CBT and GET are often compared against usual care provided by health care practitioners (e.g. family doctors) or pacing, self-pacing used by sufferers to manage their physical activity or pacing therapy (PT) guided by a therapist. However, vigorous debate has emerged concerning the appropriateness and efficacy of these treatment approaches. The PACE trial is a randomised controlled trial (RCT) that compared CBT, GET and PT, against standard care ( White et al., 2011 ) with a 59–61 per cent improvement rate and a 22 per cent recovery rate following CBT-GET ( White et al., 2013 ). However, a 2-year follow-up study from PACE revealed that between-group differences fell away as the standard medical care and PT groups showed a similar level of improvement ( Sharpe et al., 2015 ). Recent reanalyses of data from the PACE trial suggest that the actual improvement and recovery rates are much lower than first reported, improvement rates fell from 60 per cent to circa 20 per cent and recovery rates fell from 22 to 4 per cent for GET and 7 per cent for CBT, with 3 per cent for PT ( Wilshire et al., 2016 ). A Cochrane Review of CBT for the treatment of ME/CFS found some benefit for a small portion of sufferers with mild-to-moderate severity, but lack of evidence of long-term benefit and inconsistent evidence that CBT has a positive impact on physical function in the disease ( Price et al., 2008 ). The US Agency for Health Care Research and Quality (AHRQ) recently downgraded their rating of CBT and GET for ME/CFS following a review that showed that the effectiveness varied according to the diagnostic criteria used in RCTs ( Smith et al., 2016 ). In the United Kingdom, the National Institute for Health and Care Excellence (NICE; 2007 ) recommends CBT and GET as evidence-based treatments for ME/CFS. In contrast, patient surveys suggest that these treatments lead to negative outcomes for a large percentage of patients. The ME Association (2015) in the United Kingdom, a leading UK ME/CFS charity, conducted a large patient survey to gauge sufferers’ experiences and views of the three main interventions, CBT, GET and PT. This article presents an independent analysis of this large patient data set and compares findings against similar previously published surveys (2000–2015).

Epidemiological estimates suggest around 1 million people in the United States ( Green et al., 2015 ) and around 250,000 in the United Kingdom ( Department of Health, 2002 ) suffer from myalgic encephalomyelitis (ME)/chronic fatigue syndrome (CFS). The World Health Organization (WHO) classifies ME, sometimes differentiated from CFS, as a neurological disease ( WHO, 2007 ). The abbreviation ‘ME/CFS’ is often used in the literature to denote the illness, taking account of the controversies surrounding the aetiology and pathogenesis. Despite a relatively large number of sufferers, the disease remains poorly understood. Doctors and scientists have yet to agree on a cause; thus, various treatments have been applied experimentally without reference to a universally accepted model of disease pathogenesis. In 2015, the US Institute of Medicine conducted an extensive review of scientific evidence and suggested renaming ME/CFS ‘Systemic Exertion Intolerance Disease’ to better reflect the profound physical disabilities most sufferers endure, including prolonged relapses and symptom exacerbation after minimal exertion ( Institute of Medicine, (IOM), 2015 ). The illness has a major negative impact on the quality of life, economic and social status ( Drachler Mde et al., 2009 ; Jason et al., 2008 ).

A survey was opened for 4 months and nationally advertised for anyone with ME/CFS to participate in the study (not only members of the ME Association). Respondents were asked a series of 228 questions and subquestions regarding treatment, particularly CBT, GET or PT. The survey had 1428 respondents. Respondents were required to confirm an affirmative diagnosis of ME/CFS from a qualified medical professional. Respondents also had to confirm that they had taken part in either one-to-one or group course therapy (CBT, GET and PT). Respondents had to indicate the severity of their illness and symptoms before and after their treatment course. We used these data to explore the effect that delivered treatment courses (CBT, GET and PT) had on patients’ reported changes in symptoms and illness severity. Of the 1428 respondents, only 954 indicated that they had a confirmed medical diagnosis of ME/CFS, had participated in at least one of the three courses of treatment and completed questions on their symptoms and illness severity status, before and after treatment (our main analysis cohort). Using ordered logistic regression, we first model the direct effect of course approach on change in symptoms (post-course); second, we consider the effects of course composition after adjusting for demographic, condition-specific and course-specific variables. We also comment on the effect that these variables have on change in symptoms (see Appendix 1 for details of our methods). We compare our results against those of other comparable patient surveys.

