“Doctors can commit scientific fraud and financial fraud and not be punished”

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Press Release by Dr Myhill concerning Dr Myhill’s Virtual Hearing 22 MARCH 2021 – vs ICO & GMC

Doctors can commit scientific fraud and financial fraud and not be punished. This is the conclusion of Dr Sarah Myhill following her recent hearing vs the ICO and GMC.

Dr Sarah Myhill tells us “That is official General Medical Council policy”.

The PACE study of 2011, which concluded that patients with CFS and ME could be effectively treated with graded exercise and cognitive behaviour therapy, has been proven to be scientifically fraudulent. This fraud is so profound that PACE recommendations have been dropped by NICE. We now know that graded exercise makes patients with CFS, ME and Long Covid much worse. CBT is of little benefit, only as a supportive measure.


In response to this fraudulent study, Dr Myhill reported the authors of PACE, and its directors, to the General Medical Council in January 2018. The GMC has a duty, and is empowered by Parliament, to regulate doctors and this includes research misconduct. Despite taking six months to consider Dr Myhill’s request, the GMC refused to investigate. Dr Myhill supplied extensive scientific proofs but in its refusal the GMC failed to supply its scientific defence.


So, Dr Myhill, through FoI legislation, asked that the GMC supply her with the scientific references on which it relied in coming to its decision not to investigate the PACE authors. The GMC refused. It gave no reasons whatsoever.


Consequently, Dr Myhill reported the GMC to the Information Commissioner who in a ruling of 30 September 2019 agreed with her. The ICO informed the GMC that it must supply her with the scientific references on which it relied in deciding not to investigate the PACE authors. This was because Dr Myhill was asking simply for scientific references already in the public area. This was only fair to the thousands of patients who have been damaged by graded exercise and who have a right to a proper explanation as to why.


At this point you would think the GMC had to comply. However, it is suspected that the GMC refusal arose for reasons of cronyism – it did not want ex-Presidents of Royal Societies up in
front of the GMC and the Police. It did not want to admit that actually it held NO scientific evidence, and it had no good reason to proceed as it did.


So, the GMC had to think up some sort of legal argument for refusal. Hitherto it had no argument – simply blunt refusal. The GMC consulted with its legal beavers within and outside the GMC and came up with the argument that to comply with the ICO demands would infringe the personal privacy of the PACE authors. What a nonsense! Dr Myhill has no interest in the personal data of the PACE authors. She simply requested scientific references which should all be in the public arena!


This was the subject of the ICO Hearing on 22 March 2021: Myhill vs GMC and ICO.


The outcome was a split decision. It boiled down to the Public Interest test. Tribunal member Mr Malcolm Clarke agreed with Dr Myhill. He stated:


“I conclude that Dr Myhill’s legitimate interest in seeking this information, if it exists, as a practising doctor with patients, who has a deep professional interest in ensuring that national recommended treatments in this area of medicine are evidence-based, is a very strong one …Dr Myhill’s legitimate interest in knowing whether the information she requests is held by the GMC is a very strong one. I therefore conclude that ……Dr Myhill’s legitimate interests are not overridden by the rights and freedoms of the data subjects.”

Ref paragraphs 42-47,EA-2020-0018 Myhill v IC & GMC


Luckily for the GMC, the Judge Hazel Oliver and Panel member Gareth Jones disagreed. They decided the other way round.


This Ruling sends a very clear message to doctors who commit scientific fraud – it is easy to get away with it, you can easily hide behind Data Protection issues and the General Medical Council will assist. Cronyism works.


………and so now to round 4. Dr Myhill will not give up.


See https://www.drmyhill.co.uk/wiki/My_Complaint_to_the_GMC_about_the_PACE_authors for more detail

ME/CFS and CBT – a basic error

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For more than three decades, the Wessely School has searched for empirical support for its psychosomatic approach to CFS. That search has been in vain. I show here, here and here that the theoretical assumptions of the Wessely approach lack support and have fallen.

The drive to show CBT and GET are effective treatments has been a key part of the failure, e.g. the PACE trial. An independent review by NICE suggests that GET is unsafe and CBT can only be weakly supported and, quite likely, is only a placebo effect.

In facing the mountain of invalidation that the Wessely School is having to endure, it has made the most basic error for any scientist: converting an inconclusive association into a conclusion of causation.

Correlation does not equal causation

Everybody knows it. It is drummed into people’s heads from the very beginning.

Yet, as a journal editor I have discovered that it is the most basic and common error by authors in psychology and healthcare, no matter how experienced the investigator. This common mistake can throw a mantle of doubt over a publication or even an entire research programme.

The fundamental distinction between correlation and causation is legendary. It is taught in first year medical and psychology classes all over the world.  Yet, the distinction can allude even the most seasoned researchers in Psychology, Psychiatry and kindred fields.

An introduction to the topic for 14-216 year old students studying Health is here.

A video for Biology GCSE students about the topic is available below.

An often cited example of the correlation=causation mistake concerns the polio epidemics in the US and Europe during the 1940s and 50s in the pre-vaccination period. Polio was crippling thousands of people, mostly children (and still is in some parts of the world). Polio epidemics occurred during summer and autumn. People eat more ice cream during summer and autumn. So for a while, children were warned not to eat ice cream or they would get polio.

Correlation is an association between two variables. In the late 1940s the polio rate (Y) and ice cream sales (X) could show a close correlation but eating ice creams did not cause polio:

X (ice cream eating) -/> Y (polio rate increase)

Causation is a cause and effect relationship between two variables. In 1949 in the US hot weather (Z) led to more people using public swimming pools and to more people eating ice creams (X) so both were caused by hotter weather (Z), which led to higher rates of polio (people swam in non-chlorinated pools more frequently) and to more ice cream eating:

The same kind of erroneous logic occur everyday in science, even among some of the most experienced researchers who are often strongly influenced by confirmation bias.

