Patient and Caregiver Experiences and Attitudes about Their Involvement in Research in Chronic Kidney Disease

Abstract

Visual 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.

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.

Table 1. - Characteristics of patients with CKD and caregivers who had previously been involved in research, n=23

Characteristic	n (%)
Patient	18 (78)
Caregiver	5 (22)
Female	13 (57)
Age, yr
<30	1 (4)
31–40	5 (22)
41–50	4 (17)
51–60	8 (35)
61–70	4 (17)
>70	1 (4)
Country of residence
Australia	10 (43)
United States	10 (43)
United Kingdom	2 (9)
Denmark	1 (4)
Ethnicity
White	19 (83)
Indian	2 (9)
Black	1 (4)
Chinese	1 (4)
Education level (highest attained)
Postgraduate degree	10 (43)
Undergraduate degree	10 (43)
Professional certificate	1 (4)
High school	2 (9)
Employment status
Full time	9 (39)
Part time	9 (39)
Retired	2 (9)
Student	1 (4)
Disabled	1 (4)
Contract	1 (4)
Involvement in research—stage
Identifying/prioritizing	20 (87)
Designing	9 (39)
Recruiting	14 (61)
Conducting	8 (35)
Reviewing	18 (78)
Disseminating	7 (3)
Implementing	6 (26)
Evaluating	5 (22)
Involvement in research—role
Advisory board a	11 (48)
Steering committee b	14 (61)
Expert working group c	7 (30)
Workshop participant	16 (70)
Involvement in research—h/wk
<1	7 (30)
1–2	9 (39)
3–4	2 (9)
5–10	3 (13)
>10	1 (4)
Interview setting
In person	11 (48)
Online	12 (52)

^aA volunteer group consisting of mostly, or all, patients who provide guidance, feedback, and advice to the research group at all stages.

^bCommittee that determines priority/agenda items and strategic planning, consisting of 1–2 patients among other health professionals.

^cGroup of experts working together on a specific subject area to achieve specified goals.

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.

