for views about the ethical appropriateness of SBR because many people may choose not to participate or enroll a loved one in research for a variety of reasons that have nothing to do with ethics. If the point is to characterize public opinion on the ethical permissibility of SBR, then respondents’ views from a societal perspective is primary.

We speculate that a sustained change in societal policy attitudes without a sustained change from the self or surrogate perspectives may be due to two factors. First, deciding from a surrogate or a self perspective may feel more personal and concrete, and may engage the emotions to a greater degree than considering a societal policy, and therefore may be less malleable. The societal perspective requires a more intellectual understanding of somewhat abstract societal goods to be balanced against individual interests, and may be more susceptible to change by information and deliberation. As one of our participants put it, “…intellectually I understand the need to have the research done and believe that this is the only way to find out more about the illness and to bring about some real progress. Okay. I support that; I really do. Emotionally speaking, I don’t think I could bear to see my mother suffer.” Second, the DD session gave our participants an opportunity to take up the perspective of societal policy-making which is something that is relatively novel for most people. This may have made their initial and final responses from that perspective more open to change. We believe that this change shows a positive effect, as some DD theorists have argued that one desirable function of democratic deliberation is to “encourage public-spirited perspectives on public issues.”[20]

But does the lack of change in personal and surrogate perspectives signal something that is ethically worrisome? We think not. First, the baseline willingness to participate in research from the self perspective is quite high, comparable to their societal perspective responses. Thus, fundamentally, our respondents are not trying to get a “free ride” by shifting burdens onto others. The fact that their societal perspective answers changed, while their self perspective responses did not, points out that they gained a new appreciation for the societal perspective rather than a retreat into a more selfish perspective. Second, our respondents were clearly less willing to enroll their loved ones in research, as compared to their willingness to enter research themselves or to their support for a societal policy of family consent. This should be seen as a reassuring result. We should anticipate and accept the cautious and conservative approach that family members will take in enrolling their decisionally incapable loved ones in research.

There are two main implications of our study. One implication is that it is feasible to create forums for democratic deliberation to obtain high quality input from the lay public on policy issues that involve moral disagreement or uncertainty, as is typical in bioethics. This may be particularly useful when the ethical issue is not widely discussed and is scientifically and conceptually complex.

The primary implication of our study is that the broad support for allowing SBR from a societal perspective will significantly increase with further education and deliberation. A dramatic example involves the gene transfer scenario, which began with 53% support. Even a month after the DD session, the support was at 76%, a substantial difference. It suggests that societal support for SBR, if elicited after educated deliberation, would be higher than for cross-sectional surveys and may imply that traditional surveys underestimate the level of support that may be attained after deeper ethical reflection by citizens.

Abbreviations used

DD

Deliberative Democracy

AD

Alzheimers’ Disease

LAR

legally authorized representative

SBR

surrogate based research

AA

Alzheimer’s Association

LP

lumbar puncture

RCT

randomized clinical trial

RAQ

Research Attitudes Questionnaire

POA

power of attorney