Z (hotter weather)-> X (ice cream eating)

and

Z (hotter weather) -> Y (polio rate increase)

Adamson, Ali,  Santhouse, Wessely & Chalder (2020)

In October 2020, Adamson, Ali,  Santhouse, Wessely and Chalder published a study in the Journal of the Royal Society of Medicine that purported to demonstrate that CBT ‘led to’ significant improvements in CFS patients.  The authors had reached the conclusion that CBT caused improvements but the evidence warranted no such thing. What exactly did the authors do in their study?

Objectives

The authors’ aim was to examine the effectiveness of CBT for CFS in a naturalistic setting and examine what factors, if any, predicted outcome. Note that ME is not mentioned because patients with ME were not included in the study. Nor should they have been because CBT could not possibly have helped them.

Design

They analysed patients’ self-reported ‘symptomology’ over the course of treatment and at three-month follow-up. They also explored baseline factors associated with improvement at follow-up.

Setting and Participants

Data were available for 995 patients receiving CBT for CFS at an outpatient, specialist clinic in the UK.

Main outcome measures

Patients were assessed throughout their treatment using self-report measures including the Chalder Fatigue Scale, 36-item Short Form Health Survey, Hospital Anxiety and Depression Scale and Global Improvement and Satisfaction. Note, these are all self-reported, subjective outcome measures.

Results

“Patients’ fatigue, physical functioning and social adjustment scores significantly improved over the duration of treatment with medium to large effect sizes (|d| = 0.45–0.91). Furthermore, 85% of patients self-reported that they felt an improvement in their fatigue at follow-up and 90% were satisfied with their treatment. None of the regression models convincingly predicted improvement in outcomes with the best model being (R2 = 0.137).”

Conclusion

As stated in the Abstract the Conclusion implies, but not does categorically state, a causal role for the CBT intervention. However, inside the main body of the article the authors state the conclusion that makes the CBT treatment causal in a manner that is unwarranted. They make the fundamental correlation equals causation error.

Enter stage left:

Brian Hughes and David Tuller (2021)

In a well-argued paper, Brian Hughes and David Tuller (2021) demonstrate that Adamson et al.’s (2020) conclusions are “misplaced and unwarranted.” They had submitted their critique to the Journal of the Royal Society of Medicine but the Editor did not accept it. Hughes and Tuller made a preprint available online and submitted it to the Journal of Health Psychology where it was reviewed and accepted and will shortly appear online. Here I quote from the Abstract:

“[Adamson et al.] interpret their data as revealing significant improvements following cognitive behavioural therapy in a large sample of patients with chronic fatigue syndrome and chronic fatigue. Overall, the research is hampered by several fundamental methodological limitations that are not acknowledged sufficiently, or at all, by the authors. These include: (a) sampling ambiguity; (b) weak measurement; (c) survivor bias; (d) missing data; and (e) lack of a control group. In particular, the study is critically hampered by sample attrition, rendering the presentation of statements in the Abstract misleading with regard to points of fact, and, in our view, urgently requiring a formal published correction. In light of the fact that the paper was approved by multiple peer-reviewers and editors, we reflect on what its publication can teach us about the nature of contemporary scientific publication practices.”

 A Few Details

In their paper, Tuller and Hughes point out that the Adamson et al. study and paper:

“are both problematic in several critical respects. For example, the Abstract – the section of the paper most likely to be read by clinicians – contains a crucial error in the way the data are described, and requires urgent correction.” They point out that a conspicuous controversy is overlooked. Adamson et al. write that the intervention is “based on a model which assumes that certain triggers such as a virus and/or stress trigger symptoms of fatigue. Subsequently symptoms are perpetuated inadvertently by unhelpful cognitive and behavioural responses” (p. 396). Treatment involves, among other elements, “addressing unhelpful beliefs which may be interfering with helpful changes” (p. 396).

The theory of unhelpful beliefs was laid out in a 1989 paper by the Wessely team that included two of the Adamson et al. paper’s authors (Wessely and Chalder). Recent posts here, here, and here show that the theory is lacking in any scientific support leaving the theory totally broken.

This fact was brushed under the carpet and simply not mentioned in the Adamson et al. paper.

Tuller and Hughes report that Adamson et al. are similarly selective in their discussion of the literature on CBT. After scrutiny of 172 CBT outcomes, the redrafted NICE guidance makes it perfectly clear that all of the research is of either “low” or “very low” quality. According to NICE, not one claim for CBT efficacy was supported by any evidence exceeding the “low quality” threshold.

To quote Hughes and Tuller, the research reviewed by Adamson et al.:

“is hampered by several fundamental methodological limitations that are not acknowledged sufficiently, or at all, by the authors. These include: (a) sampling ambiguity; (b) weak measurement; (c) survivor bias; (d) missing data; and (e) lack of a control group. Given these issues, in our view, the findings reported by Adamson et al. are unreliable because they are very seriously inflated.”

I consider here the last point only for its relevance to cause and effect.

Lack of a control group

Causality can never be established without a control group or a control condition. Adamson et al. did not include a control group and so their data cannot possibly support an inference about causality.

Period.

Yet Adamson et al. write:

“The cognitive behavioural therapy intervention led to significant improvements in patients’ self-reported fatigue, physical functioning and social adjustment” (p. 400).  

This direct statement of causality is unjustifiable and, most likely, plain wrong.

The authors realise this – or were made to realise it by the editor or reviewers – because they state:

the lack of a control condition limits us from drawing any causal inferences, as we cannot be certain that the improvements seen are due to cognitive behavioural therapy alone and not any other extraneous variables” (p. 401).

As Brian Hughes and David Tuller (2021) point out, this statement includes another assertion of causality which is also self-contradictory: “In one sentence, therefore, the authors draw a causal inference while denying the possibility of being able to do just that given their study design.”  

Ironically, this kind of assertion is what some psychiatrists used to call ‘schizophrogenic’. Not a bad descriptor in this case. It is also a little piece of ‘doublethink‘ in which the reader is expected to simultaneously accept two mutually contradictory beliefs as correct.