Table 2. - Illustrative quotations

Theme	Illustrative Quotations
Grappling with CKD diagnosis
Overwhelmed by the burden of illness	It’s time consuming. It’s scary… all of a sudden, it’s permeating their whole life. I’m supposed to have time to get involved in research? I don’t have time to do anything except take care of myself…. They want to distance themselves from an all-pervasive disease, I think, in many instances. (Caregiver, United States) There were times that I could not even fathom, don’t talk to me about anything, just keep her alive right now. (Caregiver, United States) It's been hard for me the last few weeks because I haven't been well…. So, I think that would be very dependent on how well they are, which is a shame because that's probably when we need them to be involved… you're experiencing CKD at perhaps its worst and you might have valuable input for researchers about this is what I'm experiencing. (Patient, Australia)
Silenced by stigma and shame	People get a bad rap with dialysis because they go three times a week, they’re fluid overloaded, they [dialysis staff] blame them. Patients get blamed a lot of the time for having CKD…. A lot of people think it’s hereditary or it’s something that you do. I can’t tell you how many times I’ve told people I’ve been on dialysis and they were like “oh, you did drugs and you used alcohol?” and I’m like no, that’s not it at all…. It has nothing to do with anything that I did. I think people shut down, because who wants to get blamed, and who wants to be told you’re not doing the right thing? It’s hard enough as it is. (Patient, United States) There’s also kind of a stigma, unfortunately, with kidney disease. Some people think it’s communicable. (Patient, United States)
Absence of advocacy and awareness	Kidney disease isn’t sexy. It’s not the attractive thing. Being a woman, I walk in and I just saw some cereal, it had a pink banner across the top. My immediate thing was recognition of breast cancer. We do not have a high recognition. (Caregiver, United States) I just like getting the word out, because it’s the only way things are going to get done. I was like, it was kind of nice that Selena Gomez came out. She didn’t have to, that was pretty cool…. I hate to say this, but sometimes you need a big marquee kind of figure. (Caregiver, United States) Not many people know how bad kidney diseases are… whereas if you have a cancer, I'm not downsizing cancer's importance, but the importance that it gets rather than the kidney disease, because if you are diabetic, you're prone to kidney disease, if you're obese, you're prone to kidney disease. If have a hereditary condition, you're prone to kidney disease. So there are a lot of chances to get a kidney disease than to get a cancer, but the people take it so easily when you have a kidney disease and the lack of awareness, and even preventing the progression of getting kidney diseases. (Patient, Australia)
Bearing the responsibility for involvement
Autonomy in their own care	Especially here, dialysis is so paternalistic, and we’re not taught that we want to advocate for ourselves. (Patient, United States) There are a lot of people who are getting more engaged in their own treatment, and I think that’s the first step before they start getting involved in other things. (Patient, United States) Obviously, the clinic is your number one captive audience, I mean you’ve got 90% of the patients in the American population sitting in clinic. Of those, let’s just say 40% are not even capable, but you’ve got still 50%…. What I call it when I’m speaking is you check your independence at the door. The 90% of the patients that choose in-center walk in, check out, sit in a chair, and they’re done. (Patient, United States)
Forced to be proactive to access opportunities	I kept telling them I wanted to be involved… there’s not a lot of opportunities to be involved in research unless you’re doing drug research. (Patient, United States) I'm cheeky, I don't mind reaching out to someone. If I know they know something, I'll just write them and introduce [myself] and usually we know some of the same people and we refer to them or whatever… personally I just have the drive. (Patient, Denmark) If you make it hard for patients to get involved in something, you may just lose them forever. It may be their only chance to interact in a different way with the organization or with the community. (Patient, United States)
Infrastructural support to connect researchers and patients	I think the biggest failing is re-engaging the people who already are interested, just relying on them to see an EOI or see something that one group puts out somewhere, I just think there's a lot of missed opportunity there for recruitment. (Patient, Australia) We’re trying to come up with that, the National Kidney Foundation, like a board on the web that patients can go to and look at research opportunities…. The Juvenile Diabetes Foundation, they have a great way of doing that. What they do is you can go in and you can register to be part of their research… it gives me options on what I’m willing to do. Am I willing to go into hospital? What am I willing to do? And where am I willing to go to? Is it local? Am I willing to go out of state? And then on the basis of those results, when research comes up that fits my demographic of whatever, they will kick that opportunity to me via email. (Patient, United States)
Battling big agendas
Struggling in a system of disincentive	There are people now running large hospital systems such as the University of Southern California, such as UCLA, that are bean counters, that are basically accountants. They’re trying to stretch that dollar as far as they can, and therefore, they dictate how much time you have with each patient. (Patient, United States) In many cases, the system is designed to be paternal. It pacifies the patient. It says come three times a week, sit down, shut up. (Patient, United States) “I don’t know how to educate my patients. I don’t have time.” The system doesn’t reward you. We’re dealing with is a dysfunctional system. (Patient, United States) Clinics don’t let researchers in, because they only want research that favorably affects their business… because our entire dialysis is a business model and not a health model, that’s an issue. (Patient, United States)
