Of the 336 participants randomized to an experimental consent document and enrolled into a genome study, 13 were deemed ineligible and were removed prior to analysis of data release decisions [19
]. Thirty-eight participants were deemed ineligible for the interview because they were debriefed by phone or mail (n = 27) or they were matched-case controls (n = 11) with other family members present who completed one interview per family. Among the 285 eligible participants invited to the structured interview, the participation rate was 80.4% (n = 229). There were no significant demographic differences between those who completed the interview and those who declined.
Interview participants’ median age was 48.8 years old (range 18–86). Participants were predominantly female (58.5%) and non-Hispanic white (58.1%). The majority reported being married (63.7%), Christian (80.8%) and completing at least some college (55.8%). Most participants also reported an annual household income over USD 40,000 (59.2%) (table ).
Interview participant characteristics
Attitudes Regarding the Risks and Benefits of Data Sharing
While participants agreed that there were benefits and risks with data sharing, participants more strongly identified with the potential benefits than risks. In response to a statement that there were benefits to sharing their genetic information, 72.7% strongly agreed and 25.1% agreed. Contrastingly, 36% strongly agreed and 38.2% agreed that there were risks in sharing this information, while 16.9% strongly or somewhat disagreed (table ).
Participants’ risk benefit assessment
Collectively, participants displayed more uniformity in their judgments about the benefits of data sharing. Asked to rank the most important benefit among an a priori list of 4 options, 62.7% selected advancing research to help others with a similar condition, 23% selected advancing general medical knowledge and 14.3% selected advancing research to help themselves or their family (table ).
Qualitative data analysis of open-ended items reinforces these findings. Although some participants expressed hopes for realizing direct benefit from this research, most recognized this personal benefit was unlikely. As one participant commented (the bracketed numbers refer to the participant ID):
I'm just really serious about allowing the information from my illness to be used to help others down the line. That to me is the only benefit. Probably isn't going to help me, but it may help future patients. (231)
Some participants noted a specific desire to help others with a similar condition.
Well, the next person can avoid what I've had to put up with these past three years with the pancreas not acting nice. I just think it will benefit others with the same kind of illness that I have. (255)
Others described the more general societal benefits of participating in research.
Sharing my genetic information may be just the missing piece that the researchers need to advance good health and avoid diseases, and there may be something in my information that stands out that they didn't get in all the other people they've been studying. (369)
Survey results suggest less consistency in participants’ views when ranking the most important risk in sharing their genetic information. A third (34.6%) selected the risk of having their identity revealed as most important, 30.1% selected not knowing what could happen with their genetic information in the future and 28.2% selected health insurance discrimination. The remaining 7.1% reported the most important risk was the fear of finding out unwanted information about themselves or their family member (table ).
Open-ended items highlight participants’ difficulties in identifying the concrete risks of data sharing. As one participant commented:
As a matter of fact, I'm even having a problem figuring out what the risk would be. Because I said – okay – so somebody learns about I have such-and-such. Well, what good does that do anybody? I'm just having a problem identifying the risks. (281)
Other concerns raised by participants included the lack of control over who could access their information in the public domain, fear of identity theft, anxiety about government access, apprehension over the potential commercialization of their DNA, and fear that their data would be used in morally objectionable research. Participants seemed to understand the inherent identifiability of DNA data, but many felt that it would be years before anyone could actually identify an individual on the basis of their DNA. This may explain why in response to open-ended items older patients seemed less anxious about future risks affecting them and therefore more likely to share their data publicly than younger patients or parents of pediatric patients. As one older participant expressed it:
If I was younger, I wouldn't choose [public], but as old as I am, I'll choose that way. I don't think anybody can go on the internet now or get this information and identify me, and I don't think they will be able to in my lifetime, but I believe in the next 50 or 100 years they'll be able to do it real good, so if I was 30 years old then I'd have to watch that 50-year stretch of time. (363)
Compared to this parent of a pediatric patient:
There's a small percentage of people out there that want to get that information for misuse and misconduct. She's only 8. Her life expectancy is maybe another 70, 80 years. I don't [know] what it will be then. You can't have her running around with that risk for 80 years. (544)
This is also consistent with our previous finding that parents of pediatric patients were significantly more restrictive in their actual data sharing decisions than adult participants [19
Comparing Hypothetical and Actual Preferences
As we previously reported, 83.9% of participants initially consented to public data release when they were enrolled into the genome study, and the majority (53%) chose public data release after debriefing [19
]. During the interview, participants were given an opportunity to review the consent documents they did not receive through randomization, express their opinions about these consents and determine what data release selection they think they would have chosen had they been enrolled with a different consent. Results indicate participants’ hypothetical choices were inconsistent with their actual data sharing decisions, as participants were generally more restrictive in their hypothetical data sharing preferences than in their actual data release selections.