We conducted a search of common scientific literature databases (PubMed and others) and patient organisation websites (grey literature) to identify similar published patient surveys to compare our primary results with previous ME/CFS patient surveys (2000–2015) that offered data on patients’ symptom profile following CBT, GET and PT. Our search revealed more than 15 relevant surveys. We excluded 5 surveys due to lack of clarity regarding treatment approach, leaving 10 included comparable surveys. Table 4 summarises results from crude analysis of these secondary surveys. We present aggregate scores concerning whether or not interventions improved symptoms, had no change or worsen/deteriorate symptoms. CBT brought about improvement in symptoms for approximately 35 per cent of respondents (65% unchanged/worse). In total, 25 per cent of GET reported improvement in symptoms (17% unchanged/54% worse), while 82 per cent reported benefit following PT with only 4 per cent deterioration. These findings are better than our primary survey findings for CBT/GET benefit (1/10 vs 3/10), but similarly show a pattern that GET brings about a worsening in symptom experience for at least 5 out of every 10 patients, with PT benefiting far more patients by a large margin (8/10).

The strongest independent association with change in symptoms was with appropriateness of the course ( χ ( 2 ) 2 = 164 . 9 ; p < 0.001); patients who rated the course ‘not appropriate’ (or only partially so) reported a significant worsening of symptoms post-therapy than patients who thought the course wholly appropriate. Course attendance was also associated with symptom change ( χ ( 4 ) 2 = 17 . 9 ; p = 0.001); patients who did not attend the full course reported a significant worsening of symptoms compared with patients who attended the full course. This was particularly true if attending less than half the course. ‘Course Dynamic’ was not associated with change in symptoms ( p = 0.711). Age at ME onset was marginally associated with change in symptoms ( χ ( 5 ) 2 = 11 . 6 ; p = 0.041); compared with 35–44 year olds, all other age groups reported some improvement in their symptoms post-therapy (apart from patients aged 12 years and under), with teenagers reporting the greatest improvement. Men reported a significantly better improvement in their symptoms post-therapy than women ( p = 0.018). Both duration of ME (significantly: χ ( 4 ) 2 = 13 . 7 ; p = 0.008) and therapist’s beliefs (weakly: χ ( 3 ) 2 = 6 . 6 ; p = 0.086) were associated with reported changes in symptoms, but not the health professional who gave the diagnosis. In the former case, compared to patients who had had ME for less than 12 months, patients who had had ME for more than 12 months reported a significant worsening of symptoms post-therapy (especially patients who had had ME for >2 years). The belief that ME was psychological (vs physical) resulted in some reported worsening of symptoms post-therapy (but not for mixed beliefs).

Without controlling for any covariates, our model shows ( Table 3 ) that a greater percentage of CBT in the course has a non-significant negative effect on symptom change post-course (β CBT = –0.0049; 95% CI (–0.0795, 0.0697); p = 0.897). A greater percentage of GET has a significant negative effect on symptom change – patients report a worsening of symptoms post-therapy (β GET = –0.3226; 95% CI (–0.3932, –0.2519); p < 0.001), while a greater percentage of PT has a significant positive effect on symptom change – patients report an improvement in symptoms post-therapy (β PT = 0.2190; 95% CI (0.1503, 0.2877); p < 0.001). These conclusions were unchanged after controlling for the demographic, condition-related and course-related variables ( Table 3 ), although the strength of association between symptom change and the percentage of GET or PT in the course substantially weakened (β GET = –0.2247; β PT = 0.1179), while the association between symptoms and percentage of CBT became positive (β CBT = 0.0591).

