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Partnering with patients in healthcare research: a scoping review of ethical issues, challenges, and recommendations for practice

Abstract

Background

Partnering with patients in healthcare research now benefits from a strong rationale and is encouraged by funding agencies and research institutions. However, this new approach raises ethical issues for patients, researchers, research professionals and administrators. The main objective of this review is to map the literature related to the ethical issues associated with patient partnership in healthcare research, as well as the recommendations to address them. Our global aim is to help researchers, patients, research institutions and research ethics boards reflecting on and dealing with these issues.

Methods

We conducted a scoping review of the ethical issues and recommendations associated with partnering with patients in healthcare research. After our search strategy, 31 peer reviewed articles published between 2007 and 2017 remained and were analyzed.

Results

We have identified 58 first-order ethical issues and challenges associated with patient partnership in research, regrouped in 18 second-order ethical themes. Most of the issues are transversal to all phases and stages of the research process and a lot of them could also apply to patient-partnership in other spheres of health, such as governance, quality improvement, and education. We suggested that ethical issues and challenges of partnered research can be related to four ethical frameworks: 1) Research ethics; 2) Research integrity; 3) Organizational ethics, and 4) Relational ethics.

Conclusions

We have identified numerous ethical issues associated with the recent approach of patient-partnership in research. These issues are more diverse than the issues associated with a more traditional research approach. Indeed, the current discussion on how we address ethical issues in research is anchored in the assumption that patients, as research participants, must be protected from risk. However, doing research with, and not on, the patient involves changes in the way we reflect on the ethical issues associated with this approach to research. We propose to broaden the ethical discussion on partnered research to not only rely on a research ethics framework, but to also frame it within the areas of research integrity, organizational ethics and relational ethics.

Peer Review reports

Background

Patient engagement initiatives in different areas of healthcare have proliferated in the last decade [1,2,3]. Patients are now more commonly integrated in the governance of healthcare institutions, in quality improvement initiatives, education and policy making. In clinical settings, patients are sometimes integrated in multidisciplinary clinical teams, assisting professionals and supporting patients in their journey of care, in rehabilitation, and end-of-life care [3,4,5]. Lastly, patients are now more often engaged in healthcare research initiatives, not as research participants, but as research collaborators, partners or even as co-researchers [5, 6]. However, in these various initiatives, and despite all the good will of the various actors involved, there is still more than often a gap between the ideal conception of patient partnership in meaningful and fruitful initiatives and the actual practices of patient engagement in healthcare. Patient engagement efforts can sometimes be tainted by misconceptions, misunderstandings, and frustrations.

Carman et al. [1] describe patient engagement as “patients, families, their representatives, and health professionals working in active partnership at various levels across the health care system […] to improve health and health care” (p. 224). In the literature, patient engagement usually refers to working and engaging with patients, i.e. individuals with first-hand experience of health issue, or with their caregivers. Carman et al. [1] also describe different levels of patient participation, and propose a continuum of patient engagement in healthcare, ranging from patient consultation, to involvement, to partnership and shared leadership. In the research sphere, Marlett et al. [7] define patient engagement as “collaborative research that is done by, with and for patients to inform health care and health research decisions and questions” (p. 1058). Macaulay et al. [8] describe research partnership as “a mutually respectful relationship based on sharing responsibilities, costs, and benefits” (p. 775). Shippee et al. [6] distinguish between a passive engagement of patient in research, in which patients are research participants, and an active engagement, which corresponds to the patients-as-partner view proposed by Karazivan et al. [2]. In this latter view, patient engagement initiatives aim at engaging patients through an “equitable collaboration with individuals, families and communities affected by a health topic at all stages of the research process, from conception of the study idea through dissemination of results/findings" (p. 314) [9].