Changing research culture	They’re very good doctors, and they’re younger. Most of them are way younger than I am. It’s very interesting, it’s a real give and take, it’s a real two-way street. We need more of that. We need more of that conversation to go on. (Patient, United States) I think I’ve seen a change in the last 2–3 years, where instead of a paternalistic type thing, where the doctors or the nurses [have a] know-best attitude. That’s really gone away, it’s going away, let’s put it that way. Where we realize it is a partnership between the patients. (Caregiver, United States) I think a good example is the American Society of Nephrology, what they’re doing. The American Society of Nephrology is making a sincere effort to change the culture because they realize they have to… the Clinical Journal of American Society of Nephrology now has patient editors on their editorial board. I think those are very tangible actions that are not words. I think there’s a lot of words, but think the American Society of Nephrology is actually leading with some actions. (Patient, United States) Sometimes it's like a bit of a “tick the box.” Do we have the consumer voice? But I think more emphasis is being put on that, so that's good…. This consumer involvement is quite new, so it's a learning curve for everybody. I think it's changing because people are becoming more aware of how to involve consumers better. It might be starting like ticking the box, but it is growing to be more than that. (Patient, Australia)
Becoming equals	But personally, for me, there is a tendency to sit back and not cut in with something that I may think is important to say or contribution I have because I'm thinking of myself as the patient, and still on a much lower level to the professors, and the doctors, and the surgeons, and whoever else might be in the room… it's taken for granted by other people that they would speak up, when that's not natural or normal for the patient who is used to laying in a hospital bed, receiving instructions about how to care for themselves. It's like role confusion. Over time, you can come to expect, “Okay, my opinion and input here is wanted and warranted,” and either time is going to be made for that or you feel confident enough at a certain point to put your hand up and say, “What about this?” (Patient, Australia) I think it has been a good experience, and I feel [like] a valued contributor. I don't feel just like the patient at the end of the line, “Oh, what do you think, by the way?” And I feel like a valued member of the team and it felt all very collaborative and very professional. Again, it wasn't a case of, “Well, there's the clinicians and here's the patients.” It was very together, which I think is really nice and really important. (Patient, United Kingdom) On a lot of the steering committees and scientific committees, because I've been doing this now for a few years, it feels like we now are only starting to get traction with some equity in respects mostly. This is where I've almost ended up in conflict resolution–type processes because with some people in the profession from the research side, there's been a real pushback against treating the patient as an equitable partner and an expert in their field. (Patient, Australia)
Seeing the person behind the patient
Utilizing broader knowledge, expertise, skills, and interests	One of the other problems you have of getting patients involved is patients are only looked at on the basis of what’s on their medical charts. Not who they are. Not the fact that they might have been a CEO of a company, or they might have been a university professor. (Patient, United States) People want to treat consumers like they can just bring their lived experience and that's it. They can just quote their emotional stories and move on. No, no, no. I want the research methodology to be strong. I want the research dissemination to be strong. (Patient, Australia) Another example I see a lot of is, “Oh, we've done this research and now we want to make an infographic.” Someone in the office who doesn't have training and experience in how to create infographics, “We'll put it together because we don't have a budget.” I go, “Well, what if you went out to your consumers and said, ‘Does anybody work in this field? Is anyone a graphic designer and would like to donate their time pro bono to create this professional infographic for us?’” Harness the ability of these people because they're here. Why do you guys only look at us as someone to ask questions about tolvaptan? (Patient, Australia)
Understanding patient needs	One of the research groups I'm on did an introductory video that explained things. Trying to limit the amount of reading, I think. And having more diagrams and really short, a few groups are pulling out lots of short research summaries. (Patient, Australia) There are so many clever people where they're involved with the research and who know so much. And occasionally, I think it's forgotten that I am only a consumer. And although I'm a nurse and have that background, there's still a lot that goes over my head. (Patient, Australia) Instead of saying renal, say kidney to me. So those simpler words, switching the words where consumers can understand. (Patient, Australia) What I ask my researchers to do is develop a simple one-page summary of your research. And then next to it have how long I need to spend on that research. For example, how many meetings I need to do if I'm going to do 3 months, or if I'm involved in this research for the next 1 year, how many meetings do you want me, and how long those meetings are? Breaking that into simpler steps, because I'm living with that chronic condition and have dialysis and I have a treatment to go through. So instead of you just coming, saying, “[Patient], are you available?” You just have to set the agenda, or set the tone, right from the start saying, “We expect you have to spend 20 minutes for a meeting, for the next 3 months in three meetings.” And what skill set do you require from me. (Patient, Australia)
Motivations for involvement	I was always on the lookout for changing things, making things better. I just feel I've got so much experience, so much knowledge, and so much sadness, I have to turn it into something positive. I need something good to come out of this. So that's always been my driving force. I have to use it somehow. (Caregiver, Australia) I started to talk to other patients and I took a leadership role, started using some of the skills I had from my career and put them into my work with the kidney groups, and started feeling a sense of purpose again. (Patient, United States) I was looking to add things to my professional résumé… it was professional development, but also, if anything, I thought I would be able to help more patients and if I were going to go to work on digital-related things, I could use those for later jobs. (Patient, United States) Research is a way to stimulate my brain and my thinking… opportunities to meet people, make connections, learn more stuff, and that all helps me. In a way, it helps me cope. It helps me cope with sometimes the realities and the crap part of having kidney disease, which is not always a great deal of fun. (Patient, Australia)
