Abstract
Background and objectives Although patient and caregiver involvement in research is widely advocated to improve the relevance and uptake of study findings, barriers and uncertainties in achieving this goal remain. This study aimed to describe patient and caregiver experiences and perspectives of their previous involvement in research, to inform strategies to strengthen patient involvement in research.
Design, setting, participants, & measurements Semistructured interviews were conducted with 23 adult patients with CKD and caregivers who had previously been involved in research, from Australia, the United States, the United Kingdom, and Denmark. Transcripts were analyzed thematically.
Results We identified six themes: grappling with CKD diagnosis (overwhelmed by the burden of illness, silenced by stigma and shame, absence of advocacy, and awareness), bearing the responsibility for involvement (autonomy in their own care, forced to be proactive to access opportunities, infrastructural support to connect researchers and patients), battling big agendas (struggling in a system of disincentive, changing research culture, becoming equals), seeing the person behind the patient (harnessing broader knowledge, expertise, skills and interests, understanding patient needs, motivations for involvement), sensitivity to complexities of payment (accounting for individual circumstances, denoting value, enabling diverse involvement), and championing the patient voice (links to important stakeholders, drivers of innovation, responsibility to end users).
Conclusions The burden of CKD, limited opportunities, and power asymmetry between patients/caregivers and researchers were challenges to meaningful involvement in research. Building trust, awareness of opportunities, and recognizing the broad expertise and value of patients/caregivers, including and beyond their illness experience, may better support patient and caregiver involvement in research in CKD.
- consumer involvement
- consumer engagement
- patient involvement
- patient engagement
- patient partnership
- chronic kidney disease
Introduction
Patient involvement in research, defined as “research being carried out ‘with’ or ‘by’ members of the public rather than ‘to,’ ‘about,’ or ‘for’ them” (1), is now widely advocated by funders, regulators, patient and professional organizations, and journals to align research with the priorities and needs of patients/caregivers and researchers (1⇓⇓⇓⇓⇓–7). Patient involvement in study goals, design, and conduct can improve recruitment and retention and the reliability and uptake of study findings (8⇓⇓–11). Although there are increasing efforts to involve patients and caregivers in research in CKD, only 20% of clinical research addresses the top 10 patient priorities (12). Barriers and challenges include the need for resources to provide logistic and financial support and adequate and appropriate training for both patients/caregivers and researchers (9,10,13,14).
Patients with CKD depend on time-consuming and invasive treatments with inflexible schedules and contend with many symptoms, complications, and unpredictable events that can impede capacity for involvement (15,16). Caregivers also have considerable responsibilities that may limit their ability to be involved in research. These burdens amplify the difficulties faced in identifying and navigating research systems.
There is limited evidence for the perspectives of patients and caregivers with CKD on being involved in research (15,16; T. Gutman et al., unpublished observations). This study aims to describe patients’ and caregivers’ experiences and attitudes to being involved in research in CKD across the research cycle (1) (including priority setting, designing research, collecting and analyzing data, disseminating and implementing study findings, and evaluating studies) to strengthen and inform best practice approaches for involvement in research in CKD.
Materials and Methods
We used the Consolidated Criteria for Reporting Qualitative Studies to report this study (17).
Participant Selection
People with lived experience of CKD (including patients and caregivers), who have been involved in at least one stage of the research process (1) (e.g., priority setting, study design) in any role (e.g., Steering Committee member, investigator), were English speaking, and aged ≥18 years were eligible to participate. Participants were known to the investigators and identified through patient and professional networks and the Standardised Outcomes in Nephrology (International Network) (Supplemental Item 1). Participants were purposively selected on the basis of their experience and involvement in research (e.g., elected position, paid role, volunteer), and to capture a diverse range of demographic (i.e., age, sex, country, profession) and clinical characteristics (i.e., CKD stage, diagnosis). T.G. or A.K. invited participants by email and explained the aims of the research and their interest in the topic. Informed consent was obtained from all participants. Ethics approval was granted by the University of Sydney.
Data Collection
T.G. and A.K. (females) conducted single semistructured interviews with participants only, face to face or through videoconference (Zoom Video Communications) from September 2017 to March 2021. The interview guide was developed on the basis of a review of the literature (10,13,14,18) and discussion among the research team. All interviews were audio recorded and transcribed verbatim, and researchers took field notes. We conducted interviews until we reached thematic data saturation.