Engaging with patients in healthcare research now benefits from a strong ethical rationale, emphasizing that active participation of patients in research has the potential to empower patients, to ensure a more responsible research, and to add value [10], quality, effectiveness, and validity to the research process, among other potential benefits [11,12,13]. However, the ideal or normative conception of active patient engagement or partnership in research is often confronted, in practice, with a more pragmatic view. As mentioned before, full partnership is intended, in theory, as engagement of patients and co-leadership in all stages of research, but the extent to which this practice is really implemented in healthcare research projects can be challenged. Also, authors recognise that effective patient partnership in research requires additional efforts when compared with a more traditional research process [11]. Researchers often feel they are not well equipped and trained to face these issues [14]. Indeed, as interest, pressures and incentives to engage patients in research increase, researchers, patients, members of research ethics boards, research institutions, funding agencies, health organizations managers and professionals encounter more frequently questions and/or ethical dilemmas related to patient engagement in health research. Research with, and not on, the patient involves significant changes in the governance of research projects, in the composition of research teams, in the relationships between team members, as well as in the design, conduct and dissemination of research itself. In many cases, actors are confronted with the various issues that emerge from these changes, without any clear direction and support.

The main objective of this paper is thus to map the literature related to the ethical issues associated with this emergent practice of partnering with patients in healthcare research, and to identify the proposed recommendations, from the literature, to deal with these issues and challenges. We focus here on patient partnership and shared leadership in research, which corresponds to the highest level of patient engagement on Carman et al.’s [1] continuum of patient engagement in healthcare. A secondary objective is to identify the “gaps” in existing literature, i.e. unaddressed issues and missing recommendations requiring future research. Globally, our aim is to help researchers, patients, research institutions and research ethics boards, reflecting on and dealing with these issues. This paper also aims to inform policy making, and to improve the practice of patient partnership in healthcare research.

Methods

To conduct our research, we adopted a scoping review methodology. This methodology has the potential to capture “key concept; gaps in the research and types and sources of evidence to inform practice, policy making and research" (p. 8) [15]. Scoping reviews “can be undertaken as stand-alone projects in their own right, especially where an area is complex or has not been reviewed comprehensively before" (p. 194) [16].

According to Arksey and O’Malley [17], there are four reasons to undertake a scoping review:

  1. 1.

    To examine the extent, range and nature of research activity in a particular area;

  2. 2.

    To determine the value of undertaking a full systematic review;

  3. 3.

    To summarise and disseminate research findings; and

  4. 4.

    To identify research gaps in the existing literature.

Reasons 1, 3 and 4 corresponded to our research objectives.

Our review was conducted following the scoping review framework proposed by Arksey and O’Malley [17]. This framework includes 6 stages: 1) Identifying the research question; 2) Identifying relevant studies; 3) Selecting studies; 4) Charting the data; 5) Collating, summarising, and reporting the results; and 6) Consulting with relevant stakeholders. The six stages are reported hereafter.

Stage 1: identifying the research question

As mentioned, the main objectives of our research project are to map the literature related to the ethical issues associated with patient partnership in healthcare research, and to identify the proposed recommendations to deal with these issues and challenges. Thus, the research question that guided our bibliographic search strategy was “What are the ethical issues associated with patient partnership in healthcare research discussed in the published literature?”

Stage 2: identifying relevant studies

Two reviewers (AM and JTM) conducted the search and review between December 2017 and December 2018. In order to identify the studies relevant to our review, we interrogated three electronic databases: (1) PubMed (medical, biomedical and life sciences literature); (2) CINAHL (nursing, allied health professions, biomedicine and healthcare literature); (3) EMBASE (biomedical and pharmacological literature). The research queries were formulated according to the specific syntax for each database. We interrogated the title and abstract fields using keywords related to four concepts: “patient”, “engagement”, “research”, and “ethics”.

In our preliminary analysis of the literature, we found out that in some cases the term “community partnership” and “patient partnership” were used interchangeably, and that the term “community” was sometimes used to designate stakeholders that are not usually part of traditional health research teams, including patients, caregivers or user committee members. We thus decided to included “community” as a search term, along with “patient”, but made sure to only include in our study selection the studies addressing patient partnership in research (see Stage 3 of our methodology).