Implications

  1. The Adamson et al. study does not and will never warrant the conclusion that CBT “led to” improvements in CFS symptoms.
  2. The draft NICE guidance establishes that the evidence in support of CBT for pwCFS is marginal. It is likely to be nothing more than a placebo effect.
  3. To quote Tuller and Hughes, “the authors have provided a partial dataset suggesting that some of their participants self-reported modest increases in subjective assessments of well-being …These changes in scores might well have happened whether or not CBT had been administered.”
  4. The flight of Adamson et al. into the illegitimate correlation-equals-causation error is possibly a sign of desperation. When nothing is working, there is little option but to make it up as you go along.
  5. The house of cards that is the Wessely School is fast tumbling down, and not before time.

ME/CFS and the PACE trial

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Here I review the disastrous trial known as the ‘PACE trial’. This updates a post from several years ago.

Readers may also be interested in seeing the Special Issue on the PACE trial in the Journal of Health Psychology (2017).

Review of the evidence indicates that none of the Wessely School’s hypotheses about the causes of ME/CFS are supported by the science (see here, here and here). Under these circumstances it would be scientifically impossible for ME/CFS treatments based on these incorrect principles to actually work. Only if there is a ‘fix’ and evidence is craftily manipulated by scientific sleight-of-hand could the therapies be made to look effective.

Which is exactly what has happened.

ME/CFS patients have known the truth for donkey’s years. Only the perpetrators of the ‘CBT/GET Illusion’ have claimed otherwise.

I review here the PACE trial and present a few details of an exposure by Carolyn Wilshire, Tom Kindlon, Alem Matthees and Simon McGrath (2017), which reveals the true null effect.

What was the PACE Trial?

Rarely in the history of clinical medicine have doctors and patients been placed so bitterly at loggerheads. The dispute had been a long time coming. Thirty years ago, a few psychiatrists and psychologists offered a hypothesis based on a Psychological Theory in which ME/CFS is constructed as a psychosocial illness. According to the Wessely School, ME/CFS patients have “dysfunctional beliefs” that their symptoms are caused by an organic disease. The ‘Dysfunctional Belief Theory’ (DBT) assumes that no underlying pathology is causing the symptoms; patients are simply being ‘hypervigilant to normal bodily sensations‘ (Wessely et al., 1989; Wessely et al., 1991).

The Wessely School Theory assumes that the physical symptoms of ME/CFS are the result of ‘deconditioning’ or ‘dysregulation’ caused by sedentary behaviour, accompanied by disrupted sleep cycles and stress. Counteracting deconditioning involves normalising sleep cycles, reducing anxiety levels and increasing physical exertion.  Attentional biases also divert the patients towards their symptoms.

To put it bluntly, the DBT asserts that ME/CFS is ‘all in the mind’.  Small wonder that patient groups have been expressing anger and resentment in their droves.

Top-Down Research

‘Top-down research’ uses a hierarchy of personnel, duties and skill-sets. The person at the top sets the agenda and the underlings do the work. The structure is a bit like the social hierarchy of ancient Egypt. Unless carefully managed, this top-down approach risks creating a self-fulfilling prophecy from confirmation biases at multiple levels. At the top of the research pyramid sits the ‘Pharaoh’, Regius Professor Sir Simon Wessely KB, MA, BM BCh, MSc, MD, FRCP, FRCPsych, F Med Sci, FKC, Knight of the Realm, king-pin and originator of the Wessely School. The principal investigators (PIs) for the PACE Trial were Professors White, Chalder and Sharpe, the ‘Inner Circle’ of the Wessely School. Another Inner Circle member, Sir Mansel Aylward, then at the Department for Work and Pensions, was a funder of the trial. The PIs all have or had connections both to the Department for Work and Pensions and to insurance companies.

The investigators obtained close to £5,000,000 of tax payers’ money to run the PACE trial.

The objective of the trial was to demonstrate that two treatments based on the DBT, cognitive behavioural therapy (CBT) and graded exercise therapy (GET), help ME/CFS patients to recover. 

There was a zero chance the PACE researchers would fail to obtain the results they wanted. As the PACE ship left port, it went directly towards its destination. Only when it struck that unfortunate iceberg called “Null Result” did things begin to go seriously wrong.

Groupthink, Conflicts and Manipulation

The PACE trial team were operating within a closed system or groupthink in which they ‘know’ their theory is correct. With every twist and turn, no matter what the actual data show, the investigators are able to confirm their theory. The process is well-known in Psychology. It is a self-indulgent processes of subjective validation and confirmation bias.  Groupthink occurs when a group makes faulty decisions because group pressures lead to a deterioration of “mental efficiency, reality testing, and moral judgment” (Janis, 1972). Given this context, we can see reasons to question the investigators’ impartiality with many potential conflicts of interest (Lubet, 2017). Furthermore, critical analysis suggests that the PACE investigators involved themselves in manipulating protocols midway through the trial, selecting confirming data and omitting disconfirming data, and publishing biased reports of findings which created a catalogue of errors.

‘Travesty of Science’

The PACE Trial has been termed a ‘travesty of science’ while sufferers of ME/CFS continue to be offered unhelpful or harmful treatments and are basically being told to ‘pull themselves together’. One commentator has asserted that the situation for ME patients in the UK is: “The 3 Ts – Travesty of Science; Tragedy for Patients and Tantamount to Fraud” (Professor Malcolm Hooper, quoted by Williams, 2017). Serious errors in the design, the protocol and procedures of the PACE Trial are evident. The catalogue of errors is summarised below. The PACE Trial was loaded towards finding significant treatment effects.

