Not whether but how: the ethics and Language of reporting back individual results

Abstract

Background

The ethical obligation of reporting back research results (RBRR) is established. Yet the ethical dimensions of how RBRR is implemented remain largely unexplored. The Environmental influences on Child Health Outcomes (ECHO) program has identified logistical challenges in RBRR, citing limited guidance resources as a key obstacle. The objective of this research was to characterize and assess ethical considerations for RBRR: (i) with or without clinical significance; (ii) within a pregnancy cohort, and; (iii) within a study population with diverse cultural and traditional contexts.

Methods

Expert Panelists (n = 20), including exposure scientists, community engagement specialists, public health researchers, clinicians and ethicists convened virtually and completed short surveys to identify ethical considerations regarding how RBRR is conducted. While the original objective was to characterize ethical guidelines for RBRR across three potential outcomes, the Panel identified a need for RBRR harmonization; conflicting and inconsistently applied terminology and definitions prevented the Panel from fully discussing the original topics.

Results

Ethical considerations for conducting RBRR were mapped to the principles of bioethics (respect for autonomy, beneficence, nonmaleficence, and justice), with Panelists suggesting that additional ethical frameworks could be used in parallel or as a replacement. However, when considering results with or without clinical significance, the Panel identified a clear need for standardized terminology when describing RBRR, particularly regarding the relevance of results to individual health, behavior, and knowledge. The Panel recommended the following related to RBRR: (i) develop centralized resources; (ii) develop multi-use materials; (iii) ensure clear assignment of RBRR responsibility within research projects; (iv) encourage active engagement of research participants in the RBRR process to foster and maintain trust; (v) recognize community-level harms and benefits; (vi) extend RBRR beyond study completion; and, (vii) establish standardized language.

Discussion

The Panel highlighted challenges such as non-standard terminology and difficulty discerning between RBRR with clear health or regulatory standards versus results without established, quantified health relationships. The Panel affirmed all research results, regardless of established health relationships, that benefit participants should be returned. While RBRR guidelines were considered universal, the Panel emphasized the need for nuanced considerations, particularly when working with culturally diverse or pregnant populations. Future work should address ethical considerations regarding RBRR’s responsibilities for depositing data in a public-use repository.

Supplementary Information

The online version contains supplementary material available at 10.1186/s12940-025-01224-0.

Introduction

ECHO is a national, federally-funded program that seeks to understand the effects of early environmental influences on child health and development across five focal areas: pre-, peri-, and postnatal health; upper and lower airway health, obesity, neurodevelopment, and overall well-being [21]. Research in ECHO can be observational in nature (identifying environmental exposures and observing children’s health) and/or can be structured as an intervention (testing changes to improve children’s health) [18]. The consortium initially had essential and recommended data collection elements across all cohorts, enabling harmonization across years and coordinating centers [18, 21, 43]. In 2016, the National Institutes of Health used the first ECHO cycle to integrate existing pediatric longitudinal observational cohorts across over 200 sites in the US and Puerto Rico, consolidating data on over 60,000 children and primary caregivers [18, 21]. In the second cycle (2023–2030), data and biospecimens are collected from existing and new ECHO participants across 72 study sites using a single standardized protocol [5].

Given the ongoing data and biospecimen collection, in Cycle 1, ECHO developed and ratified the ECHO Cohort Consortium’s Return of Individual Research Results Policy, which seeks to establish a clear framework for returning individual research results to ECHO participants who express interest in receiving such information [14]. There has been substantial interest in, and research on, the report-back of research results (RBRR), with comprehensive evidence detailing the benefits of RBRR across various populations [22, 30]. However, there is limited guidance for how to conduct RBRR, particularly with pregnant individuals. Preliminary work indicates that the same benefits of increased research transparency and increased knowledge exist [37] but to supplement this work further, an in-depth exploration into the ethical dimensions of reporting individual results in a pregnancy cohort was undertaken. The investigation aimed to address the bioethical complexities of providing ethically informed reports to a culturally diverse group of pregnant individuals. This work was undertaken to support RBRR to culturally diverse pregnant individuals and is responsive to recent guidance from ECHO’s RBRR policy document [14].

In January 2020, the ECHO consortium ratified an “ECHO Individual Return of Research Results Policy” statement that outlined the principles and framework for returning individual-level results to participants interested in receiving them [14]. The policy rests on four fundamental principles: (1) participant engagement is essential to the ECHO program; (2) participants live in families and communities that are also ECHO research stakeholders; (3) the ECHO program encourages the creation of enhanced capacity to engage participants and communities; and (4) the ECHO program is transparent and trustworthy [14].

Previous research has carefully examined the ethical responsibilities for RBRR. Researchers have applied the bioethics framework to determine the appropriateness of returning data to study participants [8, 9] National Academies of Sciences and Medicine 2018). Research has further evaluated the question within the framework used by Institutional Review Boards [10, 33]. Ethical considerations were mapped to the principles of respect for autonomy, beneficence, nonmaleficence, and justice, with additional considerations about environmental justice [38]. Report-back of research results is beneficial to participants, even without known health effects, regulatory standards, or health guidelines [1, 9] National Academies of Sciences and Medicine 2018, [44]. In sum, a robust ethical rationale supports RBRR in studying participants [1] National Academies of Sciences and Medicine 2018). However, ethical considerations forhowto return data have not been similarly characterized [22]. Further, how to conduct RBRR in the context of unknown health impacts and within a culturally diverse pregnancy cohort remains uncharacterized [26]. This is a key challenge for researchers responsible for returning individual and/or aggregated results to study participants in their respective study sites [24]. Here, the results of a study conducted to characterize and assess the ethical considerations about the return of results with and without clinical significance to individuals within a pregnancy cohort are presented. We describe research results for RBRR as the interpretation of research findings, which may include clinical results (e.g., blood lead levels, or health-based levels for arsenic in well water) as well as the results of personal chemical exposures. As described in the 2018 National Academies of Science report, individual environmental exposure results, as well as biospecimen results, may be reported back to study participants (National Academies of Sciences and Medicine 2018). This initial work focused solely on the ethical development and dissemination of reports and, therefore, will underrepresent the totality of ethical considerations. In particular, the study was informed by the following key ethical considerations, recognizing that many of the examples provided below may apply to more than one principle:

• Respect for Autonomy: This principle is broadly defined as ensuring that research respects and supports autonomous decisions and ensures that research participants are able to make informed decisions about their choice to participate in research activities [3, 29]. As such, RBRR should be developed to aid comprehension and designed for accessibility [12, 44, 46] to support participants autonomous interpretation and use of results. RBRR should additionally respect and address the cultural considerations of the study population and larger community [42].

