VIEWS, VISIONS AND VISTAS IN DIALYSIS

Determining Research Priorities Through Partnership with Patients: An Overview Lianne Barnieh,*† Min Jun,* Andreas Laupacis,‡§ Braden Manns,*†¶** and Brenda Hemmelgarn*†¶** *Department of Medicine, University of Calgary, Calgary, Alberta, Canada, †Interdisciplinary Chronic Disease Collaboration, Calgary, Alberta, Canada, ‡LiKa Shing Knowledge Institute of St. Michael’s Hospital, Toronto, Ontario, Canada, §Faculty of Medicine, University of Toronto, Toronto, Ontario, Canada, ¶Libin Cardiovascular Institute and Institute of Public Health, University of Calgary, Calgary, Alberta, Canada, and **Department of Community Health Sciences, University of Calgary, Calgary, Alberta, Canada

ABSTRACT There is an increasing level of emphasis being placed on health care providers and funders to incorporate patientcentered care into research. Involving patients and caregivers in establishing research priorities ensures the relevance of the research produced. Priority setting is a process that can be used to produce a robust set of research questions that researchers can address over the coming years. One of the methods for determining research priorities that involves patients, caregivers and clinicians is the James Lind Alliance priority setting part-

nership model. This method is focused on being exclusive, transparent, and evidence-based. Using a recent example of patients on or nearing dialysis, we highlight the key steps to assess research priorities in patients, caregivers and clinicians: (i) formation of a steering committee to guide the overall process; (ii) form priority setting partnerships; (iii) identify and gather research uncertainties; (iv) process and collate submitted research uncertainties; and (v) final priority setting workshop to determine the top 10 research priorities.

Background

as a priority by health funders, with the goal of building partnerships in research and ensuring the relevance of the research produced to patients and their caregivers (4,5). Patients and their caregivers should be included when establishing priorities for research in health care. The premise is simple: because they live with their disease, patients, and the individuals who care for them—the consumers of health care—should have a voice in setting research priorities (2). Traditionally, the process of setting research priorities in health care has not involved patients or their caregivers. Shedding light on the priorities of patients and clinicians in research can change clinical practice, by increasing the priority assigned to research topics that are relevant to both (6). Interventions are more likely to be effective when voiced by those who are impacted by them—patients—thereby potentially increasing the likelihood that research will be adopted into practice. Several countries have developed strategies for patient engagement in research (5,7,8). These strategies range from higher levels of engagement of patients in research and knowledge translation processes, to involving patients in determining the priorities for research. Canada’s Strategy for Patient-Oriented Research (SPOR) is a coalition of federal, provincial and territorial partners which emphasize a higher level of engagement of patients

The translation of research into practice is heavily influenced by the relevance and credibility of the research undertaken. While clinicians (physicians, nurses, and other allied health care professionals) and researchers often assume they understand the needs, views, and concerns of patients and caregivers, both as they relate to patient care and what they might consider research priorities, this may not be the case (1). Furthermore, research that has low relevance to advancing knowledge or immediate action can be viewed as wasteful (2). Increasing emphasis among health care providers and funders has been placed on patient-centered care, defined as care that is respectful of patient preferences and in which patient values guide clinical decisions (3). Given this, involving patients as key stakeholders in the research process has also recently been identified Address correspondence to: Brenda Hemmelgarn, Department of Community Health Sciences, Cumming School of Medicine, University of Calgary, TRW3D07-4 3280 Hospital, Dr. NW, Calgary, AB, T2N 4Z6, Canada, Fax: (403) 210-9286, or e-mail: [email protected]. Seminars in Dialysis—Vol 28, No 2 (March–April) 2015 pp. 141–146 DOI: 10.1111/sdi.12325 © 2014 Wiley Periodicals, Inc. 141