When Disaster Strikes

The claimed benefits of GET and CBT for patient recovery are entirely spurious. The explanation lies in a sequence of serious errors in the design, the changed protocol and procedures of the PACE Trial. The investigators neglected or bypassed accepted scientific procedures for a RCT, as follows:

ErrorCategory of errorDescription of error
1Ethical issue: Applying for ethical approval and funding for a long-term trial when the PIs knew already knew CBT effects on ME/CFS were short-lived.On 3rd November 2000, Sharpe confirmed: “There is a tendency for the difference between those receiving CBT and those receiving the comparison treatment to diminish with time due to a tendency to relapse in the former” (www.cfs.inform/dk). Wessely stated in 2001 that CBT is “not remotely curative” and that: “These interventions are not the answer to CFS” (Editorial: JAMA 19th September 2001:286:11) (Williams, 2016).
2Ethical issue: Failure to declare conflicts of interest to Joint Trial Steering Committee.Undeclared conflicts of interest by the three PIs in the Minutes of the Joint Trial Steering Committee and Data Monitoring Committee held on 27th September 2004.
3Ethical issue: Failure to obtain fully informed consent after non-disclosure of conflicts of interest.Failing to declare their vested financial interests to PACE participants, in particular, that they worked for the PHI industry, advising claims handlers that no payments should be made until applicants had undergone CBT and GET.
4Use of their own discredited “Oxford” criteria for entry to the trial.Patients with ME would have been screened out of the PACE Trial even though ME/CFS has been classified by the WHO as a neurological disease since 1969 (ICD-10 G93.3).
5Inadequate outcome measures.Using only subjective outcome measures.The original protocol included the collection of actigraphy data as an objective outcome measure. However, after the Trial started, the decision was taken that no post-intervention actigraphy data should be obtained.
6Changing the primary outcomes of the trial after receiving the raw data.Altering outcome measures mid-trial in a manner which gave improved outcomes.
7Changing entry criteria midway through the trial.Altering the inclusion criteria for trial entry after the main outcome measures were lowered so that some participants (13%) met recovery criteria at the trial entry point.
8The statistical analysis plan was published two years after selective results had been published.The Re-definition of “recovery” was not specified in the statistical analysis plan.
9Inadequate controlSending participants newsletters promoting one treatment arm over another, thus contaminating the trial.
10Inadequate controlLack of comparable placebo/control groups with inexperienced occupational therapists providing a control treatment and experienced therapists provided CBT.
11Inadequate controlRepeatedly informing participants in the GET and CBT groups that the therapies could help them get better.
12Inadequate controlGiving patients in the CBT and GET arms having more sessions than in the control group.
13Inadequate controlAllowing therapists from different arms to communicate with each other about how patients were doing.
14Lack of transparencyBlocking release of the raw data for five years preventing independent analysis by external experts.

Credit where credit is due

A significant amount of investigation about the PACE trial was carried out in 2015 by David Tuller.

Please see:

Tuller D (2015) TRIAL BY ERROR: The Troubling Case of the PACE Chronic Fatigue Syndrome Study. http://www.virology.ws/2015/10/21/trial-by-error-i/

Tuller D (2015) TRIAL BY ERROR: The Troubling Case of the PACE Chronic Fatigue Syndrome Study (second installment). http://www.virology.ws/2015/10/22/trial-by-error-ii/

Tuller D (2015) TRIAL BY ERROR: The Troubling Case of the PACE Chronic Fatigue (final installment) http://www.virology.ws/2015/10/23/trial-by-error-iii/

Tuller (2016) http://www.virology.ws/2016/02/01/trial-by-error-continued-a-few-words-about-harassment/

Other significant pieces of exposure were:

Goldin R. PACE: The research that sparked a patient rebellion and challenged medicine. www.stats.org/pace-research-sparked-patient-rebellion-challenged-medicine

This paper by Dr Mark Vink was described in a letter to the Editor of Lancet from Professor Malcolm Hooper as “The final coup de grace“:

The PACE Trial Invalidates the Use of Cognitive Behavioral and Graded Exercise Therapy in Myalgic Encephalomyelitis/Chronic Fatigue Syndrome: A Review. J Neurol Neurobiol 2(3).

PACE-Gate: the Cover-Up

Blocking release of the raw data for five years and preventing independent analysis by external experts was tantamount to a cover-up of the true findings. An editorial by Keith Geraghty (2016) was entitled ‘PACE-Gate’. ME/CFS patient associations were rightly suspicious of the recovery claims concerning the GET arm of the trial because of their own experiences of intense fatigue after ordinary levels of activity which were inconsistent with the recovery claims of the PACE Trial reports. For many sufferers, even moderate exercise results in long ‘wipe-outs’ in which they are almost immobilized by muscle weakness and joint pain. In the US, post-exertional relapse has been recognized as the defining criterion of the illness by the Centers for Disease Control, the National Institutes of Health and the Institute of Medicine. For the PACE investigators, however, the announced recovery results validated their conviction that psychotherapy and exercise provided the key to reversing ME/CFS.

Alem Matthees Obtains Data Release

When Alem Matthees, a ME/CFS patient, sought the original data under the Freedom of Information Act and a British Freedom of Information tribunal ordered the PACE team to disclose their raw data, some of the data were re-analysed according to the original protocols. The legal costs of the tribunal at which QMUL were forced to release the data, against their strenuous objections, was over £245,000. The re-analysis of the PACE Trial data revealed that the so-called “recovery” under CBT and GET all but disappeared (Carolyn Wilshire, Tom Kindlon, Alem Matthees and Simon McGrath, 2016). The recovery rate for CBT fell to seven percent and the rate for GET fell to four percent, which were statistically indistinguishable from the three percent rate for the untreated controls. 

Graded exercise and CBT are still being routinely prescribed for ME/CFS in the UK despite patient reports that the treatments can cause intolerable pain and relapse. The analysis of the PACE Trial by independent critics has revealed a catalogue of errors and provides an object lesson in how not to conduct a scientific trial. The trial can be useful to instructors in research design and methodology for that purpose.

Following the re-analyses of the PACE Trial, the DBT is dead in the water. There is an urgent need for new theoretical approaches and scientifically-based treatments for ME/CFS patients. Meanwhile, there is repair work to be done to rebuild patient trust in the medical profession.