• Beneficence: Research should be designed to maximize benefit, either to the study participant directly, and/or to the broader community, within the context of balancing potential risks and benefits [3]. Research indicates that RBRR supports participants better understanding how their environmental exposures may impact their health, a concept commonly referred to as environmental health literacy. Therefore, RBRR development should follow the principles of environmental health literacy [16, 20, 38] and support participant decisions to understand and reduce their exposures, even after their child has been born [26, 40].

• Nonmaleficence: While beneficence seeks to maximize benefit, the principle of nonmaleficence seeks to characterize and minimize harm [3]. In recognition of this principle, reports should be designed to explain the limitations of studies evaluating personal exposure to contaminants without known health effects (National Academies of Sciences and Medicine 2018). Additional considerations for pregnancy cohorts may include acknowledging and addressing the time lag between data collection (e.g., during pregnancy) and the return of results (e.g., after the baby has been born).

• Justice: Bioethics recognizes that research benefits, risks, and costs, should be equitably distributed [3]. Under the definition of vulnerable populations as “likely to be vulnerable to coercion or undue influence (Office for Human Research Protections [32],” medical ethicists have argued that pregnant individuals may be considered vulnerable, especially if they encounter an identifiable higher exposure to risk due to lack of knowledge, susceptibility of coercion, and vulnerability to the fetus [48]. However, further clarification suggests that pregnant individuals may be more vulnerable given historical exclusion from research and, therefore, lower scientific knowledge (e.g., unequitable distribution of research benefits) [48]. As a result, this may prevent the participation of pregnant individuals in research and, therefore, RBRR [23]. RBRR is designed to ensure that all individuals benefit from research, including pregnant people, as aligned with the principle of justice [26].

We convened a 20-member Expert Panel of exposure scientists, community engagement specialists, public health researchers, clinicians and ethicists to identify and characterize ethical considerations related to returning data without clinical significance to a culturally diverse pregnancy cohort. The Expert Panel was tasked with critically examining the ethical considerations surrounding data return, specifically focusing on the following three key scenarios: (i) data lacking clinical significance; (ii) within a pregnancy cohort; and (iii) within a study population with diverse cultural and traditional contexts.

Methods

Recruitment & meeting structure

We convened an Expert Panel of 20 individuals representing diverse organizations, disciplines, and expertise (Table 1) from November 2022 to February 2023. Prior to recruitment, the research team predetermined three topics for discussion with the Panel: (1) results with/without clinical significance; (2) reporting back results to a culturally diverse study population; and, (3) reporting back results within a pregnancy cohort. To facilitate robust, informed discussion amongst the Panel, we sought expertise across several disciplines: community engagement, clinical medicine, exposure science (toxicology, chemistry, and environmental epidemiology), public health, perinatal health, and bioethics. The research team purposefully selected individuals across these disciplines; three research team members independently developed a list of Panelists. The lists were compared, and each team member identified the qualifications and expertise of the individuals on their list and their applicability to the discussion topics. Full consensus was reached on a final list of 25 potential Panelists; 20 accepted the invitation, providing expertise across bioethics (n = 4), exposure science and/or perinatal health (n = 7), clinical medicine (n = 3), and community engagement (n = 6), as shown in Figure S1. When we reached out to individuals we included a brief background of the research, the anticipated schedule, and a description of the Expert Panelist roles and time requirements.

Table 1.

Expert panelist affiliations and experience with report back of research results

Expert Panelist Affiliations	N (%)
Institutional Review Board/Human Protection Administration/Human research ethics	4
Research (toxicology, environmental exposure, children’s health, epidemiology, medical ethics, health policy)	7
Clinical medicine	3
Community Outreach Liaisons/Community engagement specialists	6
Total	20
Meeting Participation (n, %)
Meeting #1 – Nov. 1, 2022	17 (85%)
Meeting #2 – Nov. 10, 2022	19 (95%)
Meeting #3 – Feb. 28, 2023	17 (85%)
Question
How familiar are you with the concept of report back of research data to study participants?0 = not at all familiar, 10 = extremely familiar
Average	6.9
Range	4–10
SD	1.73
Responses (n, %)	10 (50%)
	Count (%)
Have you returned research data with clinical significance to participants?
Yes	5 (25%)
No	3 (15%)
Unsure	1 (5%)
No response	11 (55%)
Have you returned research data without clinical significance to participants?
Yes	8 (40%)
No	1 (5%)
Unsure	0 (0%)
No response	11 (55%)
Have you returned research data to a pregnancy cohort where exposure was assessed during pregnancy?
Yes	2 (10%)
No	6 (30%)
Unsure	0 (0%)
No response	12 (60%)

The members of the Expert Panel brought varying levels of familiarity with RBRR, particularly RBRR of data without known health outcomes within pregnancy cohorts. The structure of the virtual Panel meetings was guided by the Delphi Method [34, 45], employing iterative rounds of questionnaires, controlled feedback, and statistical representation of the group responses (Figure S1; Table S1). After each meeting (n = 3), Panelists received a brief survey using Likert scales, multiple-choice, and open-text questions (Tables S2-4). The responses were summarized, and at the subsequent meeting, Panelists were engaged with concepts and topics related to ethically designed report-back.

