Before you enroll your child in the MAGENTA chronic fatigue syndrome study: Issues to be considered

[October 3 8:23 AM Update: I have now inserted Article 21 of the Declaration of Helsinki below, which is particularly relevant to discussions of the ethical problems of Dr. Esther Crawley’s previous SMILE trial.]

Petitions are calling for shutting down the MAGENTA trial. Those who organized the effort and signed the petition are commendably brave, given past vilification of any effort by patients and their allies to have a say about such trials.

Below I identify a number of issues that parents should consider in deciding whether to enroll their children in the MAGENTA trial or to withdraw them if they have already been enrolled. I take a strong stand, but I believe I have adequately justified and documented my points. I welcome discussion to the contrary.

This is a long read but to summarize the key points:

  • The MAGENTA trial does not promise any health benefits for the children participating in the trial. The information sheet for the trial was recently modified to suggest they might benefit. However, earlier versions clearly stated that no benefit was anticipated.
  • There is inadequate disclosure of likely harms to children participating in the trial.
  • An estimate of a health benefit can be evaluated from the existing literature concerning the effectiveness of the graded exercise therapy intervention with adults. Obtaining funding for the MAGENTA trial depended on a misrepresentation of the strength of evidence that it works in adult populations.  I am talking about the PACE trial.
  • Beyond any direct benefit to their children, parents might be motivated by the hope of contributing to science and the availability of effective treatments. However, these possible benefits depend on publication of results of a trial after undergoing peer review. The Principal Investigator for the MAGENTA trial, Dr. Esther Crawley, has a history of obtaining parents’ consent for participation of their children in the SMILE trial, but then not publishing the results in a timely fashion. Years later, we are still waiting.
  • Dr. Esther Crawley exposed children to unnecessary risk without likely benefit in her conduct of the SMILE trial. This clinical trial involved inflicting a quack treatment on children. Parents were not adequately informed of the nature of the treatment and the absence of evidence for any mechanism by which the intervention could conceivably be effective. This reflects on the due diligence that Dr. Crawley can be expected to exercise in the MAGENTA trial.
  • The consent form for the MAGENTA trial involves parents granting permission for the investigator to use children and parents’ comments concerning effects of the treatment for its promotion. Insufficient restrictions are placed on how the comments can be used. There is the clear precedent of comments made in the context of the SMILE trial being used to promote the quack Lightning Process treatment in the absence of evidence that treatment was actually effective in the trial. There is no guarantee that any comments collected from children and parents in the MAGENTA trial would not similarly be misused.
  • Dr. Esther Crawley participated in a smear campaign against parents having legitimate concerns about the SMILE trial. Parents making legitimate use of tools provided by the government such as Freedom of Information Act requests, appeals of decisions of ethical review boards and complaints to the General Medical Council were vilified and shamed.
  • Dr. Esther Crawley has provided direct, self-incriminating quotes in the newsletter of the Science Media Centre about how she was coached and directed by their staff to slam the patient community.  She played a key role in a concerted and orchestrated attack on the credibility of not only parents of participants in the MAGENTA trial, but of all patients having chronic fatigue syndrome/ myalgic encephalomyelitis , as well as their advocates and allies.

I am not a parent of a child eligible for recruitment to the MAGENTA trial. I am not even a citizen or resident of the UK. Nonetheless, I have considered the issues and lay out some of my considerations below. On this basis, I signed the global support version  of the UK petition to suspend all trials of graded exercise therapy in children and adults with ME/CFS. I encourage readers who are similarly in my situation outside the UK to join me in signing the global support petition.

If I were a parent of an eligible child or a resident of the UK, I would not enroll my child in MAGENTA. I would immediately withdraw my child if he or she were currently participating in the trial. I would request all the child’s data be given back or evidence that it had been destroyed.

I recommend my PLOS Mind the Brain post, What patients should require before consenting to participate in research…  as either a prelude or epilogue to the following blog post.

What you will find here is a discussion of matters that parents should consider before enrolling their children in the MAGENTA trial of graded exercise for chronic fatigue syndrome. The previous blog post [http://blogs.plos.org/mindthebrain/2015/12/09/what-patients-should-require-before-consenting-to-participate-in-research/ ]  is rich in links to an ongoing initiative from The BMJ to promote broader involvement of patients (and implicitly, parents of patients) in the design, implementation, and interpretation of clinical trials. The views put forth by The BMJ are quite progressive, even if there is a gap between their expression of views and their actual implementation. Overall, that blog post presents a good set of standards for patients (and parents) making informed decisions concerning enrollment in clinical trials.