The Final Sinking

Caroline Wilshire, Tom Kindlon, Alem Matthees and Simon McGrath asked a very simple question:

Can patients with chronic fatigue syndrome really recover after graded exercise or cognitive behavioural therapy?

These authors gave a critical commentary and preliminary re-analysis of the PACE
trial. I quote their Abstract:

BACKGROUND: Publications from the PACE trial reported that 22%
of chronic fatigue syndrome patients recovered following graded
exercise therapy (GET), and 22% following a specialised form of
CBT. Only 7% recovered in a control, no-therapy group. These
figures were based on a definition of recovery that differed
markedly from that specified in the trial protocol.
PURPOSE: To evaluate whether these recovery claims are justified
by the evidence.
METHODS: Drawing on relevant normative data and other research,
we critically examine the researchers’ definition of recovery, and
whether the late changes they made to this definition were
justified. Finally, we calculate recovery rates based on the original
protocol-specified definition.
RESULTS: None of the changes made to PACE recovery criteria were
adequately justified. Further, the final definition was so lax that on
some criteria, it was possible to score below the level required for
trial entry, yet still be counted as ‘recovered’. When recovery was
defined according to the original protocol, recovery rates in the
GET and CBT groups were low and not significantly higher than in
the control group (4%, 7% and 3%, respectively).
CONCLUSIONS: The claim that patients can recover as a result of
CBT and GET is not justified by the data, and is highly misleading

Implications

  1. The PACE trial is/was/and always will be an unmitigated disaster. I use it in my textbook as an example of how not to do a trial.
  2. The authors and sponsors have done a disservice to science and to patients that will be hard to forget.
  3. An apology is the least that the principal investigators can do to make amends for this dreadful piece of pseudo-science.
  4. The universities involved should return the public funds that were wasted on the PACE trial project.
  5. A government enquiry is necessary to investigate the full facts in relation to the connections between the investigators, the insurance industry and the UK Department of Work and Pensions.

Note: This post is dedicated to Alem Matthees who has dedicated his life to the search for the truth about ME/CFS and was responsible for obtaining the release of the PACE trial data.

Reference:

David F Marks et al. (2020) Health Psychology. Theory, Research & Practice (6th ed.) SAGE Publications Ltd.

ME/CFS and unhelpful beliefs

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The Wessely School approach to ME/CFS and MUS hypothesizes a causative role for unhelpful beliefs (H1), deconditioning (H2), and biased attention (H3).

The evidence related to these three hypotheses is reviewed in turn. Here I consider H1: that unhelpful beliefs cause, or exacerbate, the symptoms of ME/CFS and MUS.

Future posts will review the evidence for H2 and H3.

Do unhelpful beliefs cause, or exacerbate, the symptoms of ME/CFS and MUS?

Unhelpful or dysfunctional illness beliefs referred to by the Wessely School include:

  • The belief that ME/CFS symptoms are the result of a physical illness (Note: if ME/CFS actually is a physical illness, the belief would be a true belief, and the only way it could be said to be unhelpful would be for a therapist wanting to claim otherwise)
  • The belief that ME/CFS needs medical treatment (physical treatment only)
  • The belief that exercise or too much activity has “harmful effects” which leads to “fear of exercise” (so-called ‘kinesiophobia’)

There is a huge evidence-base from studies relevant to beliefs, ME/CFS and MUS, which can be estimated to number in the thousands. However not all beliefs are dysfunctional beliefs. Also, as is typical in psychological research, the vast majority consist of cross-sectional studies which carry no evidential weight for or against any causal hypotheses.

Another sizable section of articles consists of narrative reviews, which, like this article, are intended to provide an interpretation of the evidence but do not and cannot provide a definitive case for or against causation. A small section of studies are randomised controlled trials (RCTs) which, if well-designed with appropriate controls and outcome measures, can indicate causal associations between treatments and outcomes.  A small number of prospective, observational studies offer support for the role of beliefs in illness, but not specifically ‘unhelpful beliefs’.

The table presents a set of studies which have been chosen to be representative of the literature.

Table 1. Research on Unhelpful Beliefs, Illness and Interventions

STUDY  DESIGNPUBLICATIONSFUNDINGCONCLUSIONS AND/OR INTERVENTIONS
Cancer prevention and prologation of life    

