Consulting patients in setting priorities in Myalgic Encephalomyelitis (M.E.) research: findings from a national on-line survey

The importance of involving patients in the development and monitoring of health services and in the conduct of health services research is widely recognised throughout the developed world [1–6]. The UK government’s commitment to patient involvement is evident through the publication of documents such as Our Health, Our Care, Our Say [7] and Equality and Excellence: Liberating the NHS [8]. Similarly, the National Institute for Health Research (NIHR) actively encourages patient involvement in NHS, public health and social care research through its advisory group INVOLVE [9]. The advantages of involving patients in health services research are well documented [10–13], and guidelines have been produced recommending the inclusion of patients at every stage of the research process [14]. Involving patients in setting the research agenda is particularly important as their priorities may differ from current research practice, and are more likely to reflect the interests of the NHS, public health and social care services [15]. In keeping with the core democratic principles of active citizenship, accountability and transparency, the legitimacy and sustainability of investment decisions made by research funding bodies will be increasingly influenced by how well they reflect the underlying values of the general public [16].

Despite examples of good practice in areas such as COPD and asthma [17], cancer [18] and chronic kidney disease [19], consulting patients when determining research priorities remains a developing area of study [15]. Myalgic Encephalomyelitis (M.E.) is a complex multi-system disorder that affects an estimated 250,000 men, women and children in the UK [20]. Symptoms include post-exertional malaise, fatigue, sleep disturbance, widespread muscle and joint pain, and difficulties with memory and concentration [21]. However, each patient experiences their own personal combination of symptoms, posing significant challenges for healthcare professionals in relation to diagnosis and management [22]. Although there have been attempts to gather the views of those affected by M.E. regarding research that individuals would be willing to participant in [23, 24], to date, there have been no comprehensive attempts to involve patients in determining the strategic direction of M.E. research at a national level. Given the lack of understanding regarding the disease mechanisms underpinning M.E .and the uncertainties associated with diagnosis and management, it would seem even more critical that the views of individuals affected by M.E. are listened to and acted upon when determining future research priorities. Further complexity arises from differing nomenclature with the terms M.E. and chronic fatigue syndrome (CFS) sometimes being used interchangeably.

In response to the paucity of literature surrounding the research agendas of individuals with M.E. the UK-based charity Action for M.E. initiated the first national on-line survey to involve people affected by M.E. in identifying priorities for future research investment. This paper will present the key findings from this work.

This survey is unique in seeking the views of patients in determining the strategic direction of M.E. research at a national level. Contrary to common perceptions [15], we have demonstrated that individuals affected by M.E. are able to engage with a broad range of issues relating to science, medicine, and healthcare delivery and can identify and agree on research priorities. The top five research priorities identified by the respondents of this on-line survey were: disease processes to achieve a better understanding of the underlying pathology of M.E.; more effective treatments; faster and more accurate diagnosis; clinical course of M.E.; outcomes and prognosis; and severely affected patients. The least popular research priorities identified were: sleep; economic research towards identifying the cost of M.E. for individuals and society; and psychological aspects. The historical research effort in M.E. does not obviously match with the prioritisation seen in this survey from consumers, highlighting how in the past the research community’s priorities may not have aligned with those of the patients or consumers. To date, there have not been national research priorities and this current work will ensure that the patients’ priorities must now be considered.

A particular strength of this work was the inclusion of free text responses, which enabled individuals affected by M.E. to articulate their views and actively influence the strategic direction of future research funding in the UK. In keeping with the research priorities set out by a collaborative clinical sub-group [26], respondents emphasised the importance of investing in research to achieve a better understanding of the disease processes underpinning M.E. Many expressed concerns at the trivialisation of M.E. by health and social care professionals, and the labelling of patients with M.E. as suffering from a psychiatric, psychological or somatic symptom disorder. Respondents demonstrated awareness of, and support for, biomedical research into the pathogenesis of M.E. at a molecular or genetic level. It was suggested that the identification of protein biomarkers would provide a clear distinction between M.E. and CFS, facilitating accurate diagnosis and timely access to tailored interventions with a particular emphasis on drug development and curative therapies. A high proportion of respondents provided their personal contact details with a view to being involved in future research prioritising disease mechanisms, a diagnostic test and specific therapies.

Given the paucity of literature surrounding the research priorities of individuals affected by M.E., it was necessary to undertake an exploratory scoping exercise. However, this survey has a number of limitations. Despite the large number of responses and willingness to participate in future research, it is important to acknowledge that the individuals with M.E. completing this survey were a self-selecting group for which no demographic information was available. It is, therefore, not possible to say whether the views presented in this paper are representative of the 250,000 people currently estimated to be living with M.E. in the UK. The survey was only available on-line via Action for M.E.’s website, Facebook and Twitter pages. For this reason, certain groups, such as the severely affected or those with lower computer literacy levels, may have been excluded from providing their opinions. Although research into the pathophysiology of severely affected individuals was identified as one of the top five research priorities, it is not possible to say whether this patient group were adequately represented in this survey. Carers for someone with M.E. or individuals with a family member of colleague with M.E. were also under-represented, as were professionals with an interest in M.E. although there were no obvious differences in research priorities between patients, carers and family members. Only six healthcare professionals provided unstructured data to support their first, second and third research priorities, limiting the potential for meaningful comparison between groups.

Although still a developing field of study, healthcare organisations throughout the developed world are increasingly involving patients in service delivery and policy decisions, including guideline and quality indicator development, programme development and evaluation, quality improvement and funding priorities. The findings from the on-line survey have been used to inform and influence the statement of intent underpinning the largest national patient charity Action for M.E.’s Research Strategy 2014–2016 [27] which seeks to provide personalised medicine for patients with M.E. through the prioritisation of four key research areas: cause and prevention; diagnosis; targeted treatments; and severely affected individuals. The rise in the number of people living with chronic disease, such as M.E., highlights the importance of productive interactions between funding bodies, research charities, patients and healthcare professionals. In the field of M.E. over recent years the coming together of the five largest national patient charities together with researchers and clinicians to form the UK CFS/ME Research Collaborative has provided the framework to consider and provide focus to address the research priorities outlined in the survey. Future work should focus on promoting further transparency and clarity in the reporting of achievements against these set targets.

The study has some limitations. The participants are clearly a self selected group who are members of the largest national patient support charity for M.E. It is therefore important to consider their responses in the context of this self selection and recognise that the respondents are not randomly selected. It is also not possible to determine the absolute reach of the survey, as it was deployed via the social media outlets and website for the charity for a very defined period of time. It is therefore important that more rigorous research is performed to explore in more detail the research priorities for those with M.E. and to specifically establish service user led priorities. We would suggest that this survey with all the associated limitations of survey work has highlighted an area where those with M.E. feel that they wish to influence policy. This is underlined by not only the numbers of respondants but also by the richness of the free text included with the quantitative responses. We believe that this is a strong platform upon which to further enhance our understanding of the priorities of those affected by M.E.

This work illustrates the importance of involving individuals affected by M.E. in the identification of strategic priorities for future research funding. As the beneficiaries of discoveries and developments in health services research, it is particularly important that the views of patients are listened to and acted upon. We have demonstrated that individuals affected by M.E. have clear views regarding the important priorities for research investment. These tended to focus on disease processes to achieve a better understanding of the underlying pathology of M.E. and would appear to be closely aligned with those of the scientific research community.