Background

Although the idea of citizen science is not new, in recent decades, it has become a popular approach to research. That approach generally describes the meaningful involvement of members of the public—known as citizen scientists—in the research process. Citizen scientists contribute to research in personal rather than professional capacities and might not have advanced scientific training relevant to the projects they support (Eitzel et al. 2017).

There are examples of citizen science projects in many scientific disciplines, including ecology, astronomy, biology, and medicine (Bonetta 2009). The biomedical citizen science landscape in particular is diverse and encompasses projects with various objectives, organized according to a wide range of designs, and taking place in traditional and nontraditional settings that include participants’ homes and community laboratories (Fiske et al. 2019; Wiggins and Wilbanks 2019). Borrowing from other citizen science typologies, these projects can be categorized by the identities and contributions of participants as: (1) top-down activities that are designed and controlled by institution-based scientists with minimal to no input from citizen scientists; (2) collaborative activities that are designed and controlled by institution-based scientists with robust input from citizen scientists, who also help to execute the research; and (3) bottom-up activities that are designed, controlled, and executed by citizen scientists with minimal to no input from institution-based scientists (Aungst, Fishman, and McGowan 2017; Fiske et al. 2019). Some examples of bottom-up biomedical citizen science projects include an online platform for individuals to publicly share their genotyping results and phenotypic information for personal exploration and research use; a community laboratory project to sequence the genome of a cuttlefish; a three-person effort to reverse engineer a gene-therapy drug; a collaboration involving multiple community laboratories to develop a process for generic manufacture of insulin; and a citizen science collaborative involving individuals self-manufacturing and self-injecting putative vaccines (bioCURIOUS n.d.; Greshake et al. 2014; Gallegos et al. 2018; Guerrini et al. 2020a; Talbot 2020).

Reflecting the diversity of the biomedical citizen science landscape, participants have described their activities or communities according to many different terms, including biohacking, community science, and do-it-yourself (DIY) biology (Trejo et al. 2021) (Figure 1). Grinders—a term that describes body modification enthusiasts who implant themselves with cybernetics in hopes of enhancing their senses and other capabilities (Mallonee 2017; Pearlman 2017)—and lifehackers—a term that describes individuals who track their health data and might adjust their behaviors to understand the effects (Wexler 2017)—are also viewed by some as participants in this landscape.

In recent years, United States (US) policy makers have made important investments in citizen science. The National Institutes of Health (NIH) formed a working group in part to facilitate the incorporation of citizen science methodologies into biomedical research (Theisz, Couch, and Gillanders 2016), and the National Cancer Institute launched a Biomedical Citizen Science Hub that provides online tools for communicating about and collaborating on projects (NCI n.d.).

At the same time, policy makers are aware that involving members of the public in the execution of research presents ethical challenges that require attention. One objective of the NIH working group, for example, is to investigate the ethical, legal, and social implications of research that uses citizen science methods (Theisz, Couch, and Gillanders 2016), and in 2015, the NIH hosted a two-day workshop focused on these issues (NHGRI 2015). In 2017, the National Science Foundation funded a working group to identify and prioritize ethical concerns in citizen science (Rasmussen 2017). More recently, the Woodrow Wilson International Center for Scholars, a policy forum supported by Congress, conducted a workshop that involved identifying ethical and governance challenges encountered by individuals conducting their own health research (Kuiken, Pauwels, and Denton 2018; Pauwels and Denton 2018).

In all of these forums, the ethical oversight of bottom-up biomedical citizen science activities has been identified as a unique concern. Although many of these activities might be considered relatively safe—for example, altering the genomes of bacteria to make them glow (Lang 2013) or studying the effect of different vitamin regimens on one’s homocysteine levels (Swan et al. 2010)—others are likely associated with elevated risk of harm—for example, subjecting oneself to unregulated interventions, such as homemade gene therapies (Lussenhop 2017).