Narrative review interpreting ‘unsafe’ data
Eysenck, H. J. (1987). Some publications by Hans Eysenck on this topic were deemed ‘unsafe’ by a KCL Enquiry. To date, 86 papers are retracted or flagged for concern (Retraction Watch Database, 2021). However, several dozen of H J Eysenck’s publications based on the same ‘unsafe’ data-sets  remain unretracted and this form part of the scientific literature.None reported. However it has been established that the tobacco industry had secretly contributed large sums towards the project (Buchanan, 2010; Pelosi, 2019).  Hans Eysenck’s conclusion was stated as follows: “A detailed discussion is given of work …on the relationship between psychosocial factors and cancer. including the prophylactic use of behaviour therapy …to make possible the prevention of cancer or the prolongation of life in patients who are incurably ill.”   In spite of the fact that Hans Eysenck’s research on personality and cancer was funded by undeclared tobacco industry sources, the publications remain in the scientific literature. Evidently, the host institution (King’s College London), the journal publishers and editors are all content to allow these grossly misleading publications to remain uncorrected and unretracted in the scientific literature as citable contributions to science and medicine.  
ME/CFS     Narrative reviewWessely, David, Butler, & Chalder (1989)None reported.“You have had an acute illness, probably infectious in origin, which forced you to become inactive for a period of time. Subsequently you have begun to experience fatigue on exertion and as a result you have started to limit or avoid activity of all sorts…This means that you develop symptoms at increasingly lower levels of exercise…When you experience these symptoms, you have also experienced associated thoughts, such as ‘If I carry on I may become worse’, or ‘There must be something seriously wrong with me to make me feel like this’. These symptoms are real, but… you may have incorrectly attributed them to a recurrence of the original infection. This is particularly likely because the symptoms of muscle pain, breathlessness, dizziness, fatigue and others are similar to those experienced initially. This has led to a vicious circle of increasing avoidance, inactivity and fatigue.”  
Coronary heart disease and mortality   Narrative review interpreting ‘unsafe’ dataEysenck, H.J. (1991).   See note above.None reported but the tobacco industry contributed large sums towards the project.“autonomy (i.e., the ability to be independent in one’s thoughts, feelings, and actions, even under stress) is important for survival and is a valuable countermeasure as far as cancer and CHD are concerned.”  
ME/CFS   Psychometric studyMoss-Morris, Petrie & Weinman (1996)Auckland Institute of Technology Interim Research Committee.  “organic CFS  patients  have  distorted  perceptions  of  effort  and sensation which  may  contribute to their  functional  disability”.
Post-traumatic stress disorder   RCT  Marks, I., Lovell, K., Noshirvani, H., Livanou, M., & Thrasher, S. (1998)Wellcome Trust, London, England.  “Patients were taught to spot dysfunctional thoughts and thinking errors, elicit rational alternative thoughts, and reappraise beliefs about themselves, the trauma, and the world.”
Rehabilitation to work   Narrative reviewWaddell & Burton (2004)Department for Work and Pensions, UK  “…symptoms are by definition subjective and therefore at least partly a matter of perceptions.” “Changing dysfunctional perceptions, attitudes and behaviour is central to rehabilitation of many common health problems.”  
ME/CFS   Narrative reviewBrowne &  Chalder (2009) “Two models of understanding CFS, a cognitive behavioural model and a deconditioning model, are then introduced alongside the treatments on which they are based. Both cognitive behavioural therapy and graded exercise therapy have been recommended by the National Institute for Health and Clinical Excellence as they are the treatments for which there is most evidence.” See Figure 3. Note: NICE changed its guidance in 2020 suggesting that GET should be avoided and CBT is less effective than previously assumed.
ME/CFS   Narrative reviewKnoop H, Prins JB, Moss-Morris R, Bleijenberg G (2010).Dutch MS Research Foundation (Stichting MS Research).“Three different cognitive processes may play a role in the perpetuation of CFS symptoms. The first is a general cognitive representation in which fatigue is perceived as something negative and aversive and CFS is seen as an illness that is difficult to influence. The second process involved is the focusing on fatigue. The third element is formed by specific dysfunctional beliefs about activity and fatigue.”
ME/CFS PACE trial   RCT with multiple methodologic-al flawsWhite et al. (2011)  UK Medical Research Council (MRC G0200434 ), the Department of Health for England, the UK Department for Work and Pensions, and the Chief Scientist Office of the Scottish Government Health Directorates.“CBT  and  GET  can  safely  be  added  to  SMC  to  moderately  improve  outcomes  for  chronic  fatigue  syndrome, but APT is not an effective addition.”
Medically unexplained symptoms    Narrative reviewDeary, V., Chalder, T., & Sharpe, M. (2007)None reported“Hotopf (2004) has compared the role of doctors in MUS to that of parents of sick children. Both can reinforce unhelpful illness behaviour and symptom interpretations.” “The links so far found between central nervous system processes, such as the HPA axis, and immunological processes are intriguing but far from conclusive; the causal relationships are unclear”. “High neuroticism may offer an underlying common mechanism for distress sensitivity and intolerance which lowers the threshold for symptom detection (both mental and physical), and leads to increased propensity to conditioned responses, more attention to threat stimuli and more avoidant coping. The neuroticism concept captures many of the factors hypothesised to be at work in MUS.”  
Multiple sclerosis   Cross-sectional studyDennison, L., Moss-Morris, R., Silber, E., Galea, I., & Chalder, T. (2010)UK MS Society. NIHR Biomedical Research Centre, South London and Maudsley NHS Foundation Trust / Institute of Psychiatry King’s College London.“Illness severity factors explained only 2.2% of the variance in distress (p>.05) while cognitive and behavioural variables accounted for 37.1% (p<.001). Unhelpful beliefs about the self were the strongest predictor.”      
Mortality and diabetic foot ulcers   Prospective observational studyVedhara K, et al. (2016).Medical Research Council, UK (MC_U145079313). JNVM receives salary support from the RAND Corporation.“illness beliefs have a significant independent effect on survival in patients with diabetes and foot ulceration.”  
Irritable bowel syndrome    Systematic reviewWindgassen et al. (2017)National Institute of Health Research, HTA 11/69/02. TC  partly funded by the Biomedical Research Centre for the South London and Maudsley NHS Foundation Trust and the Institute of Psychiatry.TC received travel expenses and fees for workshops on irritable bowel syndrome.“change in illness‐specific cognitions is a key process by which psychological treatments may have an effect on the outcomes of symptom severity and QoL.”   The only significant effect related to so-called identity beliefs (‘How much do you experience symptoms?’), not beliefs that are specifically ‘unhelpful’.    
Mortality  and predialysis chronic kidney disease   Prospective observationalstudy  Muscat, P., Weinman, J., Farrugia, E. et al. (2020)None reported.  “CKD patients’ perceptions of treatment control, perceptions of a chronic timeline and perceived illness identity predict survival independently of clinical prognostic factors, including kidney function and co-morbidity. Illness perceptions are important and potentially modifiable risk factors in CKD.” ­­­­­­________________________ The beliefs that were evaluated cannot be specifically classified as ‘unhelpful’.  
Paediatric chronic fatigue syndrome   Cross-sectional study  Loades, M., Crawley, E., Chalder, T., & Flannery, H. (2021)National Institute for Health Research; Department of Health via the NIHR Specialist Biomedical Research Centre for Mental Health award to the South London and Maudsley NHS Foundation Trust and the Institute of Psychiatry at King’s College London.“the Lightning Process, based on Neurolinguistic Programming (NLP), when offered in addition to treatment as usual (TAU), was more clinically and cost effective compared to TAU alone (Crawley et al., 2017).”

Interpretation of the findings

An overall appraisal of the findings suggests that no published study has yet provided any empirical evidence of a causal relationship between unhelpful beliefs and ME/CFS symptoms.