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in the research and knowledge translation process, with a focus on the interventions and outcomes considered important by patients, caregivers, and the clinicians caring for them. Despite the emphasis on patient engagement, a recent systematic review on initiatives that have engaged both patients or clinicians in setting research agendas identified only nine studies (of 258 which addressed the topic), where both patients and clinicians were involved in prioritizing research questions (9). If patients are to be engaged in setting research priorities, how does this happen? There are few established methods for eliciting patient, caregiver, and clinician research priorities. The Cochrane Agenda and Priority Setting Methods Group has identified three methods for priority setting in research that involve engagement from both patients and clinicians: the dialog model (10), the health equity lens model (11), and the James Lind Alliance (JLA) priority setting partnership model (12). The dialog model comprises four phases: (i) exploration, during which disease-specific patient groups are identified; (ii) consultation and prioritisation, where the research agenda is explored in more detail via interview; (iii) integration, which analyzes the data and integrates the priorities identified; and (iv) follow-up, which involves determining the extent to which the patient views were incorporated into recommendations (10). The global evidence mapping priority setting method focuses on systematic reviews and includes five main steps, with an overall consideration of health equity and social determinants of health. The research team first identifies systematic reviews relevant to the topic under study. Then, with the help of a facilitator, two workshops are conducted to identify the top research topics to prevent, treat and manage: one with patients only and one with clinicians, researchers and patients. In the next steps, the research team develops detailed research questions from the broad topics identified, and then the top 10 research topics are identified. The last step involves a different set of patients completing an online survey to prioritize the top 10 research questions (11). To date, the most common method used in engaging patients in setting research priorities is the JLA method which brings together patients, caregivers and clinicians in the priority setting process, creating a truly representative partnership between the groups, which is central to patient engagement in research (7,13). The JLA method focuses on identifying treatment uncertainties and defines an uncertainty as an area where there are no existing or up-to-date, reliable systematic reviews of research evidence (or large definitive trials) addressing either the uncertainty or the effects of a treatment. Uncertainties can include health care interventions, prevention strategies, testing, or rehabilitation. The JLA priority setting process is focused on being inclusive, transparent, and evidence-based, and has been used in more than 13 chronic diseases (14,15). The JLA method for research priority setting has become such an integral

part of the research community in the United Kingdom that in April 2013 the National Institute of Health Research took over the coordination of the JLA work (16). Our research group recently used the JLA method to assess the research priorities of patients on or nearing dialysis within Canada and their caregivers and clinicians. For the remainder of this paper, we will use this example to highlight the JLA process (17). The James Lind Alliance (JLA) Process The JLA process begins with creation of a steering committee, which guides the overall process. Creating a steering committee is a critical component of the JLA priority setting process, as they lead and contribute to all stages of the process. Members of the steering group need to commit to each stage of the process, including: publicizing the initiative to potential partners, participating in the initial awareness meeting, developing and distributing information and forms to gather uncertainties, collecting and collating uncertainties, checking uncertainties against existing systematic reviews, managing interim priority setting, publicizing and participating in the final priority setting exercise, and publicizing the final top 10 uncertainties as research questions to funders. Although there is no set formula on whom to include on the steering group, for the priority setting process for patients on or nearing dialysis, the 11 person steering group comprised four patients, one caregiver, three clinicians, an employee of the Kidney Foundation of Canada (an important funder of kidney research in Canada), an expert in the JLA approach, and a researcher. The steering group included individuals from across Canada. In accordance with the JLA’s fundamental aims, only patients and clinicians (who would not normally be involved in setting the research agenda) are able to participate in the priority setting exercise. An overview of the steering group responsibilities are available elsewhere (18). Once the steering committee is established, the JLA process follows four key steps to assess research priorities: (i) form priority setting partnerships; (ii) identify and gather research uncertainties; (iii) process and collate submitted research uncertainties; and (iv) final priority setting workshop to determine the top 10 research priorities (Fig. 1). Form Priority Setting Partnerships Partner organizations are important collaborators to assist not only in the distribution of the surveys (see below) but also in the final dissemination of information about the priority setting process. Partner organizations can be identified through a process of peer knowledge and consultation within the network of the steering group. In our example, the partner organizations included the Kidney Foundation of Canada, the

DETERMINING RESEARCH PRIORITIES THROUGH PARTNERSHIP WITH PATIENTS

Step 1:

Form priority setting partnership

Step 2:

Gather research uncertainties

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Electronic and paper-based surveys

Process and collate submitted research uncertainties

Step 3:

Systematic search for published uncertainties and research recommendations

Combining research uncertainties

Ranking research uncertainties

TOP 30 research priorities

Final priority setting workshop

Step 4:

Consensus meeting with patients, caregivers, clinicians and policy-makers

TOP 10 research priorities

Reporting and dissemination

Fig. 1. Overview of JLA method.

