We interviewed 9 CF participants and 15 biobank participants (). All 24 interviewees self-reported as White and non-Hispanic, reflecting the racial and ethnic composition of the TGFβ1 study population. The respondents were generally well educated, particularly the biobank participants. Because the biobank’s original recruitment base included regional universities and research-related entities, several biobank participants had significant experience working in scientific research. Three of the biobank participants, but none of the CF participants, recalled (before being shown the consent form during the interview) that they received information about genetic inclusion criteria during their recruitment for the TGFβ1 study.
Demographics of interview participants
Understanding eligibility criteria
All the CF participants reported that they had been recruited frequently for studies because of their disease condition; in contrast, biobank participants’ recruitment to the TGFβ1 study was predicated only on a single previous biospecimen contribution to the EPR. The participants’ understandings about why they had been chosen for a follow-up study depended heavily on these two very different routes to eligibility.
Undergoing CF treatment at a major research center comes with many opportunities to participate in research. For most of the CF participants we interviewed, merely the fact that they had CF was a sufficient explanation for being recruited to the TGFβ1 study. Danny, a CF participant, did not recall why he was recruited for this particular study but said, “Typically, I just figure it’s because of cystic fibrosis.” A few CF participants already knew that their CF genotype was uncommon in some way, and assumed that this genotype was of particular interest to the researchers. One of these, Jodi, said that she had “one of the worst CF mutations,” but was remarkably healthy, which made her “kind of weird.” Another, Alex, noted that he was not surprised to be recruited for a study, because his unusual CF genotype made him “an atypical mutant.” Previous willingness to take part in research was also seen as a likely explanation for recruitment: Patti presumed that her frequent participation in other CF studies was an important factor, “because they know that I’m always going to say yes.” Patti was usually the first to be recruited for a study, she joked, “because I’m a research slut.”
Biobank participants attributed their recruitment to a variety of reasons. Some believed their selection from the biobank was “more or less random,” as Bob put it: “There wasn’t anything that I felt I was being singled out because of.” Others, including Lauren, guessed they were being matched on demographic characteristics: “Perhaps I just happened to fit into the age and the gender and the race and the body type that they needed.” Many, like Kenneth, remembered or suspected that researchers had looked at their blood or DNA and found “something of interest.” For Kenneth it seemed like fun to be a “guinea pig” for the follow-up study, although in the interview he wrestled briefly with how to describe himself. Asked to describe his feelings about being recruited for research based on his biobank contribution, Kenneth responded, “Glad to be of help. Just kind of fun to be, you know—oh, gosh, you know, a guinea pig.” This term, though used jokingly, may have reflected an underlying sense that his DNA was being used in ways he did not fully understand, and that were not likely to benefit him. Amanda recalled, “My impression was, everyone was contacted for the initial blood [biobank enrollment], and then it’s more like a subgroup that stands out from that.” What it might mean to be recruited for the TGFβ1 study as part of this “subgroup,” however, was less clear for Amanda: “Was there something bad, and that’s why you’re being contacted, or is it a good thing that you fell in that one group? So I thought, why not continue with it and find out?”
“We could cure CF”: CF participants’ perceptions of their research role
CF participants generally spoke about their roles in studies as usually involving giving biological samples, a frequent activity for them both as patients and as research participants. “Whenever they ask you for something they usually want blood work,” Henry noted. Amber did not mind being approached with an invitation to participate in a research study while she was at a clinical appointment. She said, “It’s a very convenient time to discuss medical-type things.” When Danny was asked whether he expected to be recruited for a follow-up study, he replied simply, “It’s never out of the question.” Henry mused, “you never know” when researchers will request participation in a follow-up study. “If they do, that’s fine. If they don’t, that’s fine.” It appeared that overlapping clinical and research contexts contributed to CF participants’ seemingly nonchalant reactions to being recruited for the TGFβ1 study.
Several CF participants explained that they were usually willing to participate in studies because they felt that they were contributing to a good cause—and they enjoyed receiving the small monetary compensation. Patti summed up her reaction to the follow-up contact as a “multiwin” situation:
“First of all, selfishly, I’m at clinic anyway. I get a little money. It’s nice just to have a little extra. It’s like finding some money on the road. … It increases knowledge of CF for the researchers to be used to find a cure. It was convenient. … I mean, how could you say no? It’s a little “stick,” [and] some questions.”
For Patti, as for most CF participants, the TGFβ1 study did not particularly stand out; it was one contact among many with the CF clinic—during which there was a possibility of receiving, along with their clinical treatment, “a little extra.” Because of the relationships that CF participants frequently form over years of interaction with physician–researchers, they often feel that they are part of a “team” that is working to help the CF community. “We need to let these old men in Chapel Hill retire!” Patti said, laughing. “Wouldn’t it be cool if we could cure CF? … And then they could just retire and enjoy life, knowing they did what they set out to do.”