The majority of ME/CFS studies have used a correlational approach, which as noted, has no evidential value in relation to causation. Cross-sectional studies are irrelevant to a causal hypothesis. The difference between correlational and causal relationships is fundamental but often it alludes even seasoned investigators.

Some studies used the Revised Illness Perception Questionnaire (IPQ-R) (Moss-Morris, Weinman, Petrie, Horne, Cameron & Buick, 2002). Although the authors often talk about illness beliefs and illness perceptions, they do not measure ‘unhelpful beliefs’. Studies that do not measure unhelpful beliefs are not relevant to H1. The IPQ-R measures identity, timeline, cyclical timeline, treatment control, personal control, coherence, causes and emotion reaction. None of these factors can be construed as a measure of unhelpful beliefs. For example, illness identity is measured by symptoms in which patients have to report whether they experienced the symptoms and, if so, whether they attributed the symptom to their illness. The number of symptoms attributed to their illness is summed with higher scores indicating increased illness identity. However, the latter score could equally be a measure of illness severity.

Petrie and Weinman (2012) claim that: “Illness perceptions change rapidly in response to diagnostic results and have been associated with emotional distress, recovery, and disability, as well as with treatment-related behaviour such as adherence”. However illness perceptions are not unhelpful beliefs and the only studies reviewed by them that used a prospective design did not measure unhelpful beliefs.

The largest RCT known as the ‘PACE trial’ (White et al., 2011) was in part concerned with the efficacy of CBT to control unhelpful beliefs but owing to a catalogue of methodological errors, the trial has been discredited (Marks, 2017; Wilshire, Kindlon, Matthees & McGrath, 2017; Wiltshire et al., 2018).  Another RCT called the ‘FINE’ trial involving gradually increasing activity designed collaboratively by the patient and the therapist and supportive listening found no evidence of efficacy (Wearden et al., 2006).

Two MUS studies with patients with foot ulcers (Vedhara et al., 2016) and chronic kidney disease (Muscat, Weinman, Farrugia et al., 2020) used a prospective, observational design enabling conclusions about causation. However the beliefs that were measured were not unhelpful beliefs but beliefs related to ‘identity’ (‘How much do you experience symptoms?’) as noted above.  This question can be interpreted as a measure of symptom severity and thus the observed effect on increased risk of mortality may only be attributed to greater symptom severity not to beliefs at all.  

Eysenck and Grossarth-Maticek’s publications on unhelpful beliefs or similar constructs (e.g. helplessness and hopelessness) are retracted, listed with expressions of concern, or regarded by this author as unsafe (see Marks & Buchanan, 2018, for details).

Implications

In the opinion of this author, the hypothesis that unhelpful beliefs cause or exacerbate symptoms of ME/CFS and MUS receives no empirical support in the research literature.

If true, this conclusion has several implications:

  1. As previously suggested, there is a plausible scientific explanation of ME/CFS in terms of a breakdown of homeostasis. If the biological explanation is true then ME/CFS patients’ beliefs that their symptoms are physically real are accurate.

2. The claim by therapists that patients who say they experience physical symptoms are holding unhelpful beliefs is a falsehood.

3. One has a right to ask the question: to whom are the so-called ‘unhelpful beliefs’ unhelpful – patients or therapists?

4. Patients who have CBT and/or GET to change their ‘unhelpful beliefs’ are fighting a losing battle. It is a no win situation. Refuse the therapy and you are being unhelpful (proving the therapist right). Do the therapy and show no improvement and when you say you’re not feeling any better, you’re being unhelpful all over again. A Catch 22 situation.

CBT Therapists out there: If you consider that my interpretation of the evidence is incorrect, please send me full details of the contradicting studies, and I will reconsider my position.

ME/CFS and treatment harms

Featured

Despite evidence of physiological and cellular abnormalities in myalgic encephalomyelitis (ME)/chronic fatigue syndrome (CFS), the dominant therapeutic approach has been cognitive behaviour therapy (CBT) and graded exercise therapy (GET).  Patients report  distress and dissatisfaction following healthcare encounters based on GET and CBT.  A significant body of research suggests that CBT and GET are harmful for many patients with ME/CFS. These findings raise ethical concerns and suggest that more collaborative working between scientists, therapists and patients would be helpful in making scientific progress in this difficult field.

CBT and GET follow the discredited biopsychosocial approach of influential psychiatrists and psychologists of the Wessely school (Wessely, David, Butler & Chalder, 1989; White et al., 2011). Multiple patient reports and studies indicate stigmatization, distress and dissatisfaction following GET and CBT. A significant body of research suggests what patients have known for a very long while that CBT and GET are not only ineffective, but harmful for many patients with ME/CFS.

Twisk and Maes (2009)

A review of CBT and GET by Twisk and Maes (2009) found that CBT and GET are  “not only ineffective and not evidence-based, but also potentially harmful for many patients with ME/CFS” (p. 284).  Twisk and Maes (2009) suggested that CBT/GET is hardly more effective than non-interventions or standard medical care, but also that “many patients report that the therapy had affected them adversely, the majority of them even reporting substantial deterioration”.  Exertion which is almost bound to occur with GET is most likely to have a negative impact on many ME/CFS patients. Exertion produces post-exertional malaise (PEM) which decreases aerobic capacity, increases muscoskeletal pain, neurocognitive impairment, “fatigue”, and weakness, and produces a slow recovery time. Twisk and Maes concluded that it is “unethical to treat patients with ME/CFS with ineffective, non-evidence-based and potentially harmful “rehabilitation therapies”, such as CBT/GET”.

Kindlon’s (2011, 2017) Studies

High rates of adverse effects have been reported with CBT and GET by ME/CFS patients. Kindlon (2011) reviewed 10 patient surveys from four countries found that 51per cent of respondents (range = 28%–82%, n = 4338, eight surveys) reported GET worsened their health, whereas 20 per cent of respondents (range = 7%– 38%, n = 1808, five surveys) reported similar results for CBT.