All Panel meetings were audio-recorded and transcribed. Before the meetings, the research team reviewed and summarized survey responses to present, in graphical form, the concepts and topics related to ethically designed report back (Figure S1). Each meeting was moderated by a single member of the research team. The moderator facilitated introductions and then provided an overview of the Expert Panel tasks for that meeting (Table S1). For meetings two and three, the moderator then provided the analysis and review of prior survey responses (Tables S2-4). As the results were presented, and as the meeting segued into new discussion topics (Table S1) Panelists were encouraged to respond and discuss. Two other research team members were present and helped facilitate the discussion by providing additional information as needed and ensuring that all Panelists had an opportunity to speak. Two note-takers were present as well. Each session typically lasted 1.5 h.

As previously stated, the research team predetermined three topics for discussion – reporting back results: (1) with/without clinical significance; (2) within a culturally diverse study population, and; (3) within a pregnancy cohort. These areas were introduced in Meeting #1. In Meeting #2, using the conversation from the first meeting, as well as initial survey results, we identified seven ethical considerations for further discussion: time lag, environmental ‘choices’ and subsequent ability to reduce exposures; results with cultural and/or traditional implications; results with potential future clinical significance; sensitive research uses of results; the potential for group/community-level harm, and; potential to harm trust relationships between researchers and those being researched.

Originally four meetings were planned. However, in Meeting #3 Panelists reported confusion regarding the terminology and differing definitions of terms, which was preventing them from answering survey questions or even discussing specific topics. Therefore, in lieu of meeting again, the research team took the results of the previous meetings and surveys to consider new language and definitions for key terms that had been identified by the Panelists and identified short example research scenarios that would exemplify the different considerations related to how to return individual results to participants (e.g., returning individual results wherein some information is known, such as chemicals that are carcinogens, but where no regulatory or health-based values exist).

This study was reviewed by the Institutional Review Board at Columbia University as part of the overarching Fair Start Study (IRB-AAAK6753) and was not considered human subjects research.

Thematic analysis

The transcripts of the audio recordings were downloaded and used for thematic analysis. Initially, the data was organized according to discussion prompts, facilitating a structured examination, by each question, as a data organizing effort. This enabled analysis of ethical considerations specific to pregnancy cohorts, recommendations, and harmonization of RBRR language. Subsequently, to apply the bioethical principles, a secondary analysis was conducted, using a priori codes (respect for autonomy, beneficence, nonmaleficence, justice) to identify overarching ethical principles. Each transcript was individually coded, with discrepancies discussed among the research team until a consensus was reached.

Similarly, the free-text survey responses from the Expert Panel were analyzed. Initially, the data was organized by question prompts and analyzed by each prompt. The open-response survey answers were de-identified and imported into Dedoose qualitative analysis software along with manual coding using colored highlighters. A hybrid inductive approach with aspects of a deductive thematic analysis was used, as has been described in the literature [11, 41]. An open coding technique was first used to generate initial codes by a single coder [25]. Subsequent rounds of modifying and refining codes (n= 2) were documented in a modified codebook where codes were named, described, examples provided, along with memos of reflexivity during the coding process and tracking of the evolution of codes for transparency [11, 41]. The inductive approach was opted for initially to ensure that no concepts would be lost if they were outside of the pre-defined codes. The question prompts, expertise involved, and context of the study drove respondents to contextualize their answers within a priori bioethical principles described above. Resultant codes were then mapped to the bioethical principles (respect for autonomy, beneficence, nonmalfeasance, justice) allowing for saturation of participant ideas and alignment with the guiding bioethical framework.

The a priori codes (respect for autonomy, beneficence, nonmaleficence, justice) are the four ethical principles of bioethics, as defined by the Belmont Report [29] and Principles of Biomedical Ethics [3]. Prior work argued the ethical imperative for conducting RBRR by analyzing these four principles [27, 28]; as such we assessed the ethical considerations for how to conduct RBRR using these same principles. Furthermore, we note that increasingly, RBRR methods and materials are being evaluated by Institutional Review Boards, which utilize these four principles to conduct their review [10, 33]. Recognizing that bioethics may not encompass all ethical considerations [27], Panelists were directly asked to recommend additional ethical frameworks, although only 5 Panelists suggested alternatives.

Quantitative analysis

Likert scale data was analyzed using a weighted rank analysis. Multiple choice question responses were summarized. Only summary statistics were compiled due to the small sample size and question format. While multiple questions were included in the surveys (Tables S2-4), only a subset of information is provided. Given the confusion expressed by Panelists in the meetings, and reiterated in Survey #2, many of the survey results were unable to be analyzed (e.g., responses clearly showed that participants misunderstood the question, or they provided a response that was not aligned with the question prompt). Therefore, a subset of data was analyzed. The focus of the meetings shifted following Meeting 3, necessitating a more narrow focus on language harmonization, and preventing additional discussion on the core topics of original discussion.

Review process

The research team developed the draft manuscript and was informed by the thematic and quantitative analysis. We conducted a modified version of member checking [4] by sharing the manuscript with all Panelists, who were given two weeks to provide feedback and approve quotes, findings, and overall recommendations (Figure S1).

Results

Description of expert panelists

As described in Tables 1 and 20 Panelists were convened. Panelists represented expertise in bioethics, exposure science, perinatal health, clinical medicine, and community engagement. Following Meeting #1, all Panelists were asked to complete a survey (Table S2; Figure S1). Unfortunately, participation was suboptimal with only 10 Panelists responding. Of the responses obtained, participants self-identified as moderately familiar with the concept of RBRR, although few had returned research results, and only two individuals stated they had returned research results to a pregnancy cohort. Survey participation improved throughout the project, with 60% of Panelists responding after Meeting #2, and 80% responding after Meeting #3. On average, 17 Panelists attended each meeting (Table 1).