Analysis
T.G. read the transcripts, inductively identified preliminary concepts, and grouped them into themes using thematic analysis (19). Themes were revised on the basis of discussion with A.T., A.K., and N.S.R. to ensure the coding structure captured the full range and breadth of the data (20). The transcripts were coded line by line to the revised themes using HyperRESEARCH software. We conducted member checking by emailing the preliminary results to participants for feedback, and additional concepts were integrated into the final analysis (21).
Results
We contacted 32 patients or caregivers and 23 were interviewed (nine did not respond or were not available). Of the 23 participants, 18 (78%) were patients and five (22%) were caregivers from Australia (n=10), the United States (n=10), the United Kingdom (n=2), and Denmark (n=1). The participant characteristics are shown in Table 1. Duration of interviews ranged from 35 to 85 minutes. Participants most frequently reported being involved in priority setting (87%), reviewing documents (78%), and recruiting participants for studies (61%). The participants were known to the investigators to have had experience in involvement in research.
Characteristics of patients with CKD and caregivers who had previously been involved in research, n=23
We identified six themes: grappling with the CKD diagnosis, bearing the responsibility for involvement, battling big agendas, seeing the person behind the patient, sensitivity to complexities of payment, and championing the patient voice. The next section describes the subthemes. Selected quotations for each theme are available in Table 2. Figure 1 shows the relationships between themes and subthemes. The themes were similar across demographics unless otherwise specified.
Illustrative quotations
Thematic schema. Individual factors: changed from individual to individual depending on their circumstances. Collective factors: shared by participants through being identified as a patient with CKD, part of the community of patients with CKD. Systemic factors: relied on broader infrastructure, and research culture and community. Participants faced challenges in being involved in research as individuals, struggling to cope with their identity as patient and partner. Collectively, they encountered stigma and shame around their CKD diagnosis and were forced to search for opportunities for involvement. They felt systemically discouraged and disempowered because they lacked support and resources needed to tackle health systems and research culture historically set up to silence the patient and recognize the clinician/researcher as the expert. However, they identified opportunities to improve involvement by addressing individual needs and circumstances, leveraging collective motivations to advocate the patient voice, and becoming recognized for their ability to contribute to research beyond their lived experience of CKD.
Grappling with CKD Diagnosis
Overwhelmed by the Burden of Illness.
Living with CKD was “all encompassing.” Time-consuming treatments (e.g., dialysis), feeling “wiped out,” and wanting to lead a “normal life” were barriers participants identified to being involved in research. Furthermore, some found it difficult to share their experiences because they were “cognitively challenged,” “fragile emotionally,” and had lost “confidence” in themselves, and felt pressure to downplay symptoms such as pain, depression, and anxiety. Caregivers felt “wired” into their roles and found it difficult to take on responsibilities over and above keeping the health of their loved one stable.
Silenced by Stigma and Shame.
Participants noted the stigma related to identifying as a patient and were thus hesitant to share their experiences due to the shame and blame from others—“oh, you did drugs and you used alcohol.” Misunderstandings that kidney failure was self-inflicted or communicable, and blame from dialysis health care professionals around adherence and fluid overload, caused patients to “shut down,” preventing further involvement in research.
Absence of Advocacy and Awareness.
Participants explained that “kidney disease isn’t sexy” and were frustrated by the lack of public awareness compared with other diseases (e.g., cancer and HIV). Some suggested that public advocacy by well-known individuals, such as Selena Gomez, to “get the word out” would encourage more involvement in research initiatives.
Bearing the Responsibility for Involvement
Autonomy in Their Own Care.
Participants viewed involvement in their own care and advocating for themselves in clinical settings as a critical first step to being involved in research. This allowed them to develop trusting relationships with their clinicians, opening opportunities for research involvement. However, participants from the United States, believed health systems pushed patients to “check [their] independence at the door,” discouraging further engagement and leading to an “inactivated population.”
Forced to Be Proactive to Access Opportunities.
Participants were forced to seek out opportunities to be involved in research (e.g., through social media, websites) and struggled to understand jargon and acronyms. Some “internally driven” participants contacted researchers but noted “not everyone can be as proactive.” Some gave up looking for opportunities because they repeatedly never heard back from researchers.
Infrastructural Support to Connect Researchers and Patients.
Participants were frustrated at researchers “having to recruit again from scratch” for each project and failing to “re-engage the people who are already interested.” Participants suggested researchers develop databases to disseminate opportunities or match researchers and patients/caregivers on the basis of interest and experience.
Battling Big Agendas
Struggling in a System of Disincentive.