Simon McGrathLate-breaking update: See also

Simon McGrath: PACE trial shows why medicine needs patients to scrutinise studies about their health

Basic considerations.

Patients are under no obligation to participate in clinical trials. It should be recognized that any participation typically involves burden and possibly risk over what is involved in receiving medical care outside of a clinical trial.

It is a deprivation of their human rights and a violation of the Declaration of Helsinki to coerce patients to participate in medical research without freely given, fully informed consent.

Patients cannot be denied any medical treatment or attention to which they would otherwise be entitled if they fail to enroll in a clinical trial.

Issues are compounded when consent from parents is sought for participation of vulnerable children and adolescents for whom they have legal responsibility. Although assent to participate in clinical trials is sought from children and adolescents, it remains for their parents to consent to their participation.

Parents can at any time withdraw their consent for their children and adolescents participating in trials and have their data removed, without requiring the approval of any authorities of their reason for doing so.

Declaration of Helsinki

The World Medical Association (WMA) has developed the Declaration of Helsinki as a statement of ethical principles for medical research involving human subjects, including research on identifiable human material and data.

It includes:

In medical research involving human subjects capable of giving informed consent, each potential subject must be adequately informed of the aims, methods, sources of funding, any possible conflicts of interest, institutional affiliations of the researcher, the anticipated benefits and potential risks of the study and the discomfort it may entail, post-study provisions and any other relevant aspects of the study. The potential subject must be informed of the right to refuse to participate in the study or to withdraw consent to participate at any time without reprisal. Special attention should be given to the specific information needs of individual potential subjects as well as to the methods used to deliver the information.

[October 3 8:23 AM Update]: I have now inserted Article 21 of the Declaration of Helsinki which really nails the ethical problems of the SMILE trial:

21. Medical research involving human subjects must conform to generally accepted scientific principles, be based on a thorough knowledge of the scientific literature, other relevant sources of information, and adequate laboratory and, as appropriate, animal experimentation. The welfare of animals used for research must be respected.

There is clearly in adequate scientific justification for testing the quack Lightning Process Treatment.

What Is the Magenta Trial?

The published MAGENTA study protocol states

This study aims to investigate the acceptability and feasibility of carrying out a multicentre randomised controlled trial investigating the effectiveness of graded exercise therapy compared with activity management for children/teenagers who are mildly or moderately affected with CFS/ME.

Methods and analysis 100 paediatric patients (8–17 years) with CFS/ME will be recruited from 3 specialist UK National Health Service (NHS) CFS/ME services (Bath, Cambridge and Newcastle). Patients will be randomised (1:1) to receive either graded exercise therapy or activity management. Feasibility analysis will include the number of young people eligible, approached and consented to the trial; attrition rate and treatment adherence; questionnaire and accelerometer completion rates. Integrated qualitative methods will ascertain perceptions of feasibility and acceptability of recruitment, randomisation and the interventions. All adverse events will be monitored to assess the safety of the trial.

The first of two treatments being compared is:

Arm 1: activity management

This arm will be delivered by CFS/ME specialists. As activity management is currently being delivered in all three services, clinicians will not require further training; however, they will receive guidance on the mandatory, prohibited and flexible components (see online supplementary appendix 1). Clinicians therefore have flexibility in delivering the intervention within their National Health Service (NHS) setting. Activity management aims to convert a ‘boom–bust’ pattern of activity (lots 1 day and little the next) to a baseline with the same daily amount before increasing the daily amount by 10–20% each week. For children and adolescents with CFS/ME, these are mostly cognitive activities: school, schoolwork, reading, socialising and screen time (phone, laptop, TV, games). Those allocated to this arm will receive advice about the total amount of daily activity, including physical activity, but will not receive specific advice about their use of exercise, increasing exercise or timed physical exercise.

So, the first arm of the trial is a comparison condition consisting of standard care delivered without further training of providers. The treatment is flexibly delivered, expected to vary between settings, and thus largely uncontrolled. The treatment represents a methodologically weak condition that does not adequately control for attention and positive expectations. Control conditions should be equivalent to the intervention being evaluated in these dimensions.