Sensitivity to complexities of payment
Accounting for individual circumstances	With the way my disability’s set up, I’m not able to accept too much without losing some benefits, such as my medical, so it’s very important. I find it more rewarding, to volunteer, because then I don’t have an agenda. It allows me to get invited to sit at the table with people that otherwise maybe I couldn’t have these conversations with… especially at a government level, where they know that I’m not there representing a corporation or anything like that. (Patient, United States) One friend of mine, she can't be compensated, nor does she want to be compensated because in the United States, she's on something called Social Security Disability Income… if she gets too much, then the government sees her as working or capable, and they're like, “Well you don't get access to that anymore, we're taking that away,” and she's afraid to lose that. So, she does more of, I want you to pay all my expenses to come to this meeting. On the other side of it, I know a patient, he does so much work, he would absolutely in a heartbeat say, “Yes, I should be compensated for this.” The most challenging piece is that it's different for each person. (Patient, United States)
Denoting value	I don’t get paid. Every time I raise that issue, I say we’re not getting paid for this. And there’s silence. It’s like, “These suckers think we’re actually going to pay them?” (Patient, United States) So if you're an academic journalist, or a senior editor, or reviewer for some group and you're asking a patient to review, whatever that person gets paid to review, the patient should get the same… equal to the other people participating. (Patient, United States) I know there's a lot of people with CKD who are also researchers who are doing an awful lot, basically, for free. I think that's almost really taken for granted because it's like, “Oh, you're a consumer,” but to me, it's just going too far. It's really disrespectful. (Patient, Australia)
Enabling diverse involvement	If there's financial support, your foods are taken care of and things like that, I think that gives patients opportunities to come down and be involved. I think sometimes it's just being aware that some people won't have the money there to even catch the train. (Patient, Australia) Because it’s not always put into the budgets. Some of the universities insist on adding overheads, so if you’ve got an expensive researcher and then you’re left with patient involvement, the tendency is to go for the person who’s geographically closer, can walk into your building. I think cost is a barrier, but that’s because it’s not included early enough in the process. Maybe they should mandate a particular percentage [for consumer involvement]. (Patient, United Kingdom) It’s very heavily dependent on a few highly motivated individuals who give up a lot of their own personal time, at personal cost to get involved. (Patient, United Kingdom)
Championing the patient voice
Links to important stakeholders	We meet with Medicare, CMS Medicare, to talk about reimbursement issues and the quick measures, the quality measures, because those have to be patient meaningful… we were invited by the Government Accounting Office, to come and give input to them when they were mandated by Congress to look into the barriers for home dialysis. (Caregiver, United States) I’m networked to other patients out there and I know people’s strong points and skills. (Patient, Australia) I do a lot of advocacies on Capitol Hill, again because of my proximity, being an hour away there’s no excuse not to. (Caregiver, United States) I chair that [clinical study group] and rally people together and encourage patient participation in all of the studies that people are planning to do…through being the CEO of the UK group, I can then reach thousands of patients to take part. (Patient, United Kingdom)
Drivers of innovation	A lot of them would be healthy, relatively, but they wouldn't have the courage to go back to work. There wasn't so much information out there on the web. And I felt recipients do need that. They need to, when they are down, when they're sick, when they're looking for information, they need to find something that they can read, and be informed before they go in for their surgery, while they are going through the experience, and even after. So that's what I did. I put together this repository of information online for recipients across all sorts of organs. (Caregiver, Australia) I proposed a research project on a device to detect your optimum fluid in your body. We went through their process, they looked at it through what’s called the Kidney Health Initiative and decided to support that idea. It’s the first patient-approved project that they’ve ever done, so it’s pretty exciting. (Patient, United States) I recruited eight patients who have quite significant experiences of pain, and we had a day and a half workshop. Afterward, the primary researcher said that there were some things that he’d never expected that came out of these 2 days. Since then, KDIGO always had a patient-carer peer group within their meetings. I do think we were pioneering in that respect, to making sure our voice was heard as equally as the geneticists’ voice and the nephrologists’ voice. (Patient, United Kingdom)
Responsibility to end users	I think the patient voice is really important. Even if we don’t necessarily speak up all of the time, we remind people why they’re doing this research. (Patient, United States) There is an opportunity for research gains and possibly policy gains or treatment gains to be made from every person contributing what they can. With my stake being a little more unique, there is a bit of an obligation that I want to fulfill in contributing what I can. (Patient, Australia) It’s a pretty huge responsibility to represent the pediatric [voice], I’m the only one in this particular, and it’s going to expand in time but right now we serve a certain term, so I’m always on the lookout for someone to supersede me when I go. I do not want to lose a pediatric voice. (Caregiver, United States)