A growing number of observers have considered the application of traditional research ethics norms to these activities (Vayena and Tasioulas 2013a; Rothstein, Wilbanks, and Brothers 2015; Aungst, Fishman, and McGowan 2017; Guerrini et al. 2018; Fiske et al. 2019; Rasmussen 2019; Wiggins and Wilbanks 2019). For traditional research studies, ethical oversight is usually mandated by some combination of the Federal Policy for the Protection of Human Subjects (Common Rule) and US Food and Drug Administration (FDA) regulations (CFR 2019). Both regulations require institutional review boards (IRBs) to evaluate research protocols according to ethical principles and ensure that participants provide their informed consent. But the Common Rule applies only to federally funded or supported studies involving human subjects or their identifiable data or biospecimens, and the FDA’s human research subject regulations apply only to clinical investigations regulated by the agency. Top-down and collaborative citizen science projects generally comply with the Common Rule and FDA human research subject protections because they involve scientists working in traditional scientific settings (such as government, academic, or industry laboratories) where compliance with ethical guidance is mandatory and monitored. Bottom-up biomedical citizen science activities, however, might be conducted with no input from institution-based scientists, are usually not federally funded or supported, and might not qualify as FDA-regulated investigations. Many of these activities are therefore outside the reach of federal ethics review requirements, creating what has been called an ethics gap in citizen science (Rasmussen 2017).

Biomedical citizen science leaders appreciate this gap and have worked to address it through the development of codes and consensus statements. One of the first efforts of DIYbio.org, which organized in 2008 to promote biomedical citizen science, was to develop codes of ethics for North American and European practitioners (DIYbio 2011). More recently, project and community laboratory leaders from around the world participated in engagement exercises at the Global Community Bio Summit that resulted in a consensus document of ethical principles to guide their work (GCBS 2019a).

Further, some biomedical citizen scientists have expressed interest in voluntarily subjecting their projects to ethical oversight. As a practical matter, some sort of independent ethical oversight must be procured by biomedical citizen scientists who aim to publish their work in journals that require it (Stone 2013). The problem is that bottom-up biomedical citizen scientists who would like to participate in ethical oversight do not usually have access to a research institution’s IRB. Many also might not be able to afford the review fees charged by independent, for-profit boards that have in the past indicated a willingness to review citizen science projects (Stone 2013).

But more fundamentally, the procedures that IRBs utilize and the considerations that they emphasize may not align with the ethos and expectations of biomedical citizen scientists. IRB review was designed several decades ago to help research institutions protect human subjects in studies designed and executed by their employee-scientists (National Commission 1979). It is far from clear that IRB review is a useful oversight mechanism for research activities designed and executed by citizen scientists outside of institutional settings. For example, one of the three ethical principles that guide IRB review—respect for persons—is usually operationalized as a requirement to obtain informed consent from research subjects. But when citizen scientists are both executing and serving as the subjects of research, it is not clear who should provide consent or how to ensure that it is sufficiently informed (Rasmussen 2021).

Recognizing these issues, several new mechanisms of ethical oversight for biomedical citizen science have been proposed. One model involves a crowdsourced review process in which citizen ethicists or citizen review boards conduct nonbinding ethical assessments of research protocols and post their opinions online (Vayena and Tasioulas 2013b). According to an expert consultation model, ethicists and other experts provide guidance to citizen scientists similar to the online program “Ask a Biosafety Officer,” hosted by DIYbio.org for several years (Kuiken, Pauwels, and Denton 2018). Alternatively, community-based committees might provide ethics education and conduct ethical review of projects (Kuiken, Pauwels, and Denton 2018).

Other than an online platform for biomedical citizen scientists conducting self-interventions that tried out a form of crowdsourced review (DIYgenomics 2014), to our knowledge, none of these models has yet been implemented in a systematic or sustained manner, so there is limited information about citizen scientists’ perspectives on or experiences with them. However, biomedical citizen science groups have tested other nontraditional mechanisms of ethical oversight and reflected on these efforts. Among them, a group known as Citizen Science Belleville registered its own IRB that follows different procedures than those followed by traditional IRBs. For example, to promote transparency, the board publishes detailed decisions (CSB 2016; Santos-Lang 2019).

Departing from the external processes that typically characterize IRB review, 21 participants in a self-experimentation study recently tested a system of ethical oversight comprising exercises of ethical self-reflection (Grant, Wolf, and Nebeker 2019). Those exercises included discussion of study risks, risk-management strategies, and potential benefits in group webinars and individual meetings, and they were described as a positive experience for the participants.