The second arm of the study:

Arm 2: graded exercise therapy (GET)

This arm will be delivered by referral to a GET-trained CFS/ME specialist who will receive guidance on the mandatory, prohibited and flexible components (see online supplementary appendix 1). They will be encouraged to deliver GET as they would in their NHS setting.20 Those allocated to this arm will be offered advice that is focused on exercise with detailed assessment of current physical activity, advice about exercise and a programme including timed daily exercise. The intervention will encourage children and adolescents to find a baseline level of exercise which will be increased slowly (by 10–20% a week, as per NICE guidance5 and the Pacing, graded Activity and Cognitive behaviour therapy – a randomised Evaluation (PACE)12 ,21). This will be the median amount of daily exercise done during the week. Children and adolescents will also be taught to use a heart rate monitor to avoid overexertion. Participants will be advised to stay within the target heart rate zones of 50–70% of their maximum heart rate.5 ,7

The outcome of the trial will be evaluated in terms of

Quantitative analysis

The percentage recruited of those eligible will be calculated …Retention will be estimated as the percentage of recruited children and adolescents reaching the primary 6-month follow-up point, who provide key outcome measures (the Chalder Fatigue Scale and the 36-Item Short-Form Physical Functioning Scale (SF-36 PFS)) at that assessment point.

actigraphObjective data will be collected in the form of physical activity measured by Accelerometers. These are

Small, matchbox-sized devices that measure physical activity. They have been shown to provide reliable indicators of physical activity among children and adults.

However, actual evaluation of the outcome of the trial will focus on recruitment and retention and subjective, self-report measures of fatigue and physical functioning. These subjective measures have been shown to be less valid than objective measures. Scores are  vulnerable  to participants knowing what condition they are assigned to (called ‘being unblinded’) and their perception of which intervention the investigators prefer.

It is notable that in the PACE trial of CBT and GET for chronic fatigue syndrome in adults, the investigators manipulated participants’ self-reports with praise in newsletters sent out during the trial . The investigators also switched their scoring of the self-report measures and produced results that they later conceded to have been exaggerated by their changing in scoring of the self-report measures [http://www.wolfson.qmul.ac.uk/current-projects/pace-trial#news ].

Irish ME/CFS Association Officer & Tom Kindlon
Tom Kindlon, Irish ME/CFS Association Officer

See an excellent commentary by Tom Kindlon at PubMed Commons [What’s that? ]

The validity of using subjective outcome measures as primary outcomes is questionable in such a trial

The bottom line is that the investigators have a poorly designed study with inadequate control condition. They have chosen subjective self-reports that are prone to invalidity and manipulation over objective measures like actual changes in activity or practical real-world measures like school attendance. Not very good science here. But they are asking parents to sign their children up.

What is promised to parents consenting to have the children enrolled in the trial?

The published protocol to which the investigators supposedly committed themselves stated

What are the possible benefits and risks of participating?
Participants will not benefit directly from taking part in the study although it may prove enjoyable contributing to the research. There are no risks of participating in the study.

Version 7 of the information sheet provided to parents, states

Your child may benefit from the treatment they receive, but we cannot guarantee this. Some children with CFS/ME like to know that they are helping other children in the future. Your child may also learn about research.

Survey assessments conducted by the patient community strongly contradict the suggestion that there is no risk of harm with GET.

alemAlem Matthees, the patient activist who obtained release of the PACE data and participated in reanalysis has commented:

“Given that post-exertional symptomatology is a hallmark of ME/CFS, it is premature to do trials of graded exercise on children when safety has not first been properly established in adults. The assertion that graded exercise is safe in adults is generally based on trials where harms are poorly reported or where the evidence of objectively measured increases in total activity levels is lacking. Adult patients commonly report that their health was substantially worsened after trying to increase their activity levels, sometimes severely and permanently, therefore this serious issue cannot be ignored when recruiting children for research.”

See also

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.

This thorough systematic review reports inadequacy in harm reporting in clinical trials, but:

Exercise-related physiological abnormalities have been documented in recent studies and high rates of adverse  reactions  to exercise have been  recorded in  a number of  patient surveys. Fifty-one percent of  survey respondents (range 28-82%, n=4338, 8 surveys) reported that GET worsened their health while 20% of respondents (range 7-38%, n=1808, 5 surveys) reported similar results for CBT.

