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Research Article

Adaptive Pacing, Cognitive Behaviour Therapy, Graded Exercise, and Specialist Medical Care for Chronic Fatigue Syndrome: A Cost-Effectiveness Analysis

  • Paul McCrone mail,

    paul.mccrone@kcl.ac.uk

    Affiliation: Centre for the Economics of Mental and Physical Health, Health Service and Population Research Department, Institute of Psychiatry, King’s College London, London, United Kingdom

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  • Michael Sharpe,

    Affiliation: University Department of Psychiatry, University of Oxford, Oxford, United Kingdom

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  • Trudie Chalder,

    Affiliation: Academic Department of Psychological Medicine, King’s College London, London, United Kingdom

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  • Martin Knapp,

    Affiliations: Centre for the Economics of Mental and Physical Health, Health Service and Population Research Department, Institute of Psychiatry, King’s College London, London, United Kingdom, Personal Social Services Research Unit, London School of Economics, London, United Kingdom

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  • Anthony L. Johnson,

    Affiliations: MRC Biostatistics Unit, Institute of Public Health, Cambridge, United Kingdom, MRC Clinical Trials Unit, London, United Kingdom

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  • Kimberley A. Goldsmith,

    Affiliation: Biostatistics Department, Institute of Psychiatry, King’s College London, London, United Kingdom

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  • Peter D. White

    Affiliation: Centre for Psychiatry, Wolfson Institute of Preventive Medicine, Barts and the London School of Medicine and Dentistry, Queen Mary University of London, London, United Kingdom

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  • Published: August 01, 2012
  • DOI: 10.1371/journal.pone.0040808

Reader Comments (28)

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Coercive practices by insurance companies and others should stop following the publication of these results

Posted by tkindlon on 06 Aug 2012 at 02:17 GMT

For over a decade now, some individual patients with Chronic Fatigue Syndrome (CFS)* in Great Britain and Ireland (and probably elsewhere) have been pressurised by insurance companies into undertaking graded exercise therapy (GET) and cognitive behaviour therapy, based on scheduling increases activity. This seems to have been largely due to hype around the efficacy of GET and CBT and extrapolations from subjective measures, as the evidence that such interventions are efficacious in restoring the ability to work is week.


A lot of the evidence has been summarised in a review (1). For some reason this is quoted sometimes as justifying claims it is is evidence-based to say that GET and CBT have been shown to restore the ability to work in CFS. However the data is far less impressive. It is summarised in table 6. The accompanying text says: "Among the 14 interventional trials with work or impairment results after intervention, there were too few of any single intervention with any specific impairment domain to allow any assessment of association."

The PACE Trial is by far the biggest trial of these therapies in the field. It shows neither CBT nor GET led to an improved rate of days of lost employment [Means (sds): APT: 148.6 (109.2); CBT: 151.0 (108.2); GET: 144.5 (109.4); SMC (alone): 141.7 (107.5)] (Table 2) (2). Neither CBT nor GET led to improvements in numbers receiving welfare benefits or other financial payments (Table 4). These results are in contrast to the self-reported improvements in fatigue, physical functioning and some other measures (3).

This information comes a few years after a major audit of Belgian CFS rehabilitation (CBT & GET) centres (4). The sample size was large, with over 600 patients with a confirmed diagnosis of CFS (using the Fukuda et al. criteria (5)) taking part. It "comprised on average per patient 41 to 62 hours of rehabilitation" It found that "physical capacity did not change; employment status decreased at the end of the therapy." Again improvements were found in some self-reported measures.

The ethics of using coercion in medical practice generally is very questionable. Coercive practices should certainly be very questionable with therapies where they are plenty of reasons to believe they can cause harm (6). Furthermore, high rates of adverse reactions have been reported by patients, particularly with GET (7).