Bioethical considerations informing the development of personal exposure report back of research results

Through three structured meetings and surveys, the Expert Panel identified several bioethical considerations when reporting research results to a culturally diverse pregnancy cohort. During the first meeting, the research team identified the three key contexts for RBRR (with or without clinical significance, RBRR for pregnant cohorts, and RBRR for culturally diverse populations). Following that meeting, Panelists engaged in discussion and provided survey responses to identify potential ethical considerations. Following thematic analysis, several considerations were identified, discussed by Panelists and then subsequently mapped to bioethical principles: Respect for autonomy - communicating results with cultural and/or traditional implications, managing the potential for non-research uses of results, and messaging around environmental ‘choices’ (e.g., smoking, use of air fresheners, etc.) and subsequent ability to reduce exposures; Beneficence – the potential for group/community benefit; Nonmaleficence – the potential for group/community harm, the potential for RBRR to harm, and the time lag between data collection and RBRR; Justice - returning results with potential future clinical significance; and; the potential for RBRR to harm. The discussions and survey responses were mapped to the bioethical principles. The considerations identified by the Panelists often addressed more than one ethical principle – we have endeavored to place them under the primary principle.

Respect for autonomy

Panelists emphasized the foundational principle of respect for autonomy in report development, highlighting accessibility, respect for cultural and community norms, and providing clear explanations to ensure participants’ autonomy. One Panelist underscored the need for reports to acknowledge and respect “differences in what is legal/acceptable in different states and Nations,” recognizing the diverse norms within participant groups and communities, which encompass geographical, cultural, sovereign, socioeconomic, and occupational distinctions.

Privacy and confidentiality in the conduct of RBRR emerged as a significant topic of discussion for the Panel. Panelists originally identified this as concerns related to non-research uses of results. While tasked with identifying ethical considerations for conducting RBRR, concerns regarding the scope of the RBRR became evident. Panelists cited legal challenges, such as Ferguson v. City of Charleston, wherein healthcare workers screened pregnant people for cocaine and released the results to local police for prosecution, citing harm to the fetus [36]. While the Supreme Court ultimately ruled that drug tests could not be done without consent or a warrant, the ramifications have led to fears that pregnant people are being criminalized for acts that could potentially harm the fetus [36]. Panelists drew a link between this legal example and concerns that the results of exposure sampling could be used for purposes other than research. While there is a noted difference between research data and healthcare data, examples such as these underscore the complex legal and social landscapes within the RBRR.

The Panel discussion expanded to discuss respect for communities. Recognizing the stratification within communities based on gender, age, socioeconomic status, and priorities, the conversation emphasized the importance of understanding these nuances for addressing ethical concerns effectively. For example, the Panelists discussed the potential harm RBRR could inflict on groups and underscored the necessity of considering cultural and traditional practices that may be impacted. A Panelist provided one example, who discussed the conventional significance of personal care products that may contain potentially harmful products. While RBRR should include optional mitigation methods to reduce exposures, these methods must be discussed sensitively and appropriately. The Panelists also acknowledged that RBRR could have ripple effects, whether negative or positive, extending beyond the immediate participant group.

Beneficence

In developing RBRR, the Panel underscored the importance of maximizing research benefits for individuals and the community. This holistic approach emphasizes identifying resources and strategies to mobilize communities and enact policy change. Such an approach draws inspiration from the practices of several Indigenous Institutional Review Boards, which acknowledge the complex interplay between individuals and their communities and can be applied to environmental health [17].

The Panel recognized the need to develop report-back strategies that empower participants to understand their results from past sampling efforts and ongoing exposures. By doing so, the results presented in reports can continue to be relevant and impactful as children are born and progress through early childhood development stages. This forward-thinking approach extends the benefits of the research beyond the individual being sampled, potentially informing behavioral changes to reduce exposure levels or promote the health of the current family and future generations. As one Panelist suggested:

When we think of maximizing benefits to the participant, whether there are known standards or not, we should be providing results in a way that (1) is clear and meaningful – using language that is accessible and presenting data in a digestive manner; (2) motivates actionable steps to reduce exposure and the potential for adverse health effects, (3) informs the participant about whether the results are clinically significant or have an unquantifiable/unknown potential for adverse health effects.

Panelists also noted that RBRR may need to be accompanied by resources for clinicians to ensure full benefits are realized. As one Panelist wrote, “Supporting documents for clinicians would be helpful. Otherwise, participants who may already feel scared from their results may feel worse after going to their healthcare provider, who has no additional information to offer them.”

Additionally, the Panel recognized that while beneficial resources and mitigation strategies must be shared, there must also be an acknowledgment of scenarios where little can be done to arrest or reverse potential health effects. For example, as Panelists noted, exposure to some contaminants, such as lead, causes irreversible health effects.

Finally, considering the breadth and depth of potential RBRR efforts, the Panel expressed support for centralized RBRR resources, particularly within the context of the ECHO consortium.

Nonmaleficence

The Panel engaged in thoughtful discussions regarding the design of reports, particularly considering the potential psychological risks, especially within pregnancy cohorts. Panelists noted that RBRR must be carefully developed to avoid accidentally placing blame on participants for their exposures. There was a shared concern that reports might inadvertently trigger feelings of guilt or hopelessness among participants. Balancing the imperative to provide information with the need to minimize distress or anxiety was a central consideration. These discussions reflected how reports should be tailored to ensure relevance and applicability, addressing questions pertinent to the community. While the primary focus was on mitigating harm, underlying these deliberations were core principles of respect for the autonomy of individuals and communities.

The Panel additionally discussed the design of reports within a cultural context, noting that research teams need to plan for different levels of understanding within specific contexts and cultures. As one participant asked: “Who helps develop these materials? Are they pilot-tested with participants? With other naive audiences? What is accessible and helpful is very subjective, of course.”