After reviewing the traditional consent, 30.8% reported they would have declined participation if enrolled with this type of consent. However, as previously reported, all participants randomized to the traditional consent initially agreed to participate and have their data released publicly [19
]. Participants reviewing the binary consent also reported that they would have been more restrictive in sharing their genetic data than those who were randomized to this type of consent; hypothetically, 32.9% indicated they would have chosen not to release their data beyond the genome study PI, and 1.3% reported they would have declined participation completely. Of the participants randomized to the binary consent, one participant declined enrollment due to data sharing concerns (the only one in the entire study), but only 15.1% of those who enrolled opted out of data sharing. Likewise, the majority (55.8%) of those reviewing the tiered consent reported that they would have chosen restricted data release, but only 19.5% of those randomized to the tiered consent initially chose this option.
Qualitatively, many participants expressed a dislike of public data sharing, despite having already agreed to full public data release upon enrollment into the genome study. For example, one participant who was randomized to the binary consent and originally chose public data release said:
The public thing; it's a little scary. Because I would think that it would only be people that were researching such a diagnosis or prognosis would need to read such a thing. (263)
Another participant also randomized to binary consent and chose public data release said:
I don't want public access. I want it to be scientific databases in this hospital only. I don't want it everywhere. Now if this hospital calls and says, this database we have … St. Jude's hospital now feels like there's just more research than they can do and we could help them – then yeah. (710)
Interestingly, not all participants who expressed concerns about public data sharing changed to a more restrictive option when provided the opportunity after debriefing.
One possible explanation for the discordance between hypothetical and actual preferences is the notion of a privacy-utility trade-off [13
]. Many of the participants in this study who opted not to change their original consent, despite noted concerns, may have done so because they considered the risks to be minimal, occurring in the future and outweighed by the scientific benefits of making their data broadly accessible. In other words, they may have ultimately decided that the utility of public data release outweighed their real, but less concrete, privacy concerns.
We tested this by asking participants how important it was for them to protect their privacy versus advance research. When asked in separate questionnaire items, the majority (84.2%) of participants strongly agreed that it is important to them to protect their privacy. A smaller, but still significant majority (74%) also strongly agreed that it is important to them to advance research. However, when forced to choose between the two in a single questionnaire item, participants predominantly (67.3%) chose advancing research as more important (fig. ). This was reflected in open-ended responses to questions about participants’ data sharing decisions. For example, one participant explained:
Fig. 1 Participants’ attitudes toward having their privacy protected and advancing research. In separate questions we asked participants how important it is to them to have their privacy protected (n = 228) and to advance research (n = 227). Response (more ...)
Like I said, the public – the word public scares me. But to me at the time, since I only had that choice, I thought it was worth the risk to help the overall good. (566)
Privacy-utility determinations were significantly associated with participants’ actual final data release selections (chi-square test, p < 0.001). After debriefing, 42.2% of participants who felt privacy protection was more important than advancing research chose restricted data release, and 26.6% chose no release beyond the study PI, while 56.8% of those who felt advancing research was more important chose full public data release (table ).
Participants’ privacy-utility determination by final data release selection and trust in medical researchers
Although participants’ trust in doctors doing medical research was not significantly associated with their final data release decisions, trust was significantly associated with their privacy-utility determination (chi-square test, p = 0.014). In general, more study participants expressed some to high trust (n = 165) than no to low trust (n = 31). While participants expressing no to low trust were evenly divided in their privacy-utility determinations, those reporting some to high trust more often selected advancing research as more important than protecting their privacy (table ).