Control, random, normal? Biobank participants’ perceptions of their research role
Not surprisingly, biobank participants did not share CF participants’ sense that they were part of a clinical/research community, although all of them stated they were glad to be of help to the follow-up study. Their understandings of their role in the TGFβ1 study ranged from being a fairly insignificant part of a large group to being a person with a uniquely helpful and interesting genetic variant. For example, Brent, recognizing researchers’ need for large sample sizes, saw himself as one among many who assisted in meeting that need: “The more samples you have, the higher the power; the higher the power, the more likely that what you find is true.” At the other end of the spectrum were biobank participants who understood that “they had identified something interesting and [wanted] to follow up on it,” as Steve put it. Another biobank participant, Scott, even found it “exciting” to hear that “I had something that somebody was interested in researching.” Remembering receiving his recruitment letter, Scott recalled it was “almost like I won the lottery.”
Whether they felt unique or incidental to the TGFβ1 study, however, biobank participants’ most common self-description was as a “control”—a concrete-sounding term that nevertheless meant very different things to different participants. To several, being a “control” meant that they were “normal” or “healthy.” Brent, who said that he had been recruited as part of the “normal individual cohort,” explained that, to him, this phrase meant: “You were randomly selected to be a participant of the normal because you didn’t fulfill any clinical criteria to make you part of a clinical treatment group.” Others, like Anne, presumed either that their selection from the biobank had been made at random, or that they were demographically or phenotypically matched with a CF participant. Anne surmised, “I probably mirrored people who have CF but [I] didn’t have it, and so I—that’s why they wanted to use a control.” In contrast, Steve expressed confusion as to his status in the study, because being a control meant to him that he did not have the genetic variant of interest: “Was I part of the control group, or did I have that particular trait? And I was curious, and it would have been interesting to know.” Kathryn, who had received CF carrier screening as part of her recent prenatal care, recalled of her recruitment letter, “I think it said I was a control who didn’t have the cystic fibrosis gene, and that was not a surprise to me because I already had been recently screened for that and knew.” For Kathryn, as for several biobank participants, having the genetic variant of interest for a cystic fibrosis study was equivalent to having “the cystic fibrosis gene”—either having the disease or being a carrier for the disease.
It was this association that primarily troubled the biobank participants who expressed concern about their recruitment for a CF study. Confident that she was a control without “the cystic fibrosis gene,” Kathryn was pleased to be recruited for the TGFβ1 study for both “the money and the chance to keep contributing to an ongoing research study.” Those who were not as sure about their status as participants, however, frequently noted that their recruitment caused some anxiety. For example, Brian’s reaction to his recruitment letter was mixed: he was “kind of excited” to be contacted, yet he was unsure of the implications. He called the phone number given in the letter, seeking more information about his selection. Although Brian did decide to participate, he did not feel fully reassured from speaking with study personnel on that call. He was particularly worried, he recalled, because he and his wife were planning to conceive, “and we had decided beforehand not to get amniocentesis or any genetic testing done on the fetus. So not having any genetic problems in her family or mine, I was wondering why they’d contacted me about cystic fibrosis.” Bill expressed similar concerns because of his infant daughter; he recalled being assured over the phone that his recruitment did not mean he was a carrier of cystic fibrosis, but this was only after reading the recruitment letter and “initially getting a little anxious.” Having already made their decision and participated in the TGFβ1 study, however, some biobank participants with anxieties downplayed their concerns during the interview, even as they were articulating them. James, for example, mentioned and then immediately dismissed his worries:
“Probably the title sort of surprised me, but then I don’t know whether I was the control or the subject group. So again that’s probably the only thing that was a little—I won’t say surprising, but unnerving, we’ll say. Not—it wasn’t super unnerving, it was just different. … So it just—again, it’s just an emotional thing. It’s not something.”
These self-understanding of one’s status as a research participant can also change over time; like Bill, several biobank participants who were initially troubled about their recruitment decided later that there was no cause for worry. On the other hand, some who were not initially worried found that anxieties developed later. Heather recalled feeling reassured after talking with a researcher; yet later she began to wonder “if there’s something freaky going on with my genes.” For a few, our interview questions about their participation raised some concerns. Julia, at the end of her interview, asked the interviewer to remind her of the specifics of the biobank and TGFβ1 study, wondering, “Was that a bigger deal than what I thought at the time?” Feeling reassured by the interviewer’s response, she decided, “I felt fine with it then. I feel fine with it now.”