Irrespective of composition, on average 64 per cent of patients in CBT reported symptoms remaining the same (with those remaining the same or worse totally at 88%). Where CBT was the main content, 91 per cent of patients reported no positive change in symptoms (88% using any treatment combination). The only real deviation from this figure was when both elements of GET and PT were present alongside the main CBT content; approximately two-thirds reported no change (65.0%) and approximately equal numbers reported worse symptoms (19.1%) or an improvement (15.9%). Where GET was the main content or included in combination with any other treatment course (CBT or PT), patients tended to state that their symptoms worsened more frequently than improved. In contrast, the addition of elements of PT improved outcomes, relative to the corresponding courses in which it was not present. These observations were formally tested using ordered logistic regression. Table 2 depicts the distribution of the demographic, condition-specific and course-specific independent variables included in the model.

Table 1 depicts ‘change in degree of severity of symptoms’ by ‘course composition’. A small number of respondents are unused here ( n = 21 from 954) due to ambiguous answers to some questions regarding treatment overlap. A total of 302 patients (32%) reported worsening of their symptoms post-therapy, 455 (49%) reported that they stayed the same and in 176 (19%) the symptoms showed improvement. When considering the main course content (ignoring other elements), Pacing Therapy (PT) brought about a greater improvement rate in symptoms (44%), versus just 8% in CBT and 12%. GET recorded the largest negative response (74% of patients) versus 18 per cent in CBT and 14 per cent in PT.

In our survey, 72 per cent of respondents reported having received a positive diagnosis of ME/CFS by a specialist, 22 per cent from a general practitioner (family doctor) and 4.5 per cent from other professionals; 17 per cent reported that their ME/CFS symptoms started prior to age 18 and the average age of ME/CFS onset fell around 35 years. This closely resembles the epidemiological evidence which suggests an average age at onset of 33 years, with ME/CFS age distribution ranging from less than 10 years to 70 years and older ( IOM, 2015 ). Klonoff (1992) reported average illness duration to be 4.4 years. In our survey, the average illness duration was close to 6 years with 6 per cent of respondents having the illness for less than 2 years (12%, 3–4 years; 33%, 5–10 years; 31%, 11–20 years; 18%, 21+ years), suggesting a bias towards more long-term sufferers. Most respondents were women (79%). This finding is consistent with other studies that found female-to-male ratio of between 3:1 and 6:1 ( Capelli et al., 2010 ; Faro et al., 2016 ). Prognostic data on recovery in ME/CFS suggest a recovery rate of between 5 and 31 per cent ( Cairns, 2005 ). Recovered sufferers are unlikely to be captured by a survey of this kind. The demographics of our survey sample appear representative of the general ME/CFS population ( Nacul et al., 2011 ).

Discussion

CBT is offered to patients based on a model of dysfunctional illness beliefs (Deary et al., 2007; Sharpe, 2010) and rests on a theory that ME/CFS is perpetuated by such factors (Moss-Morris et al., 2003). In our primary survey, patients were split on the appropriateness of CBT, with over half finding it inappropriate (46% appropriate/partly appropriate) to their needs. Findings from our patient survey and secondary survey analysis show that CBT has little impact on symptom improvement for approximately 70–90 per cent of patients, raising questions about the utility of the CBT model of ME/CFS. For those who benefit from CBT, CBT may be an adjunct therapy that helps ME/CFS patients deal with the emotional distress of illness, the anxiety generated by suffering troubling physical symptoms, and the secondary depression that is associated with most chronic illnesses (Harris, 2012). Our data indicate that CBT therapists who regard ME/CFS as a physical illness are more likely to have a better chance of helping patients improve symptoms and relieve distress, compared to those who view the illness as psychological.

