Defining the prevalence and symptom burden of those with self-reported severe chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME): a two-phase community pilot study in the North East of England


Victoria Jane Strassheim, Madison Sunnquist, Leonard A Jason, Julia L Newton


Research and Innovation Hub/CRESTA Fatigue Clinic, Newcastle upon Tyne Hospitals NHS Foundation Trust; Institute of Ageing and Health, Newcastle University, Newcastle upon Tyne, UK; Center for Community Research, De Paul University, Chicago, Illinois, USA



To define the prevalence of severe chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) and its clinical characteristics in a geographically defined area of Northern England. To understand the feasibility of a community-based research study in the severely affected CFS/ME group.


A two-phase clinical cohort study to pilot a series of investigations in participants own homes.


Participants were community living from the area defined by the Northern clinical network of the UK.


Adults with either a medical or a self- reported diagnosis of CFS/ME. Phase 1 involved the creation of a database. Phase 2: five participants were selected from database, dependent on their proximity to Newcastle.


The De Paul fatigue questionnaire itemised symptoms of CFS/ME, the Barthel Functional Outcome Measure and demographic questions were collected via postal return. For phase 2, five participants were subsequently invited to participate in the pilot study.


483 questionnaire packs were requested, 63 were returned in various stages of completion. 56 De Paul fatigue questionnaires were returned: all but 12 met one of the CFS/ME criteria, but 12 or 22% of individuals did not fulfil the Fukuda nor the Clinical Canadian Criteria CFS/ ME diagnostic criteria but 6 of them indicated that their fatigue was related to other causes and they barely had any symptoms. The five pilot participants completed 60% of the planned visits.


Severely affected CFS/ME individuals are keen to participate in research, however, their symptom burden is great and quality of life is poor. These factors must be considered when planning research and methods of engaging with such a cohort.


Strassheim et al, BMJ Open, 2018 September 19; 8(9):e020775


We thank ME Research UK for funding the study that allowed this pilot project.

Comment by ME Research UK

We have reported before on the lack of information about those people with ME/CFS who are especially ill; that is, wheelchair-, house- or bed-bound because of the severity of their condition. This may represent a quarter of all people with ME/CFS, and they tend to have worse prospects of recovery and may be vulnerable to developing secondary medical conditions.

And yet this important subgroup is often neglected. While ME/CFS is already under-researched compared with other chronic conditions, these most severely ill patients are under-represented in what little research is done.

A large part of the problem is that their challenging circumstances mean these individuals have difficulty accessing medical care and engaging in medical research. Is there any way of improving this situation?

With funding from ME Research UK, Victoria Strassheim and colleagues at Newcastle University have been conducting an ongoing programme of research concentrating on severely affected ME/CFS patients. Victoria has published three articles over the last couple of years, including a review of existing research on severe ME and an exploration of the effects of deconditioning in these patients.

A third paper was recently published in the journal BMJ Open and, among other objectives, looks specifically at how to include severely affected ME/CFS patients in research.

The first part of the project was to attempt to contact and evaluate patients with severe ME/CFS within the Northern England Clinical Network, which very roughly extends from the Scottish border down to the northern halves of the Lake District, Yorkshire Dales and North York Moors National Parks.

The participants were adults with a medical or self-reported diagnosis of ME/CFS who were wheelchair-, house- or bed-bound, and they were recruited by advertising in GP practices, local ME/CFS services, and national and local charities, and on social media.

A total of 483 questionnaire packs were sent out to those people identified, including the Barthel Functional Outcome Measure, which assesses performance in ten activities of daily life, and the De Paul fatigue questionnaire, which assesses ME/CFS symptoms.

Unfortunately, only 63 packs were returned, although 76% to 88% of participants managed to complete the questionnaires successfully. The responses provided a host of information on the burden of symptoms and functional difficulties they have to live with, and the findings are freely available to download from the BMJ Open website.

The second part of the project involved making a series of home visits to five severely ill ME/CFS patients in the Newcastle area, and attempting to complete assessments that had formed part of a previous study conducted in people with mild or moderate ME/CFS.

Over the course of four visits, a number of activities were attempted, including an ECG assessment, an active stand test and other physical tests, various respiratory tests, cognitive assessments and several questionnaires.

Two patients were able to complete all of the assessments, while the other three achieved around 50% and were unable to perform the other tests, refused the activities or could not attend due to ill health.

The investigators conclude that people severely affected by ME/CFS can engage with research, but they have a considerable burden of symptoms and a poor quality of life, and they need more support during the research process. The use of ‘research advocates’ is suggested, to help engage and recruit these individuals into clinical studies.

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