The search terms used are summarized in Table 1. Research results were saved using the bibliographic management software EndNote.

Table 1 Search terms

Stage 3: selecting studies

Our research included peer reviewed studies published in English from 2007 to 2017. We initially conducted the search in English and in French (French is the first language of the three authors of the paper), however, we did not identify any article matching our inclusion criteria in the French literature, so we focused only on the English results. We focused on this 10-year window, since our goal was to identify current ethical issues related to the emergent practice of patients being engaged as partners in health research initiatives. Patient engagement in research is indeed a recent approach in healthcare research, which has become more prominent over the past 10 years, notably since research funding agencies around the world have started to support this approach.Footnote 1

AM analyzed the title and abstract of each identified article (n = 187) to determine if it addressed the ethical issues associated with patient partnership in research. After removing duplicates (n = 45), errata (n = 1), commentaries (n = 10), clinical studies (n = 52), editorial (n = 4), thesis (n = 1), poster abstracts (n = 4), erroneously identified references (n = 1), book chapter (n = 1), as well as one article with no accessible full text (n = 1), AM analyzed the full text of all remaining studies (n = 67) to exclude those that did not address partnership with patients in research (n = 38), such as in the case of patients engaged as research participants, or in the case of patients only engaged as advisors, which does not qualify as patient partnership, according to Carman et al. [1] continuum of patient engagement in healthcare and the patient-as-partner view presented in the background section [2, 5, 6]. We also added references identified from hand search (n = 2). At the end of our search strategy, 31 articles remained, which consisted in our final study sample (final n = 31). A flow chart of the search results is presented as Fig. 2 in the “Results” section.

Stage 4: charting the data

After having identified all relevant articles to our review, we collated bibliometric data to qualify our study sample, as suggested by Peters et al. [18]. This data is presented in Table 2, in the “Results” section.

Table 2 Description of included studies

We proceeded with the full text analysis of each article in our study sample. AM conducted the initial review, and JTM randomly selected 15 references for a second independent review in order to verify intercoder agreement. There was substantial agreement between both reviewers on the identification of the issues in the reviewed articles, and the few disagreements were resolved during one research meeting between AM and JTM.

All issues, barriers and challenges associated with partnering with patients in every steps of the research process were extracted from the selected articles, as well as their associated recommendations for practice, when addressed in the sampled articles. These first-order issues and recommendations correspond to the initial set of codes in our coding process. In line with our scoping review approach, we decided at this point to include all issues, difficulties, barriers and challenges mentioned in our sampled articles, using a broad understanding of what can be considered as an ethical issue. For example, we encountered issues that were deemed at first more practical, such as the absence of compensation offered to patient-partners, but we could also see the underlying ethical aspect to these issues, such as a search for justice and equity between research team members in the case of compensating patient-partners. We thus decided on including all pertinent issues and challenges identified in our sampled articles.

To organize our results, we used Shippee et al. [6] framework of phases and stages of patient engagement in research presented in Fig. 1. As patient partnership aims to engage with patients in all steps of the research process, we figured issues and recommendations about this practice should also be addressed for every research step.

Fig. 1
figure1

Framework of phases and stages of patient engagement in research, adapted from Shippee et al. 2015, p. 1157

Stage 5: collating, summarising, and reporting the results

During our review, it was first found that several issues were transversal to all research phases and stages of research and were identified as such by the authors of our reviewed papers. We thus decided to create a separate result section for those transversal issues (see Table 6 in the Results section).

To synthetize our results in a conceptually pertinent way, we regrouped the first-order issues (our initial codes) in second-order themes of ethical issues (see first column of Tables 345 and 6), through thematic analysis [45]. The predominant themes were elaborated using an inductive and iterative process through multiple meetings between research team members (JTM, AM and AB). During this iterative process, all disagreements between the reviewers concerning the classification of the first-order issues into the second-order themes were resolved. In these meetings, we worked on collating and summarizing all identified first-order issues in these second-order themes, organized in the three phases and eight stages framework of patient engagement in research proposed by Shippee et al. [6]. The labels of the second-order themes of issues were also determined collectively and inductively. Results are discussed and presented in summary tables in the “Results” section.