Canadian Society of Nephrology, the Canadian Associations of Nephrology Nurses and Technologists and the Canadian Kidney Knowledge Translation and Generation Network, among others. Identify and Gather Research Uncertainties Creating a taxonomy of potential issues for the disease under consideration can help guide the development and scope of the survey, and to categorize the survey responses. This taxonomy can be developed through guidance of clinicians. Uncertainties can also be identified through a search of clinical practice guidelines relevant to the care of patients under consideration. When searching clinical practice guidelines, the purpose is to identify guidelines with limited evidence as well as areas identified as requiring further research, thus representing additional potential research uncertainties. In the priority setting exercise for patients on dialysis, a taxonomy of 12 categories was created (Appendix 1). Relevant guidelines were identified form the following guideline groups: Kidney Disease Improving Global Outcomes, National Kidney Foundation/Kidney Disease Outcomes Quality Initiative, and the Canadian Society of Nephrology. A survey is used to identify and gather a broad list of potential research uncertainties. Survey questions should be phrased in a sufficiently broad manner that

helps elicit ideas about research and what is needed to improve the disease under consideration from respondents. Patients, caregivers, and clinicians are generally invited to complete the survey (often available online) through: communications from partner organizations, including through social media; advertising in newsletters of partner organizations; e-mails through partner organizations; and direct contact with relevant individuals (where applicable). The uncertainties identified by survey respondents and from guidelines are then combined in one document, and those deemed not relevant or that are unclear are removed. The remaining uncertainties are then assigned to one of the categories in the taxonomy. Returning to our dialysis example, patients, caregivers, and clinicians were invited to complete the online survey through communications from organizations such as the Kidney Foundation of Canada, Canadian Society of Nephrology and Canadian Associated of Nephrology Nurses and Technologists, the Renal Pharmacist Network, and the Canadian Association of Nephrology Administrators. The survey consisted of broad prompts including questions about the overall management of severe kidney failure and dialysis as well as issues related to the diagnosis, prognosis, and treatment of kidney failure. The 1820 uncertainties were then examined to identify those not relevant to patients

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on dialysis (e.g., management of a patient with a kidney transplant or questions about preventing renal disease). There were also 87 uncertainties identified from clinical practice guidelines. Process and Collate Submitted Research Uncertainties The interim priority setting stage may be carried out by the whole partnership, or in the initial stages, by the steering group. The goal is to categorize uncertainties into similar groups, and in some cases, develop a summary question from like questions. The JLA does not impose a strict method for this stage. A document is then circulated to the Steering Group members with the summary questions in each of the categories, all of the uncertainties, including the number of times the uncertainty was identified by patients, caregivers, or clinicians, and the source of the uncertainty (survey or clinical practice guideline). Each member next independently ranks his or her top 30 research uncertainties. The ranked priorities are then scored and combined to determine the interim prioritized list. This process is closely monitored by the JLA to ensure transparency and minimization of bias. At this stage, the steering group should also determine whether any of the uncertainties have been answered by high-quality research. At the end of this stage, a shortlist of the top 30 research priorities is produced which is then considered at the workshop. In our priority setting exercise, of the 1570 uncertainties noted above, 144 summary questions across the 12 categories were identified. Members of the steering group then worked in pairs, with consideration given to combining a clinician with either a patient or caregiver to ensure a balanced perspective, to assign uncertainties into similar groups, with the goal of identifying a summary question for each group. Table 1 provides an example for vascular access, one of the 12 categories identified a priori. There were also 85 unique summary questions that did not fit into one of the 12 categories. Review of the clinical practice guidelines provided 57 uncertainties that were combined into summary questions, of which 30 were unique. The summary document was prepared with the 259 uncertainties identified from the survey and the guidelines, from which the Steering Group chose the top 30 for the in-person workshop.