GET fails to help the majority of ME/CFS patients improve symptoms and has a marked negative impact on approximately 50 per cent of patients. GET also had a marked negative impact on perceived degree of illness severity, particularly for those with severe to very severe presentations, with 21 per cent more patients reporting being more severely afflicted after GET. Not surprisingly, 78 per cent of patients in our survey reported GET not to be appropriate to their needs. The beliefs of GET therapists have an effect on outcomes, with 80 per cent of patients reporting no benefit if the therapist believed ME/CFS to be a psychological illness. This evidence contrasts with RCTs that report benefits using GET in CFS (Moss-Morris et al., 2005; White et al., 2011). The high rate of adverse symptom reactions to GET observed in our survey and secondary survey analysis may well be connected to the advice of therapists to continue exercising even if symptoms worsen, with pushing ‘beyond limits’ a key feature of GET (Bavinton et al., 2004). While physiological deconditioning is important to combat in chronic illness, alternative physical rehabilitative therapies may need to be developed and tested that take account of ME/CFS-specific features, including orthostatic intolerance (Frith et al., 2014) and symptom flare post-exertion (Twisk and Geraghty, 2015).

Pacing alone brought about the greatest positive impact on symptom experience with 44 per cent of patients reporting improvement, compared with 8 per cent in CBT and 12 per cent in GET treatment. Following PT, 14 per cent reported worsening symptoms, compared with 18 per cent in CBT and 74 per cent in GET, making pacing the least negative treatment approach. Much more detailed empirical research is needed to qualify these findings. PT is overwhelmingly favoured by patients (84% finding it appropriate/partly appropriate) and has a moderate impact on reducing the degree of illness severity. Secondary surveys show that 82 per cent of patients report improvement with PT, compared with 35 per cent in CBT or 28 per cent in GET. The beneficial outcomes of pacing were strongly correlated with the beliefs of the therapists, with 53 per cent of patients reporting benefit if the therapist believed ME/CFS to be a physical illness, compared to just 5 per cent if the therapist believed ME/CFS to be psychological. Unlike CBT and GET, that are interventions to emerge from a theoretical model of ME/CFS (Deary et al., 2007; Surawy et al., 1995), pacing is less of a formal therapy and more of a personal approach to energy management practised by ME/CFS sufferers. The benefit of PT may relate to the way in which it allows a sufferer to adapt to the illness and work within limits, while testing boundaries. This approach is less invasive than CBT or GET programmes and may be more appropriate for the most severely afflicted. It is worthwhile remembering that most RCTs of CBT/GET only recruit participants well enough to attend clinics (mild to moderate cases).

Benchmarking key findings Findings from our primary and secondary survey analysis conflict with numerous RCTs that report CBT and GET to be superior and safe treatments for ME/CFS (e.g. PACE trial). However, the PACE trial has attracted much criticism (Kindlon, 2011b). Reanalysis of data from PACE reveals alterations to methods that make CBT and GET appear more beneficial than would have been the case if the original protocol had been adhered to (Goldin, 2016). Detractors point to the way in which recovery was operationally defined as not requiring return to normal, or near normal, levels of physical activity (Geraghty, 2017; Wilshire et al., 2016). A Cochrane review of CBT in CFS found that 40 per cent of CFS patients report a reduction in self-rated fatigue following CBT, with 26 per cent improving in usual care, a differential of only 14 per cent added benefit of CBT above standard care using subjective measures. Price et al. (2008) concluded that the benefits of CBT are not sustained over the long term and that there is little evidence of improvements in physical function following CBT. Moss-Morris et al. (2005) observed a decrease in self-rated fatigue in CFS using GET, but Wiborg et al. (2010) observed that reduced fatigue did not correlate with an increase in physical activity measured objectively with actometers. Other RCTs have found no substantive benefits using CBT or GET (Núñez et al., 2011; Wearden et al., 2010). A Cochrane review of non-pharmacological interventions for functional syndromes, including CFS, noted multiple methodological concerns in psychotherapy trials, including high drop-out rates and selective biases in sampling (Van Dessel et al., 2014). ME/CFS is difficult to diagnose and it is speculated that clinical trials often involve volunteer bias and include patients with psychiatric illnesses and milder cases. These patients may respond better to CBT or GET than patients with more moderate-to-severe cases that are captured by surveys.