Table 3 Issues associated with partnering with patients in the Preparatory phase of research
Table 4 Issues associated with partnering with patients in the Execution phase of research
Table 5 Issues associated with partnering with patients in the Translational phase of research
Table 6 Transversal issues associated with partnering with patients in healthcare research

We also used a data extraction chart to visually map the literature, identify recurring issues and recommendations, as well as to help us identify gaps and unaddressed challenges and issues. In the chart, we indicated the presence of all themes of issues in all sampled articles, regrouped in stages of research. Note that the presence of a theme of issue, as indicated by a “x” in the chart, does not qualify the information about this issue, but only the presence of the issue in the article reviewed, might it be discussed in depth, or only in passage. The extraction chart is provided in Additional file 1.

Stage 6: consultation exercise

Although the consultation stage is optional in Arksey and O’Malley’s [17] framework, we did integrate a consultation exercise with a committee composed of stakeholders and practitioners with expertise on patient partnership in research. According to Arksey & O’Malley [17], the consultation exercise is an “additional, parallel element” of the scoping study framework that can “inform and validate findings from the main scoping review” (p. 9). Since the overarching aim of this research was to inform policy making about the ethical issues associated with the practice of patient partnership in research and to make recommendations to researchers, patients, research institutions, etc. to improve its practice, we assumed that working with stakeholders would increase the relevancy of results, help identify unaddressed ethical issues, and facilitate knowledge transfer.

Our stakeholder committee was composed of two researchers experienced in conducting research with patients, two patient-partners in research, two representatives from distinct research ethics boards from healthcare research institutions in Quebec, Canada, an ethicist from the Fonds de recherche du Québec (FRQ), the main public funding research agency in the province, and an ethics expert from the Canadian Institutes of Health Research (CIHR).

A first meeting was organized prior to the reviewing process and aimed at presenting the objectives of the research and to have a better idea of the needs of each stakeholder in the matter of knowledge transfer following our study. A second consultation was organized after the literature review was completed. For that, we sent all members of the stakeholders committee the results of our review by email, and asked them to comment it and identify gaps, i.e. unaddressed issues and challenges, based on their own experience. AM and JTM collected and then proceeded to code all first-order issues, recommendations and comments from the written answers received. We then collated the results in a table, eliminating redundancies and unoriginal issues, i.e. those that were already covered in the scoping review. We synthetized all the new first-order issues and recommendations conveyed by the stakeholder committee, as well as the comments that we found pertinent and enlightening, i.e. those giving a different perspective or adding an important precision to the issues already identified in the review. The results of this consultation are presented in Table 7, in the “Results” section.

Table 7 Results from the stakeholders’ consultation exercise

Results

A flow chart of the search strategy and search results is presented in Fig. 2. Following, we present a description of our sampled articles, in Table 2.

Fig. 2
figure2

Flow chart of search strategy and search results

Interestingly, only few papers in our sampled articles reflected the patient’s perspective on the ethical issues associated with patient partnership in research (for example De Wit et al., 2013), which means that the following results are thus principally the view of researchers on the topic.

We hereafter present the results of our review of the ethical issues and challenges associated with patient partnership in research identified in our sample articles, as well as associated recommendations, for all steps of the research process. We also added a table presenting issues that are transversal to all phases and stages of research. Findings are presented in complete tables, in which first-order ethical issues (identified with a black dot) are regrouped in second-order themes of ethical issues, following our thematic analysis.