Final Priority Setting Workshop The purpose of this final stage is to determine the top 10 research priorities. A minimum of 12 and a maximum of 30 patients, caregivers, and clinicians representing both different clinical profiles and geographic locales are generally identified through the steering group networks as well as through the partner organizations. Each participant must be justifiable as an “expert” in some way, be it clinician (health professional caring for those with the disease) or patient/caregiver (direct experience with the health problem). These participants are invited to participate in a 1-day workshop. Facilitators with experience in the JLA methods lead the workshop, and focus on enabling participants to express their views, hear different perspectives and think more widely about treating or helping people with the disease under question. The format of the workshop is structured but flexible enough to allow participants to express their opinions or raise concerns. Challenges that can arise with this format of workshop include ensuring the choice of participants is balanced, avoiding domination by any one person, cost, and finding unanimity. These workshops often use a nominal group technique approach (19), which is group process involving problem identification, solution generation, and decision-making and is often used in groups who want to make a quick decision, but want everyone’s opinions taken into account. This technique also involves small and large group exercises, soliciting input from all members of the group, followed by a ranking of the potential priorities. All participants are made aware of at the beginning of the workshop that the purpose is to determine the top 10 research priorities representing the views of all those who participated. Using our previous example, the final priority setting workshop was a 1-day event including 30 participants from across Canada, including 11 patients, five caregivers, eight physicians, six nurses and a social worker, pharmacist, physiotherapist, and dietician, representing a broad spectrum of people with or caring for kidney disease. Table 2 provides an example of the ranking form for vascular access only (and not all 12 categories). The top 10 research uncertainties identified at this workshop are highlighted in Table 3.

TABLE 1. Example of uncertainties and summary question across one taxonomy category Respondent type Health professional Patient with kidney disease Patient with kidney disease Patient with kidney disease Patient with kidney disease Patient with kidney disease

Uncertainty What is the best access option if a fistula cannot be made or fails? Does having a second fistula put in the same place as first fistula have any negative effects? Is there a better way that can replace the current fistula so it would not have to be replaced? When a person has had two permanent catheters and has used up all of their arms with failed fistulas and grafts, what options are left? How many fistulas can a person have before access is no longer available? What will happen when all access options fail?

Summary question: After fistula failure, what is the best access option?

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TABLE 2. Example of form for vascular access ranking exercise Rank

Summary question

Frequency

1

What are the benefits and risks of grafts vs. fistulae vs. catheter?

35

2

Can we create new types of access?

14

3

11

4

How to dress and manage a dialysis line to prevent infections and other complications? What is the best way to needle a fistula?

5

How can we improve AVF survival?

10

10

Respondent type Health professional (17 + 1 duplicate), Patient (4 + 4 duplicates), Care provider (4 + 1 duplicate), Clinical guidelines (1 + 2 duplicates), Other (1) Patient (4), Health professional (4), Other (4 + 1 duplicate), Care provider (1) Patient (5), Health professional (4), Care provider (1), Clinical guidelines (1) Health professional (4), Patient (4), Other (1), Clinical guidelines (1) Health professional (3), Clinical guidelines (1 + 6 duplicates)

TABLE 3. Top 10 research uncertainties 1. What is the best way to enhance communication between health care professionals and patients and to maximize patient participation in decision-making with regard to the advantages and disadvantages of different forms of dialysis, and access to test results to facilitate self-management? 2. How do different dialysis modalities compare in terms of their impact on quality of life, mortality and patient acceptability, and are there specific patient factors that make one modality better for some patients with kidney failure than others? 3. What are the causes and effective treatment(s) of, and ways to prevent, itching in dialysis patients? 4. What is the best strategy to increase kidney transplantation; including access to transplantation, increasing the efficiency of the recipient workup, and increasing the availability of donor kidneys? 5. What is the psychological and social impact of kidney failure on patients, their family, and other caregivers, and can this be reduced? 6. What are the best ways to promote heart health in dialysis patients, including management of blood pressure? 7. For people with kidney failure, what is the impact of each of the dietary restrictions (sodium, potassium, phosphate) separately, and when taken in combination, on important outcomes including quality of life? 8. What are the best ways to manage symptoms in people on or nearing dialysis including poor energy, nausea, cramping, and restless legs? 9. What are the causes and effective treatment(s) of depression in dialysis patients? 10. What is the best vascular access (among both new and existing types of access) for people on hemodialysis?