The unpublished results of Dr. Esther Crawley’s SMILE trial

 A Bristol University website indicates that recruitment of the SMILE trial was completed in 2013. The published protocol for the SMILE trial

[Note the ® in the title below, indicating a test of trademarked commercial product. The significance of that is worthy of a whole other blog post. ]

Crawley E, Mills N, Hollingworth W, Deans Z, Sterne JA, Donovan JL, Beasant L, Montgomery A. Comparing specialist medical care with specialist medical care plus the Lightning Process® for chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME): study protocol for a randomised controlled trial (SMILE Trial). Trials. 2013 Dec 26;14(1):1.

States

The data monitoring group will receive notice of serious adverse events (SAEs) for the sample as whole. If the incidence of SAEs of a similar type is greater than would be expected in this population, it will be possible for the data monitoring group to receive data according to trial arm to determine any evidence of excess in either arm.

Primary outcome data at six months will be examined once data are available from 50 patients, to ensure that neither arm is having a detrimental effect on the majority of patients. An independent statistician with no other involvement in the study will investigate whether more than 20 participants in the study sample as a whole have experienced a reduction of ≥ 30 points on the SF-36 at six months. In this case, the data will then be summarised separately by trial arm, and sent to the data monitoring group for review. This process will ensure that the trial team will not have access to the outcome data separated by treatment arm.

A Bristol University website indicates that recruitment of the SMILE trial was completed in 2013. The trial was thus completed a number of years ago, but these valuable data have never been published.

The only publication from the trial so far uses selective quotes from child participants that cannot be independently evaluated. Readers are not told how representative these quotes, the outcomes for the children being quoted or the overall outcomes of the trial.

Parslow R, Patel A, Beasant L, Haywood K, Johnson D, Crawley E. What matters to children with CFS/ME? A conceptual model as the first stage in developing a PROM. Archives of Disease in Childhood. 2015 Dec 1;100(12):1141-7.

The “evaluation” of the quack Lightning Treatment in the SMILE trial and quotes from patients have also been used to promote Parker’s products as being used in NHS clinics.

How can I say the Lightning Process is quackery?

 Dr. Crawley describes the Lightning Process in the Research Ethics Application Form for the SMILE study as   ombining the principles of neurolinguistic programming, osteopathy, and clinical hypnotherapy.

That is an amazing array of three different frameworks from different disciplines. You would be hard pressed to find an example other than the Lightning Process that claimed to integrate them. Yet, any mechanisms for explaining therapeutic interventions cannot be a creative stir fry of whatever is on hand being thrown together. For a treatment to be considered science-based, there has to be a solid basis of evidence that these presumably complex processes fit together as assumed and work as assumed. I challenge Dr. Crawley or anyone else to produce a shred of credible, peer-reviewed evidence for the basic mechanism of the Lightning Process.

The entry for Neuro-linguistic programming (NLP) in Wikipedia states

There is no scientific evidence supporting the claims made by NLP advocates and it has been discredited as a pseudoscience by experts.[1][12] Scientific reviews state that NLP is based on outdated metaphors of how the brain works that are inconsistent with current neurological theory and contain numerous factual errors.[13][14

The respected Skeptics Dictionary offers a scathing critique of Phil Parker’s Lightning Process. The critique specifically cites concerns that Crawley’s SMILE trial switched outcomes to increase the likelihood of obtaining evidence of effectiveness.

 The Hampshire (UK) County Council Trading Standards Office filed a formal complaint against Phil Parker for claims made on the Lightning Process website concerning effects on CFS/ME:

The “CFS/ME” page of the website included the statements “Our survey found that 81.3 %* of clients report that they no longer have the issues they came with by day three of the LP course” and “The Lightning Process is working with the NHS on a feasibility study, please click here for further details, and for other research information click here”.

parker nhs advert
Seeming endorsements on Parker’s website. Two of them –Northern Ireland and NHS Suffolk subsequently complained that use of their insignias was unauthorized and they were quickly removed.

The “working with the NHS” refers to the collaboration with Dr. Easter Crawley.

The UK Advertising Standards Authority upheld this complaint, as well as about Parker’s claims about effectiveness with other conditions, including  multiple sclerosis, irritable bowel syndrome and fibromyalgia

 Another complaint in 2013 about claims on Phil Parker’s website was similarly upheld:

 The claims must not appear again in their current form. We welcomed the decision to remove the claims. We told Phil Parker Group not to make claims on websites within their control that were directly connected with the supply of their goods and services if those claims could not be supported with robust evidence. We also told them not to refer to conditions for which advice should be sought from suitably qualified health professionals.