Also chronic fatigue syndrome causes a reduced amount of energy to be available to individuals. It can be very challenging to be ill with CFS, trying to balance the different aspects of one's life with reduced energy levels. People with CFS shouldn't be forced without good reason to have to do a time- and energy-consuming CBT or GET course. The data shows there isn't a good reason. Of course even if the results were better, its still very questionable whether coercion is justified: we don't coerce (healthy) people to exercise for at least 30 minutes five times a week even though it would be good on average in terms of people's health. Similarly, we don't force people to drink less than the recommended limit for daily and weekly alcohol assumption. And just to be clear again, the benefits (in comparison to the risks) of CBT or GET are not nearly as clear cut as the benefits (in comparison to the risks) of exercising regularly or avoiding excesses of alcohol are for people in the general population.

Hopefully the publication of this trial will stop coercive practices in the CFS field once and for all.

* I'll use the term for consistency.

References:

(1) Ross SD, Estok RP, Frame D, Stone LR, Ludensky V, Levine CB. Disability and chronic fatigue syndrome: a focus on function. Arch Intern Med. 2004 May 24;164(10):1098-107. http://archinte.ama-assn.... or http://archinte.ama-assn....

(2) McCrone P, Sharpe M, Chalder T, Knapp M, Johnson AL, et al. (2012) Adaptive Pacing, Cognitive Behaviour Therapy, Graded Exercise, and Specialist
Medical Care for Chronic Fatigue Syndrome: A Cost-Effectiveness Analysis. PLoS ONE 7(8): e40808. doi:10.1371/journal.pone.0040808

(3) White PD, Goldsmith KA, Johnson AL, Potts L, Walwyn R, et al. (2011) Comparison of adaptive pacing therapy, cognitive behaviour therapy, graded exercise therapy, and specialist medical care for chronic fatigue syndrome (PACE): a randomised trial. Lancet 377: 823–836.

(4) [Fatigue Syndrome: diagnosis, treatment and organisation of care]
KCE Reports 88. (with summary in English). Accessed: 6th August, 2012. https://kce.fgov.be/publi...

(5) Fukuda K, Straus SE, Hickie I, Sharpe MC, Dobbins JG, Komaroff A. The chronic fatigue syndrome: a comprehensive approach to its definition and study. International Chronic Fatigue Syndrome Study Group. Ann Intern Med. 1994 Dec 15;121(12):953-9.

(6) Twisk FN, Maes M. A review on cognitive behavorial therapy (CBT) and graded exercise therapy (GET) in myalgic encephalomyelitis (ME) / chronic fatigue syndrome (CFS): CBT/GET is not only ineffective and not evidence-based, but also potentially harmful for many patients with ME/CFS. Neuro Endocrinol Lett. 2009;30(3):284-99. Review.

(7) Kindlon T. Reporting of Harms Associated with Graded Exercise Therapy and Cognitive Behavioural Therapy in Myalgic Encephalomyelitis/Chronic Fatigue Syndrome Bulletin of the IACFS/ME. 2011;19(2):59-111. http://www.iacfsme.org/BU...

No competing interests declared.

RE: Coercive practices by insurance companies and others should stop following the publication of these results

tkindlon replied to tkindlon on 06 Aug 2012 at 02:19 GMT

Oops, I was trying to add line gaps to that and it got posted by accident before I had a chance to complete the competing interests section.

Competing interests declared: I work in a voluntary (i.e. unpaid) capacity for the Irish ME/CFS Association

RE: Coercive practices by insurance companies and others should stop following the publication of these results

tkindlon replied to tkindlon on 06 Aug 2012 at 02:42 GMT

I also hadn't finished checking for typos, etc. before it was prematurely posted, hence some embarrassing spelling mistakes, etc.

No competing interests declared.

RE: RE: Coercive practices by insurance companies and others should stop following the publication of these results

Catharguy replied to tkindlon on 06 Aug 2012 at 13:01 GMT

A quite brilliant dissertation that shows what most individuals/patients know already, that CBT and GET are ineffective in treating either Chronic Fatigue Syndrome or Myalgic Encephalomyelitis. This evidence based thesis also gives the leading M.E. Charities inside the U.K. the impetus now to challenge the CBT/GET doctrine through the law courts.

No competing interests declared.