The survey and subsequent discussion also asked who would develop the reports. While the survey results (Fig. 1) showed disagreement, when we examined the written responses, we saw respondents coalesce around the idea that a multi-person approach is needed. The Panel agreed that while the principal investigator (PI) should interpret the data, a committee approach will ensure the information is easily understood, sensitive to community concerns, can integrate medical advice as appropriate, and can represent the voices of the cohort.

Fig. 1.

The Expert Panel was shown several statements regarding who should be responsible for interpreting results and preparing the reports. Respondents ranked how strongly they agreed or disagreed on a Likert scale. PI = Principal Investigator. Individual points represent a single respondent. Horizontal lines indicate the weighted rank

I think we need the PI, of course, to interpret the data and make it understandable. The committees described above, which involve others and not just researchers, would be able to bring in other perspectives or ways of framing and presenting information so it can be easily understood and presented with sensitivity.

I am not sure community engagement specialists should be responsible for interpreting the data. I think the interpretation must include individuals with a scientific or medical background. Community engagement specialists can help with identifying best messaging or communication strategies.

Thus, considering the potential for RBRR to harm trusted relationships, Panelists emphasized the iterative process for RBRR, advocating for participant involvement from research initiation. They stressed the importance of participants in decision-making regarding data-sharing, suggesting transparent communication about research timelines, results dissemination, and ongoing updates. The Panelists also noted that the RBRR process should extend beyond result reporting to continued follow-up, resource provision, and participant engagement as research evolves or new information emerges. Through such sustained efforts, the RBRR process can facilitate the “sharing of information with other members of participants’ local community,” thereby fostering broader knowledge sharing and engagement.

Justice

Related to the principle of justice, the Panel underscored the need for prioritizing messaging highlighting the parent-child dyad’s health. While the recommendations were intended to be universally applied, the Panel emphasized the importance of sensitivity when working with sensitive populations, such as pregnant individuals.

Ethical considerations regarding the timing of RBRR, specifically the lag between data collection and the return of results, commanded significant attention. This topic was addressed in two ways: (1) return of results when clear connections to health are known, and; (2) return of results in the absence of known health effects. For preliminary conversations, the term ‘clinical significance’ was used. Initial results showed strong consensus regarding results with clinical significance (e.g., blood lead levels), yet when asked about the timing of when to conduct RBRR, Panelists did not agree regarding the return of results lacking clinical significance (Fig. 2). Further, there was strong disagreement about the latter and whether RBRR should occur before or after the peer-review of associated scientific articles. Conventionally, research data lacking established health guidelines undergoes a meticulous process involving collection, analysis, interpretation, and peer review by subject matter experts before being made publicly available. However, ethical concerns arise when considering whether to provide results immediately, risking potential misinterpretation or inaccuracies, or delay until the completion of the peer review process, thereby withholding potentially beneficial information from participants that could inform behavioral or preventative measures. A Panelist noted that this time lag becomes particularly contentious with emerging chemicals of concern, citing the ongoing community concerns around PFAS, or per- and poly-fluoroalkyl substances [49]. Withholding research results for internal scientific processes, such as peer review, may thus cause harm.

Fig. 2.

Expert Panelists were asked to agree or disagree with the following statements using a Likert scale: (1) results WITH clinical significance should be returned to participants as soon as they are available; (2) results WITHOUT clinical significance should be returned to participants as soon as they are available; results WITHOUT clinical significance should go through peer review before they are reported back to participants. Individual points represent a single respondent. Horizontal lines indicate the weighted rank

A concept related to data equity emerged during the discussion. Panelists discussed the current norms governing chemical data, observing that depending solely on established standards needed to be increased. They emphasized that waiting for guidelines before acknowledging potential harm may be unethical, as outlined by the prior PFAS example. They highlighted the importance of transparency in reporting data even in the absence of standards, noting that it facilitates comparison in the future and fosters a more open approach to simply providing a binary present or absent report. Notably, research participants find benefit in receiving results, even when the association between exposure and health is unknown or nebulous [1, 7, 9, 40].

Finally, the Expert Panel discussed potential actions or changes in behavior that a participant may take upon receiving their results and the implications this might have for how results are interpreted, and the subsequent impacts on health.

For instance, consider any socio-economic impacts if a family decides that they want to mitigate exposure by getting all new furniture, or any social consequences upon learning that some action or inaction on their part led to increased exposure for their child.

In sum, the Panel agreed that follow-up, resource provision, and the implementation of mitigation strategies are indispensable elements for RBRR. These provisions must be accessible across various economic strata and culturally sensitive to ensure program effectiveness. However, this conversation was ultimately stalled as Panelists began to query the definitions and applications of the terms being used, particularly ‘clinical significance.’ These discussions led the Panel to an unexpected discussion around RBRR harmonization, such as developing generally agreed-upon definitions for terms within the discipline, which was the focus of the final meeting and survey.

Considerations specific to RBRR with pregnant research cohorts

Throughout the discussions, the Panel was tasked with contextualizing the above bioethical concerns within the framework of a pregnancy cohort. Through survey responses, the Panel identified the following additional considerations: (i) legal and political contexts unique to pregnancy; (ii) balancing conflicting priorities between the fetus and the parent; (iii) acknowledging the intimate parent-child bond during pregnancy (one Panelist referred to this as the “mama bear” instinct); (iv) recognizing the cumulative stressors inherent in pregnancy, such as increased medical visits, concern about fetal health, and balancing work and family responsibilities; and, (v) understanding that pregnancy is a critical developmental period for the fetus, influencing parental behavioral choices and decision-making. However, further discussion was tabled by the Panelists as they grappled with the terminology used to define research results with or without clinical significance.