Unrelated to specific oversight models, researchers are also beginning to collect data on stakeholder opinions relevant to ethical oversight of unregulated research, including biomedical citizen science projects. For example, in the context of a study focused on unregulated mobile health (mHealth) research, Beskow et al. (2020) conducted interviews with 41 individuals representing four stakeholder groups—patient and research participant advocates; researchers; legal and policy professionals; and mobile app and device developers—about the advisability of external ethical oversight and attitudes regarding how and by whom oversight should occur. Thirteen interviewees were researchers who used mHealth technologies in their studies and were defined to encompass independent researchers and citizen scientists. Over 90% of interviewees believed unregulated researchers should seek out independent ethical oversight, and some endorsed alternatives to IRBs that included mechanisms for obtaining informal feedback from experts.

All of these efforts have contributed to understanding perspectives on ethical oversight of specific biomedical citizen science activities and experiences with distinct models. To our knowledge, however, no study has sought to capture ethical priorities and oversight preferences of biomedical citizen scientists participating in various projects. These empirical data are useful in developing solutions to the problem of the ethics gap in citizen science because they provide a basis for evaluating the likely effectiveness and acceptability of specific features of oversight mechanisms from the perspectives of those whose projects would be reviewed. Here, we report data from qualitative interviews with 35 stakeholders in bottom-up biomedical citizen science from six countries on four continents focused on understanding their attitudes toward ethics and ethical oversight of their work. Based on these data, we consider whether six oversight mechanisms proposed in the literature or considered by citizen science groups align well with the ethical priorities and preferences of diverse biomedical citizen science communities.

Methods

In fall 2019, we conducted in-depth qualitative interviews with individuals engaged in biomedical citizen science projects at two conferences: Biohack the Planet, held in Las Vegas, NV, which is a conference “run by BioHackers, designed for BioHackers, with talks solicited from BioHackers” (Kamau 2017), and the Global Community Bio Summit, held in Cambridge, MA, which is “a space for the global community of DIY biologists/community biologists/biohackers/biomakers and members of independent and community laboratories to convene, plan, build fellowship, and continue the evolution of our movement” (GCBS 2019b). Whereas Biohack the Planet tends to attract citizen scientists who work independently in home laboratories, the Bio Summit is geared toward citizen scientists who work in community laboratories and other nontraditional laboratory settings. We conducted interviews at both meetings in an effort to capture diverse opinions and experiences.

Before each conference, potential candidates were identified from published lists of conference attendees, with input from one author (AP) who is a veteran attendee of both conferences and was a consultant to this study. To be considered for inclusion, each candidate was required to be at least 18 years old; speak fluent English; and attend one of the conferences. Three authors (MT, CG, IC) conducted the interviews using a semi-structured interview guide, which included questions relevant to the interviewee’s attitudes about, preferences for, and experiences with ethics, ethical oversight, and six oversight mechanisms. (All mechanisms were not covered during every interview owing to time constraints.) Interviews were recorded with permission and professionally transcribed. Each interviewee was asked to verbally consent at the start of the interview and was offered a $75 gift card at the end of the interview. The mean interview length was 50 minutes.

Four authors (MT, CG, WB, IC) developed a preliminary codebook based on analysis of two interview transcripts. The preliminary codebook was pilot tested and refined as a result of that process. Each transcript was then independently coded according to the final codebook by different combinations of two of the same four authors. Finally, coded reports were reviewed by all authors to identify salient themes and to organize the data.

The study was designed, analyzed, and reported consistent with the Consolidated Criteria for Reporting Qualitative Research (COREQ) (Tong, Sainsbury, and Craig 2007). Additional details regarding methods can be found in Supplemental File 1: Methods.

Results

Results are reported as follows: (1) descriptive characteristics of interviewees; (2) ethical priorities; (3) attitudes relevant to ethical conduct of research; (4) preferences for features of ethical oversight; (5) opinions of select ethical oversight models; and (6) barriers to implementation. Although some interviewees chose to participate on a nonconfidential basis, no interviewee is identified by name. Rather, to avoid any bias from consideration of identified versus de-identified data, quoted information is attributed to each interviewee using an assigned number.

Opinions of select oversight models

Interviewees’ opinions of specific oversight models for biomedical citizen science provided further information about the acceptability and feasibility of potential features of ethical oversight. Six models that have been proposed in the literature or considered by citizen science groups were discussed with interviewees: traditional IRB review, expert consultation, community review, crowdsourced review, systematized self-reflection, and codes of ethics (Figure 2).

Figure 2 

Six ethical oversight models.