Future Challenges and Directions Priority setting is a process that can be used to produce a robust set of research questions that researchers can address over the coming years. As nephrology, of all the internal medicine specialties, has the fewest clinical trials to guide management (19), identifying top research priorities may aid funders in ensuring that the most important research questions can be prioritized within funding competitions. While the JLA method has many strengths, as illustrated by the recent priority setting process in people on or nearing dialysis, it remains a challenge to determine the priorities of all groups, particularly vulnerable groups. Furthermore, anecdotally, it would appear that the uncertainties identified by patients and researchers differ. For example, in the survey, six of the ranked top ten uncertainties identified by patients related to symptoms that are difficult to manage (i.e., itching, cramping, restless legs, poor energy); whereas only two of the ranked top ten uncertainties identified by physicians were related to symptoms. There is increasing emphasis on patient-centered care and the importance of including patients and their caregivers in establishing research priorities that inform clinical care is clear (6,8,20). Incorporating research priorities identified from all those involved in the disease—patients, caregivers and clinicians—can increase the relevance of the research undertaken and the potential it is adapted into practice. As determining research priorities through

a partnership with patients gain interest from potential funders, it is likely that this process will increase over time, benefitting both researchers and those affected by the disease. Future work needs to include determining the impact of these research priorities on the research agenda. References 1. Jung H, Wensing M, Grol R: What makes a good general practitioner: do patients and doctors have different views? Br J Gen Pract 47:805–809, 1997 2. Chalmers I, Bracken MB, Djulbegovic B, Garattini S, Grant J, Gulmezoglu AM, Howells DW, Ioannidis JP, Oliver S: How to increase value and reduce waste when research priorities are set. Lancet 383:156–165, 2014 3. Institute of Medicine: Crossing the Quality Chasm: A new Health System for the 21st Century. Committee on Quality Health Care in America. Washington, DC: Institute of Medicine, 2001 4. Brett J, Staniszewska S, Mockford C, Herron-Marx S, Hughes J, Tysall C, Suleman R: Mapping the impact of patient and public involvement on health and social care research: a systematic review. Health Expect 17(5):637–650, 2012 5. National Institute for Health Research: INVOLVE. Available at: http://www.invo.org.uk/, accessed November 13, 2014 6. Lloyd K, White J, Chalmers I: Schizophrenia: patients’ research priorities get funded. Nature 487:432, 2012 7. Canadian Institutes for Health Research: Strategy for patient-oriented research (SPOR). Available at: http://www.cihr-irsc.gc.ca/e/41204.html, accessed November 13, 2014 8. Patient-Centered Outcomes Research Institute: www.pcori.org, accessed November 17, 2014 9. Stewart RJ, Caird J, Oliver K, Oliver S: Patients’ and clinicians’ research priorities. Health Expect 14:439–448, 2011 10. Elberse JE, Pittens CA, de Cock Buning T, Broerse JE: Patient involvement in a scientific advisory process: setting the research agenda for medical products. Health Policy 107:231–242, 2012 11. Jaramillo A, Welch VA, Ueffing E, Gruen RL, Bragge P, Lyddiatt A, Tugwell P: Prevention and self-management interventions are top priorities for osteoarthritis systematic reviews. J Clin Epidemiol 66:503–510, e504, 2013

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12. Berry DL, Halpenny B, Hong F, Wolpin S, Lober WB, Russell KJ, Ellis WJ, Govindarajulu U, Bosco J, Davison BJ, Bennett G, Terris MK, Barsevick A, Lin DW, Yang CC, Swanson G: The Personal Patient Profile-Prostate decision support for men with localized prostate cancer: a multi-center randomized trial. Urol Oncol 31:1012–1021, 2013 13. Stanley MA, Calleo J, Bush AL, Wilson N, Snow AL, Kraus-Schuman C, Paukert AL, Petersen NJ, Brenes GA, Schulz PE, Williams SP, Kunik ME: The peaceful mind program: a pilot test of a cognitive-behavioral therapy-based intervention for anxious patients with dementia. Am J Geriatr Psychiatry 21:696–708, 2013 14. Gadsby R, Snow R, Daly A, Crowe S, Matyka K, Hall B, Petrie J: Setting research priorities for Type 1 diabetes. Diabet Med 29:1321– 1326, 2012 15. Pollock A, St. George B, Fenton M, Firkins L: Top ten research priorities relating to life after stroke. Lancet Neurol 11:209, 2012 16. National Institute for Health Research: NIHR Evaluation, Trials and Studies Coordinating Centre becomes the new home for the management of the JLA Priority Setting Partnerships. April 8, 2013. 17. Manns B, Hemmelgarn B, Lillie E, Dip SC, Cyr A, Gladish M, Large C, Silverman H, Toth B, Wolfs W, Laupacis A: Setting research priorities for patients on or nearing dialysis. Clin J Am Soc Nephrol 9:1813–1821, 2014 18. http://www.jlaguidebook.org, accessed November 17, 2014 19. Strippoli GF, Craig JC, Schena FP: The number, quality, and coverage of randomized controlled trials in nephrology. J Am Soc Nephrol 15:411–419, 2004 20. Tinetti ME, Basch E: Patients’ responsibility to participate in decision making and research. JAMA 309:2331–2332, 2013

Appendix 1. Example of taxonomy used for dialysis research uncertainties 1. Stage 4/5 CKD (renal-replacement therapy estimated within 6–12 months) • Treatment (preservation of kidney function) • Preparation for renal-replacement therapy & Modality selection (comparison across any dialysis subtypes) & Kidney Transplantation ○ Referral, eligibility and work up ○ Comparative effectiveness against dialysis & Timing of dialysis initiation/symptoms related to kidney failure • Prognosis • Other 2. Peritoneal dialysis • Dialysis prescription / adequacy • Catheter related issues • Peritonitis and exit site infection • Peritoneal dialysis technique failure • Other 3. Hemodialysis • Comparison across HD modalities (home, nocturnal, short daily etc) • Dialysis prescription / adequacy • Complications during dialysis ○ Hypotension ○ Muscle cramps ○ Headache • Other 4. Hemodialysis vascular access • Comparative effectiveness of different access options • AVF (fistula) /AVG (graft)

○ ○

Prevention of complications Treatment of complications • CVC (central venous cathether or “line”) ○ Prevention of complications ○ Treatment of complications • Needling • Other 5. Chronic complications related to kidney failure • Cardiovascular (HTN, CVD, CHF, fluid over load) • Diabetes – Type 1 and II: Prognosis and management • Anemia • Metabolic complications (bone disease, fractures, phosphate/calcium/PTH abnormalities) • Other 6. Diet • Dietary restriction • Impact of fluid management and restriction • Dietary supplements • Hyperkalemia • Other 7. Symptoms • Sleeping problems • Restless legs • Cramps • Itching • Nausea / vomiting • Fatigue • Pain • Sexual Dysfunction • Depression • General QOL • Other 8. Health system / health services • Effectiveness and Costs • Quality of care / standards • Service delivery • Optimal geographic location • Other 9. Communication across patients and/or providers • Other 10. Education of patients and families • Other 11. End of life • Other 12. Other • Travel • Lifestyle • Exercise • Psychosocial care • Pharmacologic management (general questions, and questions about compliance) • Vaccinations • Other • Work 13. Out of scope 14. Unclear uncertainty

Determining research priorities through partnership with patients: an overview.

There is an increasing level of emphasis being placed on health care providers and funders to incorporate patient-centered care into research. Involvi...
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