 As we will see, these upheld charges of quackery occurred when parents of children participating in the SMILE trial were being vilified in the BMJ and elsewhere. Dr. Crawley was prominently featured in this vilification and was quoted in a celebration of its success by the Science Media Centre, which had orchestrated the vilification.

Captured cfs praker ad

The Research Ethics Committee approval of the SMILE trial and the aftermath

 I was not very aware of the CFS/ME literature, and certainly not all its controversies when the South West Research Ethics Committee (REC) reviewed the application for the SMILE trial and ultimately approved it on September 8, 2010.

I would have had strong opinions about it. I only first started blogging a little afterwards.  But I was very concerned about any patients being exposed to alternative and unproven medical treatments in other contexts that were not evidence-based – even more so to treatments for which promoters claimed implausible mechanisms by which they worked. I would not have felt it appropriate to inflict the Lightning Process on unsuspecting children. It is insufficient justification to put them a clinical trial simply because a particular treatment has not been evaluated.

 Prince Charles once advocated organic coffee enemas to treat advanced cancer. His endorsement generated a lot of curiosity from cancer patients. But that would not justify a randomized trial of coffee enemas. By analogy, I don’t think Dr. Esther Crawley had sufficient justification to conduct her trial, especially without warnings that that there was no scientific basis to expect the Lightning Process to work or that it would not hurt the children.

 I am concerned about clinical trials that have little likelihood of producing evidence that a treatment is effective, but that seemed designed to get these treatments into routine clinical care. it is now appreciated that some clinical trials have little scientific value but serve as experimercials or means of placing products in clinical settings. Pharmaceutical companies notoriously do this.

As it turned out, the SMILE trial succeeded admirably as a promotion for the Lightning Process, earning Phil Parker unknown but substantial fees through its use in the SMILE trial, but also in successful marketing throughout the NHS afterwards.

In short, I would been concerned about the judgment of Dr. Esther Crawley in organizing the SMILE trial. I would been quite curious about conflicts of interest and whether patients were adequately informed of how Phil Parker was benefiting.

The ethics review of the SMILE trial gave short shrift to these important concerns.

When the patient community and its advocate, Dr. Charles Shepherd, became aware of the SMILE trial’s approval, there were protests leading to re-evaluations all the way up to the National Patient Safety Agency. Examining an Extract of Minutes from South West 2 REC meeting held on 2 December 2010, I see many objections to the approval being raised and I am unsatisfied by the way in which they were discounted.

Patient, parent, and advocate protests escalated. If some acted inappropriate, this did not undermine the righteousness of others legitimate protest. By analogy, I feel strongly about police violence aimed against African-Americans and racist policies that disproportionately target African-Americans for police scrutiny and stoppng. I’m upset when agitators and provocateurs become violent at protests, but that does not delegitimize my concerns about the way black people are treated in America.

Dr. Esther Crawley undoubtedly experienced considerable stress and unfair treatment, but I don’t understand why she was not responsive to patient concerns nor  why she failed to honor her responsibility to protect child patients from exposure to unproven and likely harmful treatments.

Dr. Crawley is extensively quoted in a British Medical Journal opinion piece authored by a freelance journalist,  Nigel Hawkes:

Hawkes N. Dangers of research into chronic fatigue syndrome. BMJ. 2011 Jun 22;342:d3780.

If I had been on the scene, Dr. Crawley might well have been describing me in terms of how I would react, including my exercising of appropriate, legally-provided means of protest and complaint:

Critics of the method opposed the trial, first, Dr Crawley says, by claiming it was a terrible treatment and then by calling for two ethical reviews. Dr Shepherd backed the ethical challenge, which included the claim that it was unethical to carry out the trial in children, made by the ME Association and the Young ME Sufferers Trust. After re-opening its ethical review and reconsidering the evidence in the light of the challenge, the regional ethical committee of the NHS reiterated its support for the trial.

There was arguably some smearing of Dr. Shepherd, even in some distancing of him from the action of others:

This point of view, if not the actions it inspires, is defended by Charles Shepherd, medical adviser to and trustee of the ME Association. “The anger and frustration patients have that funding has been almost totally focused on the psychiatric side is very justifiable,” he says. “But the way a very tiny element goes about protesting about it is not acceptable.

This article escalated with unfair comparisons to animal rights activists, with condemnation of appropriate use of channels of complaint – reporting physicians to the General Medical Council.