In the following Fig. 3, we present a synthesis of the second-order themes of ethical issues identified in our literature review. We have listed all the second-order themes of ethical issues presented in our result tables together, independently of the different phases and steps of research, and regrouped similar themes, to obtain a final list of themes of ethical issues, as well as the importance given to each of them in the reviewed literature, measured in occurrences. More precisely, the theme “lack of resources to support partnership” was associated with the preparatory phase of research (Table 3) but was also mentioned by authors as a transversal issue (Table 6). In Fig. 3, we regrouped both themes, and removed redundancies in order to only count one occurrence per reviewed article. We did the same regrouping for the theme “tokenism” (from Table 3 and Table 6); the theme “conflict of interest” (from Table 3 and Table 6); the theme “patients’ exclusion from research stage” (from Table 4 and Table 5); and the theme “disagreement on priorities” (from Table 3 and Table 5).

Fig. 3
figure3

Themes of ethical issues associated with partnering with patients in healthcare research –Occurrences in the literature

Unaddressed issues and recommendations

Following our consultation with the stakeholders committee, we were able to identify the followings gaps in the literature, i.e. the missing ethical issues and/or the lack of associated recommendations for an ethical partnership with patients in healthcare research. These also include the issues that have been identified and presented in the result tables, but that haven’t been the object of much research or discussion in the literature we scoped. We collated the new first-order issues, new recommendations and the comments obtained in this consultation exercise in the Table 7.

The 4 new issues highlighted by the stakeholder committee were coherent with the 18 second-order themes of issues presented in Fig. 3. Indeed, the issue of the lack of engagement due to prior negative experiences of engagement researchers may have had or heard from is encompassed by the second-order theme “absence of a shared vision of patient partnership”. The two new issues about the influence of patient engagement in the early phase of research on the research agenda and types of research undertaken are coherent with the second-order theme “disagreement on priorities”. Finally, the last new issue about patient-partnership impacting team efficiency is encompassed by the second-order theme “logistical and practical barriers to research partnership”.

Discussion

We have identified a total of 58 first-order ethical issues and challenges associated with partnering with patients in healthcare research from the main scoping review (see Tables 345 and 6). Some issues were specific to the Preparation phase of research (n = 11), the Execution phase of research (n = 6) and the Translational phase of research (n = 6). However, the majority of the issues identified in our scoping review were classified as transversal issues (n = 35), pertaining to all steps and phases of research or to the general practice of engaging with patients as partners in different healthcare initiatives. We believe most of these transversal issues, such as the absence of compensation for patients, and the lack of training on patient-partnership for team members, to be also common to other spheres of patient-partnership initiatives, such as engaging with patients in governance committees, in quality improvement initiatives, or in healthcare education. Indeed, it appears that a lot of these issues are not specific to partnered research, but rather to the establishment of a sincere, satisfying, and functional partnership between patients, healthcare professionals and administrators.

We regrouped the first-order issues in 18 second-order themes of ethical issues. As presented in Fig. 3, the most prominent second-order themes of issues associated with partnered research found in the literature, i.e. those that presented the most occurrences in our sampled articles, were: a) the lack of resources available to carry research in partnership with patients; b) the challenges in the selection of patient-partners; c) the lack of support offered to research teams and patients; d) the absence of a shared vision on research partnership with patients; e) a tokenistic patient engagement in research; and f) the logistical and practical barriers to partnered research.

The results from the consultation exercise added interesting precisions and new perspectives on the issues and recommendations already scoped in the literature review, and also added 4 new first-order issues to the list (see Table 7). We were particularly interested in the identified unaddressed issues relating to patient engagement in the early phase of research, notably in the research agenda setting. It was brought forward by the stakeholder committee that patient engagement in the beginning of a research project may favor or overemphasis certain themes of research, on more common pathologies such as cancer or diabetes, and could also favor certain types of research, which would be more focused on getting results that can be quickly transferred to care, as opposed to fundamental research. These important issues are definitely to be explored further.