RE: Coercive practices by insurance companies and others should stop following the publication of these results

dtfraser replied to tkindlon on 07 Aug 2012 at 02:10 GMT


In their recent editorial "Does Conflict of Interest Disclosure Worsen Bias?", the PLoS Editors concluded that [1]:

"Indeed, if disclosure worsens bias, then this is a game-changer for discussion and debate about managing conflicts of interest in medicine.

Journals, professional associations, clinical guideline developers, and others need to worry not just that disclosure provides a band-aid to the real problem of the COI itself, but that any attempt to stem the trouble through disclosure policies may actually be worsening the problem".

Several years ago, Ioannidis pointed out that [2]:

"Conflicts of interest and prejudice may increase bias, u.

Conflicts of interest are very common in biomedical research, and typically they are inadequately and sparsely reported.

Prejudice may not necessarily have financial roots.

Scientists in a given field may be prejudiced purely because of their belief in a scientific theory or commitment to their own findings.

Many otherwise seemingly independent, university-based studies may be conducted for no other reason than to give physicians and researchers qualifications for promotion or tenure.

Such nonfinancial conflicts may also lead to distorted reported results and interpretations.

Prestigious investigators may suppress via the peer review process the appearance and dissemination of findings that refute their findings, thus condemning their field to perpetuate false dogma".

Three authors of this paper [3], who are also the inventors of GET and CBT for "CFS", and the principal investigators who designed and obtained funding for the PACE study [4], declare interests which apparently conflict with their involvement in their own trial.

The main competing interests are described by them as having "done voluntary and paid consultancy work" for Government and Insurance Companies.

On the topic of competing interests (or Conflicts of Interest), Resnik has argued that: "Indeed, one of the best ways to destroy trust is to not disclose a relevant financial (or other) interest to a subject when they are being recruited for a study, only to have the subject find out after the study is underway or has been completed. A patient (or subject) who learns about important information that was not disclosed is likely to feel betrayed or manipulated.

"Researchers should always strive to help subjects understand and interpret information, whether it is information about benefits, risks, and alternatives, or information about COIs".

The following brief extracts may give some idea about the nature and quality of advice to Insurance and Government by one (or more) of the Principal PACE investigators [6]:

"ref:059874 there is strong evidence that symptoms and disability are shaped by psychological factors".

"ref: 059874 social factors are almost certainly of great importance in shaping functional illness"

"ref: 059874 The current system of state benefits, insurance payments and litigation remain potentially major obstacles to effective rehabilitation".

"ref: 059874 The demands such persons make on health service, social benefit systems, and insurers are likely to increase".

"ref: 059874 how much is it costing you doing nothing?"

"Research in several functional syndromes has found that a strong belief and preoccupation that one has a 'medical disease' and a helpless and passive attitude to coping is associated with persistent disability"

"Some persons appear to exaggerate symptoms but this is often hard to prove".

"A shift towards a more consumer-based approach to health is also likely to increase the prominence if not the prevalence of such syndromes as the authority of medicine to define what is a legitimate illness is diminished.

Indeed, increasingly consumer oriented and privatised doctors will collude with the patient’s views that they have a disabling and permanent disease".

"Relevant factors include the information patients receive about the symptoms and how to cope with them.

This information may be helpful or may stress the chronicity of the illness and promote helplessness.

Such unhelpful information is found in “self-help” (!) books and increasingly on the Internet (see for example www.meassociation.org.uk).

Unfortunately, doctors and especially “specialist private doctors” and complementary therapists may be as bad.

Other social factors that perpetuate illness are anger with the person or organisation the illness is illness attributed to, or toward the insurer for not believing them.

It has been pointed out that: “if you have to prove you are ill you can’t get well”.

Both State and private insurers pay people to remain ill.

Recent systematic reviews of randomised controlled trials have confirmed that psychologically informed rehabilitative treatments (often called cognitive behaviour therapy or CBT) and “antidepressant” drugs have some effectiveness in treating most functional syndromes.

There is however a great shortage of skilled providers of CBT in particular and rehabilitative facilities in general.