Considerations specific to RBRR with culturally diverse cohorts

When prompted to identify specific ethical considerations for collaborating with culturally diverse cohorts, the Panel agreed that while standard bioethical considerations remain pertinent, there is greater emphasis on RBRR development. It was underscored that involving study participants in the RBRR development process is important, ensuring that research practices are respectful, inclusive, and culturally appropriate. According to a Panel participant,

…whenever possible, it is important to have people who share the background and sensitivity to traditions/culture be involved because they have knowledge and experiences that can be helpful. Sharing drafts of the documents to present results can be helpful in making the document and communication of information meaningful and relevant to the group.

Again, Panelists stated that unclear understandings of what constituted results with clinical significance stymied their ability to further explore RBRR within culturally diverse cohorts.

Additional ethical frameworks

While bioethics serves as the foundational ethical framework in bioethical research, Panelists suggested the following: (i) integrating the principle of utility into bioethics, emphasizing the importance of maximizing overall well-being and utility in decision-making; (ii) recognizing the significance of self-determination by incorporating its elements into bioethical considerations, ensuring that individuals have the autonomy to make decisions aligned with their own values, beliefs, and preferences; (iii) integrating narrative bioethics, which emphasizes the importance of understanding and respecting individuals’ lived experiences and personal narratives in ethical analysis; (iv) including care and relational bioethics principles, which prioritize the ethical significance of caregiving relationships and responsibilities; and (v) incorporating casuistry into bioethical frameworks, which involves the careful examination of specific cases and the application of ethical principles in contextually sensitive ways. By considering these additional perspectives and approaches, the practice of RBRR can more effectively address the diverse challenges from a bioethically informed perspective.

Standardizing language across disciplines and data contexts

Throughout the process, the Panel encountered challenges in using existing terminology to refer to the report-back of research results and potential health implications from exposure research data. In instances where health effects are known, such as in the case of blood lead levels, recommendations often focus on straightforward actions like testing and adopting behaviors to reduce exposure to lead, thus lowering blood lead levels. Yet, RBRR can similarly lead to health promotion behaviors even when exposure-health relationships are not fully quantified. For example, pregnant individuals with greater general knowledge of phthalates, commonly found in plastic, were more likely to avoid personal care products containing these chemicals [47].

When discussing the exposure-health relationship within the context of RBRR, the National Academies of Science used the term clinical significance (National Academies of Sciences and Medicine 2018) [30]. When considering genetic risk, which has a clear clinical significance, RBRR is somewhat straightforward [28]. However, when applied to personal chemical exposure, the Panel experienced difficulty understanding how this term could be appropriately applied. One Panelist succinctly stated, “How do we define clinical significance?”

In discussions, Panelists attempted to distinguish between results governed by established regulatory or health standards (e.g., blood lead levels, radon, or chemicals regulated in drinking water) and those lacking such standards. During these discussions, Panelists offered replacement terms for characterizing the exposure-health association. Subsequently, the Panel participated in a survey in which three distinct research scenarios were presented, with varying levels of direct to indirect health relevance. Panelists were asked to identify each scenario’s most applicable term(s), and could select as many terms as they desired per scenario (Table 2).

Table 2.

Scenarios presented to the expert panel, from the perspective of an academic researcher conducting personal chemical exposure research

Scenario
Scenario 1: Annika is participating in a study to look at her exposure to volatile polycyclic aromatic hydrocarbons (PAHs) and PM2.5 over seven days. In her sampler, researchers find that her average exposure to PM2.5 is 21ug/m3. PM2.5 concentrations in ambient air are regulated by the National Ambient Air Quality Standards, with recommendations that the average over a 24 h period not exceed 12ug/m3. This value is not clinically actionable but can inform behavior changes for Annika to reduce her exposure to PM2.5.
Scenario 2. In Annika’s sampler, the researchers also quantified three PAHs, including one that has been associated with exacerbated asthma (phenanthrene). There are no risk-based values, health guidelines, or regulations for phenanthrene. Substantial research however indicates that phenanthrene is a respiratory inhalant. This data therefore cannot be contextualized with the use of existing standards or regulations and is not clinically actionable. However, Annika may still find benefit in this information.
Scenario 3. Benzo[a]pyrene, a known carcinogen, is also identified in Annika’s sampler. While benzo[a]pyrene is a carcinogen, the risk of developing cancer following exposure is not well quantified at the individual level. Cancer risk assessments rely on population-level exposures. As a result, this value from Annika’s sampler is not clinically actionable, and there are no quantitative risk-based values to contextualize exposure (e.g. are levels too high?). Yet, like with the phenanthrene exposure, Annika may find benefit to this information, particularly when she learns that benzo[a]pyrene is found in cigarette smoke, and she is a smoker.

All scenarios were strongly viewed as having behavioral and preventative utility (selected 42 and 39 times, respectively), followed by the term ‘clinical relevance’ (22 selections) (Table 3). The least chosen terms were Clinical Action Levels (n = 6) and Clinical Significance (n = 11). Selections were equivalent between the remaining terms, ranging from 14 to 19. The Panel considered only the first scenario to have existing health guidelines or standards; this scenario also scored the highest for clinical significance.

Table 3.

Summary of times a term was selected for each scenario. Respondents could select as many terms per scenario as they felt appropriate

Term	Scenario #1	Scenario #2	Scenario #3	Respondents	No response
Clinical Significance	7	1	3	7 (43.8%)	9
Clinical action levels	3	2	1	3 (18.8%)	13
Clinical relevance	8	5	9	12 (75%)	4
Quantitative risk-based values	10	2	2	12 (75%)	4
Existing regulations (either health-based or technology based)	12	0	2	12 (75%)	4
Existing health guidelines or standards	13	1	5	14 (87.5%)	2
Preventative utility	12	12	15	15 (93.8%)	1
Behavioral utility	14	14	14	15 (93.8%)	1

Recommendations and future work

The initial aim of the Expert Panel was to explore the ethical considerations related to how RBRR is conducted in three key contexts: (i) in the absence of clinical significance, (ii) in pregnancy cohorts, and (iii) in culturally diverse cohorts. Early discussions around who should develop RBRR materials, and ways to support RBRR, were well characterized. However, as the Panel delved into these conversations, it quickly became clear that actual discussions were dependent on clear and unambiguous definitions of the term ‘clinical significance.’ In conversation with the Panel, we pivoted, using our final survey opportunity to assess how terms associated with RBRR (clinical significance, behavioral utility, etc.) would be applied to research scenarios.