Table 2 summarizes key advantages and disadvantages of each model from the perspective of interviewees. Briefly, interviewees explained that the use of traditional IRBs to evaluate their work could increase its perceived legitimacy. However, IRBs were criticized as elitist, hierarchical, and prone to abuses of power, as well as too formal and time-consuming for many biomedical citizen science activities. It was generally agreed that expert consultation could provide valuable insights and guidance as a “sounding board” for community members (033). But interviewees worried about the difficulty of identifying experts who understand their communities’ values and priorities and would engage with them over the long term.

Table 2

Interviewees’ opinions of select ethical oversight models.

OVERSIGHT MODEL	ADVANTAGES	DISADVANTAGES
Traditional IRB review	Obtain recommendations from experts in the ethical review of scientific research Might increase perceived legitimacy Might preempt/mitigate criticisms by demonstrating serious commitment to ethics review	Difficulty accessing an established IRB or developing one’s own IRB Elitist and hierarchical Too formal and time-consuming for low-risk activities
Expert consultation	Obtain recommendations from variety of experts Might facilitate mentorships	Difficulty finding the right experts Unclear how to engage experts long term
Community review	Obtain recommendations from peers in community Community driven Might be similar in structure to existing safety review boards in the community	Could be elitist and hierarchical depending on who is allowed to participate Resource intensive
Crowdsourced review	Obtain feedback from designated peers in community or outsiders Decentralized and not elitist or hierarchical	Difficulty identifying the right citizen ethicists If online, potential for trolling and misunderstanding
Systematized self-reflection	Elicit reflections from those directly engaged in projects Might yield insights for other projects	Unclear how to manage different ethical opinions Resource intensive
Codes of ethics	Identify and communicate shared values Community driven Useful as a general guide to decision making	Incapable of answering ethical questions in specific circumstances

With respect to internal governance models, interviewees noted that advantages of community review include that it is culture-built and can be modeled after existing community safety review boards. Depending on the structure, however, it might be elitist or hierarchical and require an unsustainable amount of time and resources. By contrast, interviewees explained that the crowdsourced review model, which invites input from designated peers and non-biased outsiders, is decentralized and not prone to abuses of power. However, concerns were raised about how to select individuals to serve as citizen ethicists, and if reviews are posted online, whether they will provoke trolling and contribute to misunderstanding, which are issues that already plague some communities’ online discussion boards.

Interviewees had a generally favorable view of systematized self-reflection because it engages all individuals directly involved in specific projects and might yield insights for other projects. However, this model might be too resource-intensive for specific projects, and interviewees were unsure how differences of ethical opinion would be managed. Finally, while many interviewees believed that codes of ethics are useful in demonstrating their commitment to ethical conduct, one interviewee questioned the value of codes given that they cannot provide specific guidance or answers to questions. Representing an extreme opinion, one interviewee described codes of ethics as “garbage” for the reason that “no one will read [them]” (001).

Discussion

To our knowledge, this is the first study to probe a global sample of biomedical citizen science stakeholders about their ethical priorities, attitudes toward ethical oversight, and ethical oversight preferences. Interviewees’ ethical priorities related to autonomy and inclusivity were endorsed by biomedical citizen scientists in prior studies. (Sanchez Barba 2014; McGowan et al. 2017; Guerrini et al. 2020b). Good science is also consistent with efforts to promote the acquisition of good-quality data in citizen science (Kosmala et al. 2016). More generally, the ethical priorities identified by our study participants align with ethics guidance developed by biomedical citizen science groups, which is not surprising given several interviewees’ involvement in those efforts (DIYbio 2011; GCBS 2019a).

In some cases, interviewees’ conceptualization of ethical priorities provided a fresh take on those described in the Belmont Report, which provides an analytical framework to guide consideration of ethical issues in US human subjects research (National Commission 1979). For example, the Belmont Report describes respect for persons, operationalized through a requirement to obtain informed consent, as a guiding principle in research ethics. Interviewees also endorsed this principle but viewed it through a lens that emphasizes rights of self-determination, so that in some cases, respect for persons might mean allowing them to take risks of harm to self. The Belmont Report also describes justice as a guiding principle and interprets it to require equitable selection of subjects and distribution of research benefits and burdens. By contrast, our interviewees conceptualized justice as commitments to the diversity of all participants in scientific knowledge production and equal and open access to the tools and fruits of research. Further, interviewees identified ethical priorities that go beyond those described in the Belmont Report, such as commitments to communities, that might serve to, as Ikemoto (2017, p. 566) observed, “situat[e] science as a tool for social justice.” These findings suggest that, depending on the biomedical citizen science project, reliance solely on the Belmont Report (and the Common Rule that builds on it) to guide its ethical review might not be appropriate or sufficient from the perspective of participants (Rasmussen 2019; Rasmussen 2021).