Ethical frameworks for patient partnership in research

When analyzing the results of our review, it appeared to us that numerous issues and challenges associated with patient partnership in healthcare research related to ethical frameworks that expanded way beyond the traditional research ethics framework that is generally used to reflect, anticipate, prevent and deal with the ethical issues associated with a more traditional research approach. Indeed, the practice of partnering with patient in research, i.e. of doing research with, and not on, the patient involves changes in the way we reflect on the ethical issues associated with this research approach. In a more traditional research approach, patients are usually involved only as research participants. The ethical issues surrounding the relationship between the research and the patients in this case are prevented and dealt with research ethics principles and processes, often managed through the process of obtaining an ethics certificate from a research ethics board. The general goal is to protect research participants from harm. But when we switch to doing research in partnership with patients, the issues are more diverse. Of course research participants are still to be protected under the principles and processes of research ethics, but there are added goals aimed in the ethical reflection on the issues engendered by partnered research, such as to protect the partnering relationship, to assure the best contribution of all actors, as well as to protect patient partners in research. Research ethics principles and processes focused specifically on protecting research participants are thus in most cases inapplicable or unrelated to the diverse and numerous issues specifically related to partnered research we have identified in our scoping literature review. This broadening of the conceptualization of ethics in the context of partnered research appeared to us as an important contribution of our review, and we thus attempted at a different categorization of the first-order ethical issues according to the areas of ethics they related to, and based on the definitions we propose hereafter. This conceptualization was not another step into the thematic analysis, aiming at reducing again the 18 second-order themes into a potential third-order classification. Differently, we wanted to present frameworks that could help researchers, patients and practitioners reflect on the variety of issues associated with the recent practice of patient partnership in research found in the literature, and to show that the traditional framework used in research was in most cases unhelpful or inapplicable. This different categorization was developed inductively and qualitatively, during meetings of the research team (JTM, AM and AB).

We have found that the 58 first-order ethical issues and challenges associated with patient partnership in research identified in our literature review, as well as the 4 issues brought forward by the consultation exercise, can be related to four ethical frameworks that we have identified as: 1) Research ethics; 2) Research integrity; 3) Organizational ethics; and 4) Relational ethics. These are illustrated in Fig. 4.

Fig. 4
figure4

Ethical frameworks for patient partnership in research

As mentioned above, research ethics is interested in the ethical issues raised by involving patients as research participants, and focuses on respect for participants, autonomy of patients, concern for welfare, protection of participants from risks, justice and issues associated with vulnerability. Principles of research ethics are included in national and institutional guidelines and are implemented by institutional research ethics boards (REBs) and researchers. In our scoping review, we have identified only 5 issues (n = 5) associated with research ethics. For example, some researchers are worried partnering with patient in research might compromise data confidentiality [25, 30], or may expose patient partners to incidental findings about patient participants [25, 31].

Research integrity is concerned with the way scientific research is carried out, and is focused on scientific rigour, honesty, reliability, referencing and plagiarism, working out authorship, conflicts of interest management, grant application and fund management, for example. Research integrity is the responsibility of researchers and research professionals, as well as research institutions, and guided by national and institutional policies and guidelines. In our review, we have identified 15 issues and challenges to partnered research associated with research integrity. For example, some researchers worry that the lack of patients’ knowledge about research methodologies may raise concerns about the integrity of scientific methods [28], or that patients’ potential conflicts of interest may influence their contribution in the research process [25, 33]. Research ethics and research integrity are now often regrouped under the umbrella of the concept of responsible conduct of research (RCR). Added together, they account for roughly a third (n = 20) of the issues identified in our review.

Differently, organizational ethics focus on the practices, programs, structures, reward allocation, and leadership needed to encourage the ethical and responsible conduct of employees and managers in organizations, and to foster ethical and responsible relationships with stakeholders. It is interested in ethical decision-making and behavior from organizational actors, and the management of misconducts. It is carried out in organizational mission, values and culture, embodied in codes of ethics and value statements. In the context of patient partnerships in healthcare organizations, it is not always clear how patient partnership initiatives are supported. Some health organization have dedicated structures responsible for supporting patient engagement, but these structures may fall under different organizational functions and hierarchical levels, and often lack resources, legitimacy, or leadership. Half of the issues and challenges identified in our review (n = 30) can be associated with a broad view of organizational ethics, most of them being practical, logistical and processual issues. These include, for example, the lack of support offered to researchers and patients to carry out partnered research, the lack of resources available to foster meaningful partnership, the disparity in patients’ access to research partnership, and the question of the compensation of patient-partners.