EOI, expression of interest; UCLA, University of California Los Angeles; CEO, chief executive officer; CMS, Centers for Medicare & Medicaid Services; UK, United Kingdom; KDIGO, Kidney Disease Improving Global Outcomes.

Figure 1.: 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.

Table 3. - Illustrative examples of participant contributions to research

Stage(s)	Activity
Priority setting	• Contributed to grant proposals and applications • Identified research priorities • Lobbied government for funding • Conducted community consultations • Contributed to developing a national strategic action plan and submitted to government for funding
Research design	• Designed and developed surveys • Designed a flyer for recruitment • Provided feedback on patient-reported outcomes • Simplified enrollment processes for a trial • Contributed to device design (risk/benefit, tolerance, etc.) • Contributed to trial design • Provided lived experience to inform intervention design • Reviewed patient-facing documents for readability • Edited research protocol with patient-friendly language
Data collection	• Conducted survey • Conducted interviews • Conducted focus groups • Recruited participants for trials • Recruited participants for workshops • Recruited participant for focus groups • Facilitated workshops • Provided access to participant groups/databases/networks
Data analysis	• Reviewed data • Analyzed qualitative data (interview transcripts)
Dissemination	• Wrote research papers/reports • Presented the research at universities to medical students • Edited scientific journals (with an emphasis on patient/caregiver involvement) • Moderated panels • Publish research reports to patient/caregiver audiences • Presented and shared research findings with other patients/caregivers • Attended and presented at scientific meetings • Held webinars for patients/caregivers • Developed plain-language summaries and infographics for patients/caregivers • Created informational videos for patients/caregivers
Implementation	• Changed policy on home dialysis without a caregiver • Wrote a submission to regulator to support drug approval after survey study
Evaluation	• NA
All stages	• Served as steering committee member • Advised on patient/caregiver involvement strategies • Cochaired project steering committee • Developed an international patient registry • Led research work groups • Patients/caregiver organizations partnered with research groups • Involved across all stages of developing an internationally validated pain score for PKD • Sought funding and conducted focus groups to develop patient decision-aid

PKD, polycystic kidney disease.

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).