Obstacles to recovery

In practice, even if treatment is available, there may be obstacles to recovery.

Over time, the patient’s beliefs may be become entrenched and be driven by anger and the need to explain continuing disability.

The current system of state benefits, insurance payments and litigation remain potentially major obstacles to effective rehabilitation.

It is often unrealistic to expect medical treatment alone to overcome these.

Furthermore patient groups who champion the interest of individuals with functional complaints (particularly for chronic fatigue and fibromyalgia) are increasingly influential;

they are extremely effective in lobbying politicians and have even been threatening towards individuals and organisations who question the validity and permanence of the illness they champion.

Again the ME lobby is the best example".


It is not obvious from the available documentation that participants were exposed to and hence helped to understand and interpret the above relevant (for some) and non-esoteric information relating to aspects of the investigators self-declared conflicting (or competing) interests.

If the PACE researchers did not strive to help subjects understand and interpret information about the conflicting interests of all three main PACE investigators, it is rather difficult to understand how fully-informed consent could have been secured.

Given that it was the UK tax-payers money being used for this experiment, some 5 or 6 million GBP, perhaps the main investigators might like to take this opportunity to explain more fully how and why doing voluntary and paid work for Government and Private Insurance Companies constitutes a Conflict of Interest?


[1] Does Conflict of Interest Disclosure Worsen Bias? Barbour V, Clark J, Norton M, Simpson P, Veitch E. 24.04.12
http://www.plosmedicine.o...

[2] Why Most Published Research Findings Are False John P. A. Ioannidis 30.08.05
http://www.plosmedicine.o...

[3] Adaptive Pacing, Cognitive Behaviour Therapy, Graded Exercise, and Specialist Medical Care for Chronic Fatigue Syndrome: A Cost-Effectiveness Analysis McCrone P, Sharpe M., Chalder T., Knapp M, Johnson A, Goldsmith K, White P. 01.08.12

[4] White PD, Goldsmith KA, Johnson AL, Potts L, Walwyn R, et al. (2011) Comparison of adaptive pacing therapy, cognitive behaviour therapy, graded exercise therapy, and specialist medical care for chronic fatigue syndrome (PACE): a randomised trial. Lancet 377: 823–836 http://www.ncbi.nlm.nih.g...

[5] Disclosing Conflicts of Interest to Research Subjects: an Ethical and Legal Analysis Accountability in Research 2004; 11: 141-59. David B. Resnik, JD, PhD http://ukpmc.ac.uk/abstra...

[6] UnumProvident: Trends in Health and Disability 2002 Chief Medical Officer’s Report
Functional Symptoms and Syndromes: Recent Developments Michael Sharpe
http://issuu.com/dtdocs/d...


No competing interests declared.

RE: RE: Coercive practices by insurance companies and others should stop following the publication of these results

EdJackson replied to dtfraser on 08 Aug 2012 at 00:31 GMT

In regard to the impact this work may have upon the way in which those with CFS are treated by insurance companies, and potential conflicts of interest, it may be worth mentioning that as part of an earlier correspondence relating to the PACE trial Trudie Chalder declared:

"TC has done consultancy work for insurance companies and has received royalties from Sheldon Press and Constable and Robinson." [1]

The current competing interests section of this paper Chalder only says:

"TC has received royalties from Sheldon Press and Constable and Robinson." [2]

This could mean that some readers fail to realise that, along with other researcher who have worked on this paper, Chalder also has prior financial relationships with insurance companies.


[1] Chalder T, Sharpe M, White PD. PACE trial clarification. The Lancet, Volume 379, Issue 9816, Page 616.doi:10.1016/S0140-6736(12)60267-0

[2] McCrone P, Sharpe M, Chalder T, Knapp M, Johnson AL, et al. (2012) Adaptive Pacing, Cognitive Behaviour Therapy, Graded Exercise, and Specialist
Medical Care for Chronic Fatigue Syndrome: A Cost-Effectiveness Analysis. PLoS ONE 7(8): e40808. doi:10.1371/journal.pone.0040808

No competing interests declared.