Participants, particularly in the United States, explained “the system is designed to be paternal. It pacifies the patient.” They noted large dialysis companies and hospital systems prioritized “profit” over “health,” placing pressure on doctors to stick to strict schedules and quotas, leaving no time to educate patients. Furthermore, some surmised that dialysis companies put up “roadblocks” to stop researchers from accessing their patients.
Changing Research Culture.
Participants wanted researchers/clinicians to acknowledge the paternalistic attitude to involvement. They noted some clinicians/researchers were “protective of their power” and “they don’t want us playing in their sandbox.” They observed this was “generational,” and younger researchers were recognizing patients/caregivers as valuable contributors. Social media aided a shift in power, allowing patients/caregivers to start “a dialogue instead of a one-sided monologue.” Government, professional organizations (e.g., American Society of Nephrology) and journals (e.g., British Medical Journal) were praised for efforts to involve patients/caregivers.
Becoming Equals.
Some participants felt valued and integrated in collaborative research teams, whereas others described a chasm between “us (patients/caregivers) and them (clinicians/researchers)” and experienced “a real pushback against treating the patient as an equitable partner.” Some initially struggled with their role because they viewed themselves “on a much lower level” than other members of the research team (i.e., clinicians). With encouragement from the researchers, they developed the confidence to “speak up.”
Seeing the Person behind the Patient
Harnessing Broader Knowledge, Expertise, Skills, and Interests.
Patients/caregivers had more to offer than lived experience and felt researchers failed to “harness” their expertise in other disciplines. They wanted opportunities to contribute to research that was of interest to them and where they could apply their skills. For example, when designing an infographic: “What if you went out to your consumers and said, ‘is anyone a graphic designer?’”
Understanding Patient Needs.
Participants wanted researchers to be proactive in ensuring they were able to speak freely and to understand discussions and communications (e.g., through a dedicated patient liaison). They emphasized the need for plain language and detailed information about the research project, including the tasks and time required of them. They appreciated updates on unexpected delays and being informed of the outcomes of the research.
Motivations for Involvement.
Participants were driven to become involved in research to “pay it forward” or “give back to the community.” Involvement empowered them to become “proactive” and turn their experiences with kidney disease into “something positive.” Others found it “rewarding” to share new knowledge with other patients. Participants enjoyed traveling, meeting other patients, and viewed involvement as an opportunity for professional development. They conceptualized their involvement in research as improving patient experiences and outcomes.
Sensitivity to Complexities of Payment
Accounting for Individual Circumstances.
Participants emphasized the need to consider each person’s individual needs and preferences regarding reimbursement and payment for involvement in research. Some were frustrated with not being financially compensated for their work, “It’s the hardest job I never got paid for,” whereas others were content to contribute without payment because they could “afford to.” Some preferred no payment because it could compromise their impartiality, particularly if the money came from a pharmaceutical company. Some cautioned that receiving payment could cause them to lose government benefits, such as for disability, but agreed for their expenses to be reimbursed.
Denoting Value.
For some, payment demonstrated that researchers valued their contributions as “equal to the other people participating” and made them feel “part of the team.” Some noted they were asked “to do an awful lot for free” and thought this was “disrespectful.”
Enabling Diverse Involvement.
Participants were concerned involvement was “heavily dependent on a few highly motivated individuals, who give up a lot of their own personal time and personal cost to get involved.” Providing payment for time and covering expenses in advance were considered enablers to involve a broader demographic. Budgeting for patient/caregiver involvement was seen to avoid “the tendency to go for the person that’s geographically closer” and allow for purposive involvement.
Championing the Patient Voice
Links to Important Stakeholders.
Participants viewed themselves as advocates for research: “connection is key.” They used their influence within patient and professional organizations to “vouch for researchers,” sway policy makers, lobby government, and disseminate findings broadly. They engaged, trained, and empowered new patient/caregiver partners through networking.
Drivers of Innovation.
Unique firsthand insights from participants allowed them to contribute compelling and creative suggestions for researchers (e.g., developing a device to detect fluid volume similar to an insulin pump) and initiate and lead new projects (e.g., developing an evidence-based information resource to help transplant recipients return to work).
Responsibility to End Users.
As lone voices in research groups, participants reminded the team of “why they’re doing this research” by sharing firsthand insights of their lived experience. Participants considered themselves as advocates for other patients/caregivers and felt a responsibility to use the opportunities they were given to represent the broader patient voice, particularly for specific groups that were often underserved, such as children with CKD.