Relational ethics focuses on the relationships and the context of the connections between individuals, and highlight values and concepts such as mutual respect, engagement, environment and embodiment [46, 47]. This view is mostly developed in nursing literature [48], but we found it to be quite suited and useful to reflect upon the issues relating to the ethics and the quality of the relationship fostered with patient-partners, within research teams and research institutions, and on the quality and the sincerity of the engagement of patients in the research process. We have identified several issues linked with this concern in our review (n = 12), mostly relating to an unmeaningful or insincere partnership being established with patients in the context of research, or to the absence of a shared vision of patient-partnership between members of the research team. For example, it has been discussed that some researchers mistrust the value of patients’ contribution [28], or find that patients aren’t knowledgeable enough and lack the expertise to contribute to research [21, 28], and that patient engagement in research can be tokenistic [5, 28, 38, 43]. For these reasons, patients are often excluded from certain phases of research. Although our review was focused on patient engagement in research, for the most part the issues relating to relational ethics might also be present in other spheres of patient engagement, such as in governance and/or teaching activities. Developing a shared vision and meaningful and ethical relationships with patient-partners is the responsibility of researchers, research professionals, and administrators, but patients also influence these relationships and share expectations.

As mentioned before, the issues mentioned in our review fall under the responsibility of different and often unrelated actors. But most importantly, there is often a lack of coordination between the actors and organizational structures responsible for research ethics, research integrity, organizational ethics and fostering qualitative and ethical relationships with patients in the context of patient partnership initiatives. Indeed, these responsibilities fall under different departments, committees, managers and professionals. For example, organizational ethics is not much developed in Quebec healthcare organizations. Issues linked to organizational ethics are often carried through by clinical ethicists who are sometimes unaware of patient engagement initiatives, especially the ones taking place in the context of research, and not coordinated with research ethics board, or patient engagement support staff. Unfortunately, this lack of coordination creates silo effects, and can exacerbate the ethical challenges research professionals and patient-partners may encounter. The lack of a more coordinated, systemic and strategic management of patient engagement initiatives, including partnered research, in healthcare organizations can prevent the ethical development of this recent research approach. In this context, we anticipate that the traditional culture of research, conducted without engaging patients, may find back its old rights. If policy makers, administrators, health professionals and patients believe patient partnership in research is an approach worth pursuing, it appears that changes will have to happen in its management by health organizations and institutions in order to ensure patient engagement and partnership initiatives in research are carried through in the most ethical way.

Limitations and future research

This review was limited by a small number of studies that specifically addressed the ethical issues and challenges of partnering with patients in healthcare research. Given the growing interest in this approach and the institutional pressures to engage with patients in research, we believe more empirical research will be needed on the ethical issues raised by this research approach.

Some aspects of the search strategy chosen to conduct our study might also have brought some limitations. First, we chose to focus on the higher level of patient engagement in research brought forward in the literature and in practice, which is the level of patient partnership in research. This focus led us to exclude articles that did address the topic of patient engagement in research, but not to the level of patients being engaged as partners in research. We excluded 38 articles on that criteria, such as papers focusing on patients being engaged as advisors in research or being consulted in research projects. It is possible that some other potentially interesting ethical issues associated with the general practice of patient engagement in research may have been found in these excluded papers. Also, the focus on the 2007–2017 window may potentially have excluded some early papers addressing the ethical issues of patient partnership in research, published before that timeline. However, our preliminary search lead us to specifically focus on that timeline, since we hadn’t found papers corresponding to the search criterion before that. Also, we chose in our review to focus on peer-reviewed articles, but this choice came with some limitations, as it made us exclude other types of articles, such as commentaries, which often add insights into ethical issues associated with a research project. Moreover, our results might be specific to certain national context, namely partnered research in the US, Canada and the UK, since 77% of our sampled articles were authored by scholars from these countries. Also, our review reflected both patients and researchers view on patient partnership initiatives in research in the available literature, however, only few papers from our sampled articles highlighted the view of the patients’ side of the partnership, as mentioned in the Results section. We thus believe there is a need for more research to better grasp the opinions and views of patient-partners on the ethical issues and challenges of partnered research.