Table 4. - Patient and caregiver recommendations for involving them in research

Theme	Suggestions and Considerations from the Participant Perspective
Grappling with CKD diagnosis	• Involve patients early in their journey so they are inducted and equipped to be involved and can contribute, even in periods of illness. Consider the support that may be required to facilitate this • Use caution and care when approaching someone who is unwell to become involved for the first time. Consider passive approaches such as posters or flyers in this instance • Encourage and support patients/caregivers to build confidence to be involved • Consider that because patients with CKD have a lifelong illness, “there are lots of people who are available to provide input” and they are potentially able to be involved “from the start through to the finish of any projects because they live with that experience for long periods of time” • Provide education and increase awareness of CKD in the broader population to discourage discrimination and stigma
Bearing the responsibility for involvement	• Encourage patients/caregivers to become active participants in their own care through strategies such as shared decision making • Cultivate relationships in clinical care to start the conversation about becoming involved in research • Educate clinicians on how to reach and communicate with patients/caregivers • Demystify research—educate patients/caregivers about research, its benefits for them, and opportunities to become involved • Use multiple channels to reach patients/caregivers and advertise opportunities for involvement (e.g., social media, flyers, email) • Develop relationships with patient/caregiver groups and provide information and education sessions explaining your research and how patients/caregivers can become involved • Develop a database of interested patients/caregivers that can be contacted and re-engaged for new projects • Invite patients/caregivers to participate in scientific meetings
Battling big agendas	• Aim to involve patients/caregivers as equal members of a collaborative research team (e.g., include in all meetings and correspondence) • Educate clinicians in medical school about how to work with patients/caregivers • Consider communication training for clinicians • Ensure simple language is used • Develop a “patient-centered research standard” • Enlist patients/caregivers to assist in developing involvement strategies and avoid tokenistic involvement • Open access to scientific meetings to patients/caregivers including patient/caregiver speakers in equal roles and sessions specifically designed for patients/caregivers
Seeing the person behind the patient	• Develop a database of patients/caregivers and include profiles that contain their interests, skills, and expertise and their lived experience • Consider relevant experience when selecting or matching patients/caregivers with research projects • Develop a terms of reference, outlining what is expected of patients/caregivers, including their roles/tasks, number of meetings, expected workload in hours, time frame of the research, and privacy and confidentiality policies • Take time to talk to patients/caregivers about any special needs they may have, including physical and cognitive challenges, technology, or logistic support • Consider providing education or training for patients/caregivers from researchers on basic research methodology and subject matter background • Provide support/assistance to help patients/caregivers voice their perspectives, particularly in settings where they are working with clinicians/researchers • Include more than one patient/caregiver in any group • When scheduling meetings, ask patients/caregivers for their availability • Consider having a dedicated patient/caregiver liaison so they know who to contact with concerns or questions • Be sensitive to patient/caregiver experiences, including taking time to understand their individual journey, and use care when talking about potentially challenging topics, e.g., mortality • Communicate often throughout the process, including explaining any delays • Report back to patients/caregivers at the end of the process to explain what has come out of the research, and how they contributed • Consider using an “interest-based” approach when selecting patients/caregivers with which to work • Highlight the potential benefits to patients/caregivers individually (e.g., education, purpose) and more broadly (e.g., improving future patient outcomes)
Sensitivity to complexities of payment	• Include patient/caregiver involvement in the budget at grant stage—view this as an investment similar to any other budget items that contribute to research quality • Use a cost calculator or budgeting tool such as the one from INVOLVE to understand rates of compensation • Be upfront with discussions about payment/compensation • Always ask the patient/caregiver for their preferences, or if payment may affect the benefits they receive • Be transparent about what other team members are receiving for their involvement, and align patient/caregiver compensations with this, ensure they feel valued as an equal team member • Cover any expenses incurred in advance—patients/caregivers may not have access to funds or credit cards to cover expenses up front
Championing the patient voice	• Utilize patient/caregiver networks and connections to expand involvement • Utilize patient/caregiver networks and connections to gain support from policy makers, government, patient organizations, and the public • Ask for input, ideas, and solutions from patients/caregivers from their lived experience • Build capacity to support patient/caregiver-led research through mentorship and training
Summary of implications for improving consumer involvement	• Education and training for both consumers and researchers to address barriers and power imbalances. This would include communication regarding expectations and roles of all involved to ensure meaningful and sustainable involvement across the research cycle • Always consider the person behind the “patient’ when working with consumers and highlighting the other skills that are brought to their involvement 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).