Discussion
Patients with CKD and their caregivers contended with a multitude of treatment and symptom burdens and caring responsibilities, which hampered their capacity to contribute to research. Initially, they lacked confidence and struggled with their role as an “expert” among health professionals and required support from researchers, and, in some cases, financial compensation, to feel accepted as equal partners. Patients and caregivers felt burdened with the responsibility to be proactive in seeking limited opportunities to become involved, often at personal cost, which they felt hindered diversity and inclusiveness. They also faced stigma of their “patient status” due to poor understanding and awareness of CKD by the general public, and even by some health professionals, who laid blame on them for their disease, discouraging involvement. Historical power dynamics of health systems and research culture, including minimal time with their clinicians, limited opportunities to build trusting relationships and better understanding of their illness, which they believed would lead to greater involvement in research. Broader skills and expertise from their personal and professional lives were overlooked, and critical connections to important stakeholders, particularly the patient community to whom they felt a responsibility to educate and advocate for, were often ignored.
Although the findings were broadly consistent across the participants, there were some differences noted by country. Shame and stigma associated with CKD diagnosis was particularly notable in participants from the United States, who identified this as a major barrier to involvement in research. Patients from the United States were also concerned that clinicians were discouraged from educating their patients due to financially motivated time constraints and, thus, were unable to develop ongoing trusting relationships. Patients receiving in-center dialysis did not seem to be supported to take ownership of their treatment decisions and care. Both led to patients who were uninformed, with little understanding of, or interest in, becoming involved in research. In contrast, participants from Australia and the United Kingdom more frequently described how they were positively supported to be involved in research projects.
Patients with HIV/AIDS have similarly reported discrimination and stigma associated with disclosing their patient status as a major barrier to being involved in research (22,23). Participants from our study believed better education and public awareness could serve to reduce the shame and stigma associated with patient status. Studies in patients with HIV/AIDS and cancer have previously identified the power imbalance between patients and researchers, highlighting patients’ lack of trust in the research community and confidence in themselves as contributors (23,24). Holding meetings in community venues and sharing benefits directly with the community were suggested to avoid “drive by” (23) research, shift the focus to implications and outcomes for the community, and build confidence and trust, which could be effective in the CKD population. A further power imbalance identified in our study was the challenge to include the patient who was unwell. A challenge that may be specific to kidney disease is that patients may be overwhelmed by kidney disease and treatment, which can be a barrier to being involved in research. Due to the lifelong nature of CKD, patients were more commonly involved in periods of good health, such as when dialysis was been well established or when their transplant was functioning well. We suggest that flexibility in terms of timing, level, approaches, and support to enable patients with kidney disease to contribute to research is likely to be required.
This study provides in-depth insights from patients/caregivers from four countries, with varied experiences of CKD and involvement in research. We reached thematic data saturation and used investigator triangulation to ensure the themes reflected the breadth and depth of the data. However, there are some potential limitations. All participants were from high-income countries, English speaking, and with high educational attainment. Participants were key informants known to investigators to have been involved in research to provide relevant data on the basis of their experiences; however, they are likely not typical of patients and caregivers broadly, or those specifically involved in research. This may limit the transferability of findings because the themes identified may not be transferable to the broader population of patients with CKD and caregivers involved in research. Also, perspectives of those who may have faced barriers to involvement and low-income settings and disadvantaged groups were not included in this study. Although we reached thematic data saturation, we are uncertain about saturation of themes within specific countries from which there were a limited number of participants. The dearth of indigenous voices and cultural diversity needs to be addressed in future work, with a focus on codesign with these communities. Most of the findings related to involvement in priority setting, study design, recruitment, and reviewing research. There were little data on involvement in the analysis, evaluation, and implementation stages of research (Tables 1 and 3). This mirrors findings from reviews on patient involvement in research more broadly that demonstrate most involvement takes place in the planning and preparation phase (8,25,26; T. Gutman et al., unpublished observations). When patients/caregivers are not involved in the execution and implementation phases of research, they are unable to ensure their contributions and priorities are carried through the research project, limiting their value and effect.
Illustrative examples of participant contributions to research
Novel to this study are suggestions and considerations from the participant perspective on the basis of their experiences being involved in research (Table 4). We suggest researchers (1) acknowledge the inherent power imbalance patients and caregivers encounter in being involved in research, and (2) implement formal mechanisms codesigned with patients/caregivers to counterbalance this. The often invisible challenges patients/caregivers face when becoming involved in research include limited experience in the research setting, unfamiliarity with jargon and acronyms, and intimidation by experts. These have been similarly identified in other health disciplines and can discourage open and honest contributions (23,27). Formal induction processes, a terms of reference outlining expectations and roles for patients/caregivers and researchers, a dedicated contact for patients/caregivers, leaders encouraging inclusivity, and ensuring time and opportunity for patients/caregivers to be heard among professionals can help optimize their involvement (28).