Table 4 presents a summary of the Panel recommendations. Broadly, the Panel recommended the following: (1) develop centralized RBRR resources; (2) develop RBRR in recognition that participants may use materials in multiple ways; (3) establish that RBRR responsibility lies with the principal investigator of the research, although a committee approach is recommended; (4) conduct RBRR following principles of community engagement; (5) develop RBRR in the context of community-level benefits and harms; (6) clarify that RBRR responsibility does not end when the study ends and; (7) establish standardized language. The Panel also identified two areas of future work: (1) RBRR considerations within the evolving legal landscape, and (2) reconsidering ethical considerations for how RBRR is developed. In addition, the Panel emphasized the importance of recognizing and respecting participants’ cultural norms, values, and preferences in how data is returned and communicated (cultural appropriateness) and maintaining ongoing communication and engagement with participants throughout the RBRR process, including soliciting feedback and addressing concerns (continued engagement). Other ethical frameworks and approaches were also considered. Although the Panel formulated recommendations, it also identified gaps related to these scenarios. Addressing these gaps will facilitate a more comprehensive understanding of the ethical considerations.

Table 4.

Description of expert panel recommendations and future work

Recommendation	Description of recommendation
Develop centralized RBRR resources	Panelists noted that, specific to ECHO, there was a need for centralized resources to help address expertise to specific report-back. For example, resources for environmental RBRR, versus resources for research looking at neurodevelopmental outcomes. In this way, researchers can pull from a diverse variety of resources.
Develop RBRR in recognition that participants may use the materials in multiple ways.	The Expert Panel recognized that often, participants may take their reports to their clinicians, or reports may even recommend participants speak to clinicians about their health concerns. However, clinicians may not be well prepared to discuss chemical-specific health concerns. Therefore, the Panel recommended that RBRR be accompanied by briefs for clinicians, either in the report itself (e.g., talking points for your clinician) or provided directly to clinicians.
Establish that RBRR responsibility lies with the principal investigator of the research, although a committee approach to RBRR development is recommended.	The Panel was in consensus that the responsibility for RBRR lies with the PI, and should not be a centralized program communication within the context of ECHO. As one Panelist stated: “It is the responsibility of researchers to report back on results that may have implications for the participant’s health in ways that explain the science in accessible language, contextualize the risk posed and provide suggestions for actions to take to reduce risk”. While the responsibility was with the researcher, the Panel recommended that a committee approach be used, integrating community engagement specialists to help develop the messaging and appropriate interpretation of results.
Conduct RBRR following principles of community engagement	Given the time lag between study initiation and RBRR, Panelists recommended that researchers think “strategically about how you stay in touch between collection and report back.” They noted that continued communication can help “maintain that trust and create a better understanding of the scientific research process.” Furthermore, engagement with participants in the development of RBRR will ensure the RBRR is culturally competent, uses appropriate examples, and addresses participant needs and concerns, and includes appropriate resources (e.g., resources across an economic and logistic spectrum), following guidance from all four bioethical principles.
Develop RBRR in the context of community-level benefits and harms.	RBRR that explicitly acknowledges potential benefits and harms, as well as clarity around what is known versus not known, has the potential to increase transparency in the research process, and help participants interpret their results.
Clarify that RBRR responsibility does not end when the study ends	The Panel agreed that RBRR would benefit from ongoing evaluation to see how people use RBRR and subsequently adopt mitigation/behavior strategies.
Establish standardized language	Language around report-back of research results has many terms, to include “data disclosure,” “report of results”, and more. Standardized language would help the field move forward. Related, the Panel stressed the need to have terminology that can differentiate between chemicals with current health or regulatory standards or guideposts versus chemicals that currently do not have such standards or guideposts.
Future work. RBRR and the evolving legal landscape.	The Panel indicated that research should continue exploring the intersection of RBRR with the evolving legal landscape, particularly in cases where sensitive research results could potentially be used for non research purposes (e.g., Ferguson v. City of Charleston).
Future work. Reconsider ethical considerations related to how RBRR is developed and disseminated.	Ultimately, the Panel was unable to address all three of the original stated objectives, due to non standardized language. However, the Panel had a brief discussion related to other ethical frameworks, which may complement bioethics, or be used as an alternative. With standardized language, the considerations identified by the Panel could be reconsidered, along with considerations of how to report-back research results from secondary datasets.

An important consideration emerged regarding the ongoing responsibility of RBRR beyond the study duration. Specifically, as researchers contribute their data to public-use repositories, subsequent or secondary analyses might identify new insights beneficial to the initial study participants. This issue emerged as having significant importance because ECHO has a centralized repository, enabling the potential for ECHO study participants to learn of new research findings. As one Panelist stated:

There should be an obligation/agreement with legal teeth [that] if new findings occur, ECHO or the cohort should be informed if they can return those additional results to their population or [if] severe enough return the results themselves.

As the Panel discussed the potential future use of data from a centralized repository, privacy considerations were again raised, as recent research showed that reidentification can occur even in environmental health studies [6].