We also wish to acknowledge a bias towards the positive value of patient engagement in research. Both JTM and AB are currently engaged in research initiatives conducted in partnership with patients, and are convinced of the importance of this approach, although sometimes critical of the way it is carried through in practice.

Finally, one of the contributions of this scoping review was to offer a conceptual framework of the categories of ethical issues that could inform future empirical research. The proposed framework, as well as other findings from this review, such as the gaps identified in the literature, should be further explored to ensure a more comprehensive and in depth understanding of the ethical issues related to patient engagement in healthcare research. This could be done through interviews and/or surveys methods, conducted with researchers and patient-partners themselves, for example. Further exploration should lead to more specific recommendations for policy making.

Conclusion

We have identified 58 first-order ethical issues and challenges associated with the patient partnership in research approach in our scoping review of the literature. These were regrouped in 18 second-order themes of ethical issues. In our consultation exercise with a committee of stakeholders, we have identified another 4 first-order issues that complemented the findings from our main scoping review. The results of our review show that most issues are transversal to all phases and stages of research, and that many of these issues could also apply to the practice of patient engagement or patient partnership in other spheres of healthcare such as governance, quality improvement, and education. Our results also show that the ethical issues associated with partnered research are more diverse than the issues usually associated with a more traditional approach to research, conducted without engaging patients. The usual discussion of how we address ethical issues in research is based on the assumption that patients, as research participants, must be protected from harm. However, doing research with, and not on, patients involves changes in the way we reflect on the ethical issues associated with this research approach. We thus propose to broaden the ethical discussion on partnered research to not only rely on a research ethics framework, but to also frame it within the frameworks of research integrity, organizational ethics and relational ethics.

Availability of data and materials

All data generated or analyzed during this study are included in this published article.

Notes

  1. 1.

    For example, in the US, the PCORI Institute was founded in 2010, and the National Center for Advancing Translational Science from the NIH was founded in 2012, and they both supports since 2012 research initiatives on patient and community engagement. The Strategy for Patient-Oriented Research (SPOR) from the Canadian Institutes on Health Research was created in 2011, and since then it supports, and funds partnered healthcare research in Canada.

References

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Acknowledgements

The authors are grateful to the steering committee of the research project, as well as the stakeholder committee who participated in this review.

Funding

This research was funded by SPOR Support Unit of Québec (Unité de Soutien SRAP du Québec), an initiative from the Canadian Institutes of Health Research (CIHR). The funding agency had no input regarding the design of the study; the collection, analysis, or interpretation of data; nor in writing the manuscript. J. T. Martineau also wish to acknowledge funding from HEC Montréal to support open access publication.

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JTM (principal investigator) conceived of the study and the paper. AM conducted the review, with JTM as second coder. JTM, AM and AB participated in analyzing the data. JTM drafted the paper, and AM and AB contributed and commented on different versions of the manuscript. All authors read and approved the final manuscript.

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Correspondence to Joé T. Martineau.

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Martineau, J.T., Minyaoui, A. & Boivin, A. Partnering with patients in healthcare research: a scoping review of ethical issues, challenges, and recommendations for practice. BMC Med Ethics 21, 34 (2020). https://doi.org/10.1186/s12910-020-0460-0

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Keywords

  • Scoping review
  • Ethical issues
  • Patient partnership
  • Patient engagement
  • Healthcare research