Kindlon (2017) also reviewed evidence that low-intensity exercise has the potential to exacerbate symptoms in CFS.  The effects of exercise can persist for more than a week after exertion e.g. gentle exercise of less than 7-minute duration can lead to worsening of fatigue, pain, sore throat and/or general health (Nijs et al., 2008; Van Oosterwijck et al., 2010). Longer-term, the effects of exercise can persist beyond 24 hours (VanNess et al., 2010). Lapp (1997) followed 31 patients for 12 days after a maximal exercise test  and found that the average relapse lasted 8.82 days with 22 per cent still in relapse at 12 days. Kindlon (2017) observed that: “interventions involving exercise could provoke a general and persistent worsening or exacerbation of symptoms in CFS. They also offer an explanation as to why it might be difficult for patients with CFS to adhere to graded activity/exercise interventions” (p. 1147). As noted, post-exertional malaise is a key symptom of ME/CFS. Numerous biological abnormalities have also been found following exertion (Lane et al., 2003; Light et al., 2009; Sorensen et al., 2009; Twisk and Maes, 2009).

Geraghty and Blease (2019)

In their review Geraghty and Blease (2019) identified seven potential modalities of iatrogenesis or harm reported by patients:

1. difficulties in reaching an acceptable diagnosis;

2. misdiagnosis, including of other medical and psychological conditions;

3. difficulties in accessing the sick role, medical care and social support;

4. high levels of patient dissatisfaction with the quality of medical care;

5. negative responses to controversial therapies (cognitive behavioral therapy, CBT, and graded exercise therapy, GET);

6. challenges to the patient narrative and experience;

7. psychological harm (individual and collective distress).

Geraghty and Blease (2019) concluded that the “biopsychosocial framework currently applied to ME/CFS is too narrow in focus and fails to adequately incorporate the patient narrative. Misdiagnosis, conflict, and harm are observable outcomes where doctors’ and patients’ perspectives remain incongruent. Biopsychosocial practices should be scrutinized for potential harms. Clinicians should consider adopting alternative patient-centred approaches.”

Friedberg, Sunnquist and Nacul (2020)

Friedberg, Sunnquist and Nacul (2020) also reviewed evidence suggesting that claims of safety of CBT and GET are “not adequately supported by the evidence and are contradicted by the experiences of clinicians and patients.”  They quote the 2014 Agency for Healthcare Research and Quality (AHRQ) review  that reported, “Harms [worsening of symptoms and/or disability] were generally inadequately reported across trials” (p. vi). The AHRQ report concluded that GET studies reported more adverse events and withdrawals. As early as 2012, experienced clinicians were not recommending PACE-type GET or CBT as treatments (Friedberg et al., 2012). They also cite Kindlon’s (2015) analysis of large international patient surveys in which more than 50% of pwME/CFS reported that CBT and GET fell short of delivering significant improvements and often led to worsened health due to ill-advised activity and exercise prescriptions.

McPhee, Baldwin, Kindlon, Hughes’ (2019) NHS Survey

McPhee et al. (2019) surveyed the National Health Service–affiliated ME/CFS specialist clinics in England to assess how harms following treatment are detected and to examine how patients are warned about the potential for harms. The researchers sent 57 clinics standardised information requests under the UK’s Freedom of Information Act. Data were received back from 38 clinics. Clinics were highly inconsistent in their approaches to the issue of treatment-related harm. Clinics placed little or no focus on the potential for treatment-related harm in their written information for patients and for staff. Furthermore, no clinic reported any cases of treatment-related harm, despite acknowledging that many patients dropped out of treatment. McPhee et al. recommended that clinics need to “develop standardised protocols for anticipating, recording, and remedying harms, and that these protocols allow for therapies to be discontinued immediately whenever harm is identified”.

NICE Draft Guidance (2020)

The draft NICE guideline released on 10 November 2020 stated:

1) NICE “recognises that ME/CFS, which is estimated to affect over 250,000 people in England and Wales, is a complex, multi-system, chronic medical condition where there is no ‘one size fits all’ approach to managing symptoms. It stresses the need for a tailored, individualised approach to care that allows joint decision making and informed choice.”

2)“Because of the harms reported by people with ME/CFS, as well as the committee’s own experience of the effects when people exceed their energy limits, the draft guideline says that any programme based on fixed incremental increases in physical activity or exercise, for example graded exercise therapy (GET) should not be offered for the treatment of ME/CFS.”

3) “Instead, it highlights the importance of ensuring that people remain in their ‘energy envelope’ when undertaking activity of any kind and recommends that a physical activity programme, in particular, should only be considered for people with ME/CFS in specific circumstances.”

4) “The draft guideline also emphasises that CBT it is not a treatment or cure for ME/CFS. However, as a supportive therapy which aims to improve wellbeing and quality of life, the draft guideline says CBT may be useful in supporting people who live with ME/CFS to manage their symptoms.”

Conclusion

It is self-evident that healthcare professionals seek to avoid and minimize harms when assisting ME/CFS patients. As suggested by Geraghty and Blease, a “concordant ‘patient-centred’ approach that give greater prominence to the patient narrative and experience of illness” is required.

In light of the findings on patient harms, extreme caution is required in offering pwME /CFS  treatments such as CBT and GET which involve increased  exercise or activity levels.

The findings on patient harms suggest that treatments using CBT and GET involve ethical dilemmas which practitioners can be helped to resolve by working with patients using a more collaborative approach.

Clinics should develop standardised protocols for anticipating, recording, and remedying harms, and these protocols should allow for therapies to be discontinued immediately whenever harm is identified.

Somewhat belatedly, the revised NICE draft guidance (2020) recognises the evidence on harms to ME/CFS patients from the use of GET and, to a lesser extent, CBT. The change in guidance is, in no small measure, a tribute to the researchers and patients who are cited here.

Dedication: This post is dedicated to Tom Kindlon and his indefatigable search for the truth about ME/CFS.