Discussion

Over the course of three meetings, the Expert Panel identified and discussed ethical considerations and frameworks for returning individual research results to a pregnancy cohort. Early in the process, the Panel experienced difficulty with the terminology being used, and difficulties discerning between RBRR of results with clear health or regulatory standards versus results concerning emerging chemicals of concern without established, quantified relationships to health [26]. Indeed, terms may be somewhat discipline specific. For example, early discussions around the report-back of genetic results used the term ‘disclosure,’ [15] which has also been used in environmental health [19]. Other terms include ‘return of results,’ [13] and ‘report-back,’ [1, 22, 24, 26] and most recently, report-back of research results (National Institutes of Health [31]. While these terms were largely understandable and interchangeable, when trying to quantify the usefulness of research results to health outcomes, the discussion around terminology became far more complex. Strategies for RBRR with known or unknown clinical significance were outlined in the 2018 report from the National Academies of Science (National Academies of Sciences and Medicine 2018). Yet, rather than focusing on quantified exposure-health relationships (e.g., established guidelines for blood lead levels), or known causative relationship (e.g., BRCA and breast cancer), the Panel expressed interest in terms that went beyond clinical endpoints and encompassed the usefulness of data to prevent exposure, or inform behavioral changes to improve health. This understanding of research results more broadly fits the movement towards right-to-know, [27] which is aligned with bioethics principles, [28] and mirrors how participants use their results [2, 35, 40].

As such, the Panel affirmed that all research results benefit participants and should be returned, even in pregnancy cohorts. This stance is further supported by other report-back studies with pregnant people, wherein participants wanted their results, and used their results to make informed decisions about their health and the health of their families [7, 26, 35]. Within this context, the Panel noted that the report-back must be handled carefully, and the cumulative stressors that a pregnant person faces must be recognized. Prior work in this area indicated that while pregnant participants want their information, they are also facing other stressors regarding the types of care products they purchase in an effort to lead a healthy life for themselves and their baby [35].

While the focus was on pregnancy cohorts, the Panel also discussed ethical considerations specific to RBRR with culturally diverse cohorts. Here, the Panel identified a greater emphasis on developing RBRR materials. The importance of including study participants or community liaisons has been previously demonstrated, as liaisons were able to identify research results with cultural or traditional implications that were unknown to the researcher [40, 42].

The study did have several limitations. Although concerted efforts have been made to include diverse perspectives and gather comprehensive data, the scope of the project, e.g., report-back to culturally diverse pregnant populations with an emphasis on research without known health effects, remained limited to this specific context. Different populations and research areas may have distinct ethical considerations that require separate exploration – a consideration the Panelists brought up regularly. We acknowledge several other nuances that may impact how RBRR is conducted, particularly across large consortiums, and regarding RBRR for biobanked specimens. Across large cohorts, such as ECHO, there are nested case-cohort studies, as well as biobanking of specimens. For nested case-cohort studies, discussions will be needed to determine if all results are reported back, necessitating different reports for participants within the nested case, or if only cohort-level results are shared. Considering biobanking, if there is a substantial time lag between data collection and analysis, this may alter the relevance of results, and therefore RBRR, although preliminary data suggests that study participants find value in RBRR, even five years after data collection [39]. Secondly, Panel members took a surprising approach to the prompts provided during the Expert Panel meeting, leading to unexpected interpretations, yet also uncovering necessary discussions around inconsistent RBRR terminology. As a result, full ethical considerations likely remain uncharacterized. Thirdly, survey response rates (~ 60%) fell below expectations. Despite extensive discussions, consensus on the topics proved challenging for the Expert Panel members, given concerns over terminology as previously discussed, or diverse interpretations of research scenarios. Employing the Delphi method in this study was intended to foster consensus among the Expert Panel members. Here, the method rather facilitated large scale discussions, and signaled areas wherein additional work Is needed, primarily around the harmonization of terms used in RBRR. We posit that the absence of unanimous agreement should uphold the importance of diverse viewpoints in informing future work. Lastly, we acknowledge that RBRR can be a time- and cost-intensive practice, which often requires specialized skillsets. In our discussions with the Expert Panelists we asked that they focus on the ethical principles alone, excluding logistical, feasibility, and economic considerations, while recognizing that once ethical guidelines are in place, these guidelines would need to be appropriately tempered and applied to fit research scenarios and logistics.

The anticipated impact of this work extends beyond its implications for returning data to pregnant individuals. The insights gained can potentially inform best practices applicable to various study populations and can be extended to other diverse cultural backgrounds across the ECHO consortium. By evaluating how data should be returned within a pregnancy cohort, we aim to contribute to the establishment of ethical guidelines that promote participant well-being and trust in research endeavors.

Abbreviations

RBRR

Reporting back research results

ECHO

The Environmental influences on Child Health Outcomes

PFAS

Per–and poly–fluoroalkyl substances

Authors’ contributions

EC: formal analysis, investigation, writing – original draft, writing – review and editing. SK: conceptualization, methodology, formal analysis, investigation, writing – review and editing, funding acquisition. TV: formal analysis, investigation, writing – original draft, writing – review and editing, project administration. JH: conceptualization, methodology, formal analysis, investigation, writing – review and editing, project administration, funding acquisition. DR: conceptualization, methodology, formal analysis, investigation, data curation, writing – original draft, writing – review and editing, visualization, supervision, project administration, funding acquisition.

Funding

Research reported in this publication was supported by the Environmental Influences on Child Health Outcomes (ECHO) program, Office of The Director, National Institutes of Health, under Award Number UH OD023290. Additionally, this research was supported by the National Institute of Environmental Health Sciences of the National Institutes of Health under Award Numbers P30 ES030287, R01 ES036256. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.

Data availability

The datasets generated and analyzed during the current study are available from the corresponding author on reasonable request. All quantitative data is included in this published article and the corresponding supplementary information files.

Declarations

Ethics approval and consent to participate

This study was reviewed and determined to be ‘not human subjects research.’ All Panelists were asked to join the Panel, and participation in meetings and surveys was optional. All Panelists reviewed the final manuscript prior to submission.

Consent for publication

Not applicable.

Competing interests

The authors declare no competing interests.