Patient and caregiver recommendations for involving them in research
Educating and involving patients in shared decision making “decreases informational and power asymmetry” (29) between patients and clinicians, helping to establish trusting relationships and set up a dynamic whereby patients can develop confidence in themselves as experts and equal contributors to their care (30). More research is needed to better understand ways in which shared decision making in clinical settings can be leveraged to develop future relationships with researchers and allow patients to conceive their role and value as contributors to research. There is potential for clinics or hospitals to connect patients/caregivers with researchers through information sessions that provide education to patients/caregivers and opportunities for them to get involved in research.
There is also a need for more consistent involvement of patients/caregivers across the research cycle (26). More work is needed to understand how to better support meaningful involvement in the later stages, particularly in evaluation and implementation of research findings, and disseminating results back to the patient community.
The burden of CKD, limited opportunities, and power asymmetry between patients/caregivers and researchers were challenges to meaningful involvement in research. Building trust, awareness of opportunities, and recognizing the broad expertise and value of patients/caregivers, including and beyond their illness experience, may better support patient and caregiver involvement in research in CKD. Optimizing involvement in these ways may serve to better align research priorities, produce higher quality research of greater importance to all stakeholders, and enhance implementation of findings to practice, resulting in better care and health outcomes for patients.
Disclosures
A. Kelly reports receiving honoraria from Amgen (2014), Bristol Myers Squibb (2018), and Roche (2017) and reports other interests/relationships as Cochair of the Medication Adherence Special Interest Group of Outcome Measures in Rheumatology. J.C. Craig reports serving as Coordinating Editor of Cochrane Kidney and Transplant, on the Editorial Boards of Clinical Epidemiology and Prognosis and Diagnosis, and as Vice President of Flinders University. N. Scholes-Robertson reports serving as Patient Editor of Cochrane Kidney and Transplant and as a scientific advisor or member of the Rural Kidney Association Inc. (NSW, Australia) and reports other interests/relationships with Better Evidence and Translation in Chronic Kidney Disease (BEAT-CKD) Consumer Advisory board (Australia) and Rural Kidney Association Inc. (NSW, Australia). S. Jesudason reports receiving honoraria from Baxter and as Associate Editor of Journal of Nephrology, reports being a scientific advisor or member of the National Indigenous Kidney Transplant Taskforce, and reports other interests/relationships with Kidney Health Australia and nongovernmental organization advisory committees. T. Gutman reports other interests/relationships on the Patient-centred Research Network coordinating committee and the Standardised Outcomes in Nephrology Initiative coordinating committee (no financial relationships). All remaining authors have nothing to disclose.
Funding
This work is supported by the National Health and Medical Research Council NHMRC, through Postgraduate Scholarships (1169149 to T. Gutman and 1190850 to N. Scholes-Robertson) and a Fellowship (1106716 to A. Tong).
Acknowledgments
The funding bodies and institutions had no role in the study design, collection, analysis, and interpretation of data; writing of the report; and the decision to submit the report for publication. We would like to thank and acknowledge the participants that contributed to their time, knowledge, and expertise to this study. The following participants consented to be named in acknowledgments: David Baron, Helen Coolican, Vanessa Cullen, Amanda Dominello, Pam Duquette, Denise Eilers, Derek Forfang, Kevin Fowler, Nieltje Gedney, Chandana Guha, Tess Harris, Luke Macauley, Michael Mittelman, Shyamsundar Muthuramalingam, David Rosenbloom, Nicole Scholes-Robertson, Henning Sondergaard, David White, and Caroline Wilkie.
Supplemental Material
This article contains the following supplemental material online at http://cjasn.asnjournals.org/lookup/suppl/doi:10.2215/CJN.05960521/-/DCSupplemental.
Supplemental Item 1. Participant organizational affiliations.
Footnotes
Published online ahead of print. Publication date available at www.cjasn.org.
See related Patient Voice, “Advancing Patient-Centered Research: Enabling the Patient Voice to Be Heard,” on pages 171–172, and editorial, “ What Patients Teach Us About Patient Engagement in Research,” on pages 176–178.
- Received May 1, 2021.
- Accepted November 16, 2021.
- Copyright © 2022 by the American Society of Nephrology
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