Indeed, given the diversity of the actors and activities in biomedical citizen science, it is clear that there will be no one-size-fits-all solution to the problem of the ethics gap. Nevertheless, interviewees were able to agree on some preferred features. Specifically, they endorsed ethical oversight mechanisms that are voluntary, community-driven, and offer guidance. Conversely, they rejected mechanisms that are mandatory, inflexible, and hierarchical. Triangulating these results with interviewees’ opinions of specific oversight mechanisms, the expert consultation and community review models appear to be consistent with the preferences, priorities, and needs of many biomedical citizen sciences communities, especially if participation in these mechanisms is voluntary and they function in advisory capacities. However, systematized ethical reflection might work well for specific projects involving a limited number of participants, and codes of ethics can be useful in establishing general standards of conduct. By contrast, traditional IRB review is generally not practical or trusted, whereas crowdsourced review is likely to be unwieldy if conducted online.

Interviewees’ concerns about IRB review in particular are similar to those voiced by investigators using community-based participatory research (CBPR) approaches. In an interview study, CBPR researchers described frustrations with having their work reviewed by outsiders who might not understand their research contexts, objectives, or ethical concerns (Shore 2007). One interviewee felt traditional IRBs rely on regulations “written originally for particular, non-participatory, more traditional kinds of research, particularly empirically based, quantitative research,” and do not translate well to community-based research (Shore 2007, p. 37). In practice, this disconnect can lead to IRBs making determinations that delay or even derail CBPR projects.

In prior research, we reported biomedical citizen scientists’ frustration with and distrust of traditional scientific and medical institutions (Guerrini et al. 2020b). Interviewees in this study echoed those sentiments and channeled them into commitments to community-based solutions to ethical oversight that did not recreate oversight mechanisms perceived to be elitist and hierarchical. However, solutions that rely on internal review might be in tension with recommendations that biomedical citizen scientists avoid participating in the review of their own work on grounds that they will not be able to objectively assess its risks and benefits (Rothstein et al. 2020).

Despite their worries about involvement by outsiders, interviewees generally did not oppose—some even welcomed—ethical guidance from independent experts, although they stressed that such experts would need to respect their communities’ culture for those partnerships to be successful. In 2018, the Wilson Center brought together regulators, scholars, and biomedical citizen scientists to identify ethical and governance challenges encountered in health self-research (Kuiken, Pauwels, and Denton 2018). Our results support continuation of these relationship-building efforts, especially at the project level. We urge bioethicists in particular to keep an open mind if approached by biomedical citizen science communities and recommend further research to clarify every party’s liability and reporting obligations, which might mitigate potential concerns associated with entering such partnerships.

Our findings are subject to several limitations. First, the data are not generalizable to all biomedical citizen scientists or their communities. Second, our interview population was limited to individuals who physically attended at least one of two conferences. Conference attendees might have had more resources or been better connected within and across communities than non-attendees. Further, ethical issues were discussed during scheduled sessions at both conferences. As such, conference attendees may have been more interested in, or might have more deeply considered, the ethical implications of their work compared with non-attendees. Third, although our sample included biomedical citizen scientists from around the world, the majority resided in the United States. As a result, our data likely underrepresent practices and norms that are common outside the United States. Finally, like other studies involving biomedical citizen scientists (Sanchez Barba 2014; Zarate et al. 2016; Guerrini et al. 2020b), the majority of interviewees were male. However, through careful pre-selection of potential candidates and snowball sampling, we sought to maximize diversity and the data reflect a wide range of experiences and perspectives.

Conclusion

Looking ahead, we support continued investigation of the use of various oversight mechanisms in biomedical citizen science communities and efforts to facilitate collaborations with experts and understanding by regulators and other stakeholders. More generally, we recommend further research of the role and application of ethics in biomedical citizen science settings. Given that traditional research ethics principles and practices do not appear to always translate well to these settings, it might be time to consider the development of new ethical frameworks for biomedical citizen science.

Supplementary Files

The supplementary files for this article can be found as follows: