Development of a new model to engage patients and clinicians in setting research priorities

Abstract

Objectives

Equitable involvement of patients and clinicians in setting research and funding priorities is ethically desirable and can improve the quality, relevance and implementation of research. Survey methods used in previous priority setting projects to gather treatment uncertainties may not be sufficient to facilitate responses from patients and their lay carers for some health care topics. We aimed to develop a new model to engage patients and clinicians in setting research priorities relating to life after stroke, and to explore the use of this model within a James Lind Alliance (JLA) priority setting project.

Methods

We developed a model to facilitate involvement through targeted engagement and assisted involvement (FREE TEA model). We implemented both standard surveys and the FREE TEA model to gather research priorities (treatment uncertainties) from people affected by stroke living in Scotland. We explored and configured the number of treatment uncertainties elicited from different groups by the two approaches.

Results

We gathered 516 treatment uncertainties from stroke survivors, carers and health professionals. We achieved approximately equal numbers of contributions; 281 (54%) from stroke survivors/carers; 235 (46%) from health professionals. For stroke survivors and carers, 98 (35%) treatment uncertainties were elicited from the standard survey and 183 (65%) at FREE TEA face-to-face visits. This contrasted with the health professionals for whom 198 (84%) were elicited from the standard survey and only 37 (16%) from FREE TEA visits.

Conclusions

The FREE TEA model has implications for future priority setting projects and user-involvement relating to populations of people with complex health needs. Our results imply that reliance on standard surveys may result in poor and unrepresentative involvement of patients, thereby favouring the views of health professionals.

Keywords

consumers discipline: survey methods equity research priority setting stroke

Introduction

Ensuring effective and transparent priority setting for health research, which reflects the views of members of the public, is both ethically desirable and operationally beneficial.^1,2 There is growing evidence that involving patients, carers and health professionals in setting research and funding agendas can improve the quality, relevance and implementation of the research conducted^1–6 and ensure that research resources address the issues which are most important to those people affected by a particular health care problem.^7–9 This is becoming increasingly important for research that targets benefits for an aging population where multiple morbidity is common and increasingly complex interventions are likely to have an impact on multiple areas of health and well being. Priorities defined by patients, carers and health professionals would ensure that scarce research resources could be allocated to tackle those areas that matter most to patients and health care providers.

Despite widespread acknowledgement of the importance of patient involvement in priority setting, there is evidence that patients have not been enabled to make equitable contributions to research priority setting. Past research priorities have often been set by (or emerged from) the interests of researchers or industry and have largely excluded the views of patients and carers who have expertise and/or life experience.^10,11 Given the importance of increasing the extent, rigour, equity and meaningfulness of patient involvement in priority setting, it is vital that acceptable, efficient and transparent methods are adopted. Research suggests that such approaches are rare and methodologically challenging to undertake.¹²

The James Lind Alliance research priority setting process

The James Lind Alliance (JLA) has been established specifically to help achieve meaningful patient involvement in research priority setting. It is a non-profit making initiative aiming to ensure that those who conduct and fund health research are aware of what gaps in knowledge matter most to patients, carers and clinicians. This aim is achieved by forming priority-setting partnerships, bringing patients, clinicians and their representative groups together within equal partnerships which identify and prioritize unanswered questions (‘treatment uncertainties’).¹⁰ The JLA defines a treatment uncertainty as a question about the effects of a health care intervention for which there is no up-to-date, reliable systematic review of research evidence or for which a review confirms uncertainty exists. In practice, treatment uncertainty can be any unanswered question about the effects of any intervention on any outcome.

The JLA process involves eliciting the treatment uncertainties of patient, carers and clinicians, checking the submitted uncertainties, an interim prioritization stage and a meeting to agree the top 10 research priorities (Figure 1). The aim is to achieve equitable participation and contributions from representative groups of patients, carers and clinicians at all stages. The methods allow for a certain amount of flexibility, in response to the individual needs and goals of the partner organizations within the priority-setting partnership.¹³ Despite this flexibility, JLA methods strive to be methodologically defensible, and aim to ensure transparency, accountability and fairness. The JLA philosophy is pragmatic, with recognition that JLA methods are developing rather than established, and reflection on and refinement of the methods occurs as an essential component.

Figure 1.

James Lind Alliance – process diagram.

In order to maximize the likely impact of users’ views on decision making and priority setting, it is imperative that all stages of the JLA process are ethically credible and enable equitable participation by members. It is important that the process of identification of treatment uncertainties is comprehensive, and failure to achieve equitable contributions risks creating an incomplete list of treatment uncertainties and consequently, final priorities which may be biased and lack ethical credibility.

The JLA encourages its priority-setting partnerships to gather their uncertainties from as wide a range of sources as possible and to ensure patients are as confident and empowered as clinicians to submit the questions about their treatment to which they have not be able to find answers.¹³ Previous JLA methods for eliciting treatment uncertainties have primarily involved stakeholder meetings^14,15 and/or surveys, both electronic (email and internet) and paper-based.^16,17 These methods of eliciting treatment uncertainties can, by their nature, create a number of barriers to accessibility, engagement, ability to participate and representativeness. For example, to take part in the surveys, people must receive and be able to read and comprehend written instructions and be capable of writing or typing their treatment uncertainties. To be able to attend and contribute at a stakeholder meeting, potential participants have to be aware of and understand the purpose of the meeting, have available time and resources to attend and be able to travel to and access the meeting venue. Once at the venue they need the ability and confidence to understand and contribute. The extent to which these barriers erode the comprehensiveness and credibility of the process of elicitation of treatment uncertainties is very likely to be related to the nature of the health care condition (including the demographics of those affected by the condition, the impairments and activity limitations caused by the condition) being investigated.

Facilitating equitable elicitation of treatment uncertainties from people affected by stroke

In March 2010, we established a priority-setting partnership (PSP) to determine research priorities relating to life after stroke. Stroke survivors can have a wide range of impairments affecting mobility, communication, vision, cognition, perception, fatigue, continence and concentration.^18,19 Furthermore, stroke survivors and their carers often lack confidence and experience social isolation, further limiting their participation in activities and their quality of life.^20–22 Consequently, we considered that existing JLA methods were unlikely to result in equitable elicitation of treatment uncertainties from stroke survivors, carers and clinicians.

We aimed, therefore, to develop a new model to engage patients and clinicians in setting research priorities, and to explore the use of this model in a priority setting project relating to life after stroke.

Methods

Population of interest

Our population of interest was people living in Scotland who were affected by stroke, including stroke survivors, carers and health professionals. Our decision to focus on the research priorities of people living in Scotland was based on members of our steering group having well established and effective networks of communication across Scotland, such that we were confident of obtaining a geographically representative response rate. Limiting our focus in this way reduced the risk of selection bias within our population. Furthermore, as our new model involved face-to-face contact with stroke survivors and health professionals, applying a geographical limitation was pragmatic, enabling practical and feasible implementation. We were aiming to identify research priorities which would be generalizable to the wider population of people affected by stroke living in high income countries.

Development of a new model to engage patients and clinicians

We designed novel methods (Figures 2 and 3) to augment the standard JLA method of mailed and electronic surveys (Standard Survey Model). We called this the FREE TEA model (Facilitating Representative and EquitablE involvement through Targeted Engagement and Assisted involvement). The acronym was selected to convey the ethos of the model, which primarily involved meetings at which the key focus was often having a ‘cup of tea and a chat’.

Figure 2.

Standard Survey Model augmented with FREE TEA model to gather treatment uncertainties. FREE TEA: Facilitating Representative and EquitablE involvement through Targeted Engagement and Assisted involvement.

Figure 3.

Key strategies for targeted engagement and assisted involvement, within the FREE TEA model. FREE TEA: Facilitating Representative and EquitablE involvement through Targeted Engagement and Assisted involvement.

Implementation of Standard Survey Model

Questionnaires were emailed to a representative sample of stroke survivor, carer and health care professionals and organizations. The latter were asked to cascade the questionnaires to their members. Individuals and organizations were identified by our JLA PSP steering group members as including all health care professionals and stroke-related groups, organizations and charities involved in life after stroke across Scotland (see Supplementary material on web for full list of organizations). Questionnaires were mailed, with reply-paid return envelopes, to individual stroke survivors, and placed on the JLA website and on a project website. Responses could be returned via mail or email, and a telephone number (with answer-phone) was provided for verbal responses.

Implementation of FREE TEA model

Targeted engagement

In order to target the engagement and facilitate submission of treatment uncertainties, we aimed to achieve face-to-face contact with representative groups from across Scotland. We targeted engagement with stroke survivors and carers via the network of stroke support groups, primarily supported by The Stroke Association, Chest Heart and Stroke Scotland and Different Strokes. We contacted, by telephone and using personal contacts from members of our steering group, stroke clubs and groups across Scotland and asked if we could visit their club/group. We aimed to visit at least two clubs/groups from every mainland area in NHS Scotland. In addition, we recruited volunteers to distribute materials at at-least one relevant club/group from each of the three island areas. All visits were arranged at a time and place convenient to the group members and generally (but not exclusively) occurred during a scheduled group meeting. During the visit (which was by a member of the PSP steering group) a standard presentation describing the project was delivered and the group members were invited to submit their treatment uncertainties. Group members chose whether to submit their individual uncertainties, or whether to discuss and submit on behalf of the group. The PSP steering group member assisted with this process as was required; particularly for patients with reading, writing and communication difficulties. In total, we attended 20 stroke groups and clubs from the 11 mainland NHS Scotland areas.

There are a range of well-attended stroke-specific meetings for health professionals across Scotland which we were able to attend. Activities which we carried out varied according to the format and agenda of these meetings. At a minimum we displayed information about the project, distributed information leaflets and copies of the questionnaire, and were available to discuss and answer questions. We attended seven stroke-specific national conferences and meetings.

Assisted involvement

In order to address barriers to participation, it was necessary to ensure that we created, and made easily accessible, materials which enabled involvement regardless of stroke-related impairments. It was also important that participants were able to submit treatment uncertainties in a format suitable to their individual needs and abilities. In order to achieve this, the steering group developed a standard presentation describing the project and requesting submission of treatment uncertainties, and adapted this into a number of different formats. These included a power point presentation, an on-line presentation with recorded narration and a table-top presentation. When we mailed questionnaires, links to the on-line presentation were provided to facilitate involvement of people with impaired ability to understand the written information. During our visits to stroke survivor groups/clubs the standard presentation was delivered using the most appropriate mode for the venue and group.

Aphasia is a common impairment after stroke, so we worked with speech and language therapists to develop an aphasia-friendly information sheet, which was available on request and – when visiting stroke clubs – offered to people known to have aphasia.

We accepted submissions of treatment uncertainties in any format including written on our questionnaire, written on a piece of paper, within email text or by telephone. We included postal, email and telephone contact details in all our correspondence.

Comparison of responses to Standard Survey and FREE TEA models

We did not plan to formally evaluate and compare the Standard Survey and FREE TEA models. However, we did collect basic data which have enabled us to make comparisons between the number of treatment uncertainties which were submitted.

Results

We gathered 516 treatment uncertainties (Figure 4). Approximately equal numbers came from stroke survivors and carers (54%) and health professionals (46%). However, only 35% of those from stroke survivors and carers were elicited from the Standard Survey Model, in contrast to 84% of the submissions from health professionals. Thus, if we had not used FREE TEA, only one-third of the responses would have been from stroke survivors/carers and two-thirds from health professionals.

Figure 4.

Comparison of number of treatment uncertainties submitted in response to the Standard Survey and FREE TEA models. FREE TEA: Facilitating Representative and EquitablE involvement through Targeted Engagement and Assisted involvement.

Our use of the Standard Survey differed for the populations of stroke survivors and health professionals and, while we only used email and electronic communication to reach health professionals, we mailed information to a large number of stroke survivors. From 200 questionnaires mailed to stroke survivors, only 22 responded (11%). This confirms the difficulty we had anticipated in involving this group of patients using only the Standard Survey.

Discussion

Main findings

We developed and implemented the FREE TEA model, successfully facilitating involvement of equal numbers of stroke survivors and health professionals in our PSP. Our successful application of this model has implications for future PSPs and user-involvement relating to populations of people with complex health needs. These results imply that a reliance on standard surveys may result in poor and unrepresentative involvement of patients, thereby favouring the views of health professionals. In order to facilitate representative and equitable involvement of people affected by complex health care conditions, we recommend a pragmatic application of novel models of engagement, and propose that our FREE TEA model may be appropriate. The additional cost of the visits to patient groups was approximately £35 travel plus half a day of research assistant time per visit. As we visited groups during one of their scheduled meetings we did not incur any additional costs for patient travel, venue hire or refreshments. Future studies should consider including a full cost-analysis of the resources required for different models of engagement. A pragmatic approach to using this model will be required and adaptations would be necessary according to the specific barriers facing the particular population.

Limitations

We have assumed that submissions by mail, email or telephone occurred in response to the Standard Survey. However, due to the process of information cascade, where people contacted were encouraged to pass on project information to any other relevant people and populations, it was not possible to be certain whether mail, email or telephone submissions occurred as a result of the Standard Survey or FREE TEA. It is therefore possible that a number of our emailed and mailed responses attributed to the Standard Survey were in fact prompted by FREE TEA visits. However, this would serve to underestimate the effect of FREE TEA meaning that the reported responses are underestimates.

Our methods were not successful in eliciting responses from carers; only 4% of the responses were from carers. A small number of participants at the stroke groups were carers, however carer involvement in our activities at the stroke groups was not common. Informal carers play an essential role²³ and their treatment uncertainties should be included in priority setting. Additional methods to facilitate the involvement of carers are required which might, for example, involve targeted engagement and assisted involvement of members of local support organizations for carers.

In comparing the responses from these two models we have explored the number of treatment uncertainties submitted by stroke survivors and health professionals. While achieving equality in the numbers of responses is a positive outcome, equality in the numbers of responses is not necessarily a measure of representative and equitable involvement. We did not gather sufficient information to enable us to explore whether we achieved truly representative and equitable involvement from different populations. Our methods of targeting stroke survivors who attended existing stroke clubs and groups may have resulted in a sample of stroke survivors which was not representative of the whole population of stroke survivors and from which certain groups of people may have been excluded. We recommend that future research prioritization projects gather basic data on demographic characteristics of respondents.

Conclusions

We achieved similar numbers of submissions of treatment uncertainties from stroke survivors and health professionals by augmenting standard survey methods with our novel FREE TEA model. It facilitated involvement of stroke survivors and, without these activities, we would have gathered substantially fewer responses from stroke survivors than from health professionals. The involvement of carers may be difficult to achieve and we propose that additional methods are required to ensure their involvement. The treatment uncertainties gathered have been used to identify the top 10 shared research priorities relating to life after stroke, following the JLA methods of prioritization.^24,25 This was achieved with participation from similar numbers of stroke survivors and health professionals at all stages of the process, creating shared priorities which we believe have ethical credibility, and reflect what matters most to those affected by stroke.

Footnotes

Acknowledgements

We would like to thank Kathleen Frew (Chest Heart and Stroke Scotland), Angela MacLeod (Stroke Association), Fiona Brodie and Katrina Brennan for their contribution to the JLA life after stroke PSP steering group.

Funding

This work was supported by funding from the Scottish Government’s National Advisory Committee for Stroke. The Nursing, Midwifery and Allied Health Professions Research Unit (NMAHP RU) is funded by the Scottish Government Health Directorate’s Chief Scientist Office.

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