The personalised nature of the campaign has much in common with that of animal rights activists, who subjected many scientists to abuse and intimidation in the 1990s. The attitude at the time was that the less said about the threats the better. Giving them publicity would only encourage more. Scientists for the most part kept silent and journalists desisted from writing about the subject, partly because they feared anything they wrote would make the situation worse. Some journalists have also been discouraged from writing about CFS/ME, such is the unpleasant atmosphere it engenders.

While the campaigners have stopped short of the violent activities of the animal rights groups, they have another weapon in their armoury—reporting doctors to the GMC. Willie Hamilton, an academic general practitioner and professor of primary care diagnostics at Peninsula Medical School in Exeter, served on the panel assembled by the National Institute for Health and Clinical Excellence (NICE) to formulate treatment advice for CFS/ME.

Simon Wessely and the Principal Investigator of the PACE trial, Peter White, were given free rein to dramatize their predicament posed by the protest. Much later, in the 2016 Lower Tribunal Hearing, testimony would be given by PACE

Co-Investigator Trudie Chalder would much later (2016) cast doubt on whether the harassment was as severe or violent as it was portrayed. Before that, the financial conflicts of interest of Peter White that were denied in the article would be exposed.

In response to her testimony, the UK Information Officer stated:

Professor Chalder’s evidence when she accepts that unpleasant things have been said to and about PACE researchers only, but that no threats have been made either to researchers or participants.

But in 2012, a pamphlet celebrating the success of The Science Media Centre started by Wessely would be rich in indiscreet quotes from Esther Crawley. The article in BMJ was revealed to be part of a much larger orchestrated campaign to smear, discredit and silence patients, parents, advocates and their allies.

Dr. Esther Crawley’s participation in a campaign organized by the Science Media Center to discredit patients, parents, advocates and supporters.

 The SMC would later organize a letter writing campaign to Parliament in support of Peter White and his refusal to release the PACE data to Alem Mattheees who had made a requestunder the Freedom of Information Act. The letter writing campaign was an effort to get scientific data excluded from the provisions of the freedom of information act. The effort failed and the data were subsequently released.

But here is how Esther Crawley described her assistance:

The SMC organised a meeting so we could discuss what to do to protect researchers. Those who had been subject to abuse met with press officers, representatives from the GMC and, importantly, police who had dealt with the  animal rights campaign. This transformed my view of  what had been going on. I had thought those attacking us were “activists”; the police explained they were “extremists”.

And

We were told that we needed to make better use of the law and consider using the press in our favour – as had researchers harried by animal rights extremists. “Let the public know what you are trying to do and what is happening to you,” we were told. “Let the public decide.”

And

I took part in quite a few interviews that day, and have done since. I was also inundated with letters, emails and phone calls from patients with CFS/ME all over the world asking me to continue and not “give up”. The malicious, they pointed out, are in a minority. The abuse has stopped completely. I never read the activists’ blogs, but friends who did told me that they claimed to be “confused” and “upset” – possibly because their role had been switched from victim to abuser. “We never thought we were doing any harm…”

 The patient community and its allies are still burdened by the damage of this effort and are rebuilding its credibility only slowly. Only now are they beginning to get an audience as suffering human beings with significant, legitimate unmet needs. Only now are they escaping the stigmatization that occurred at this time with Esther Crawley playing a key role.

Where does this leave us?

stop posterParents are being asked to enroll in a clinical trial without clear benefit to the children but with the possibility of considerable risk from the graded exercise. They are being asked by Esther Crawley, a physician, who has previously inflicted a quack treatment on their children with CFS/ME in the guise of a clinical trial, for which he is never published the resulting data. She has played an effective role in damaging the legitimacy and capacity of patients and parents to complain.

Given this history and these factors, why would a parent possibly want to enroll their children in the MAGENTA trial? Somebody please tell me.

Special thanks to all the patient citizen-scientists who contributed to this blog post. Any inaccuracies or excesses are entirely my own, but these persons gave me substantial help. Some are named in the blog, but others prefer anonymity.

 All opinions expressed are solely those of James C Coyne. The blog post in no way conveys any official position of Mind the Brain, PLOS blogs or the larger PLOS community. I appreciate the free expression of  personal opinion that I am allowed.

 

 

 

 

 

 

Hans Eysenck’s contribution to cognitive behavioral therapy for physical health problems: fraudulent data

  • The centenary of the birth of Hans Eysenck is being marked by honoring his role in bringing clinical psychology to the UK and pioneering cognitive behavior therapy (CBT).
  • There is largely silence about his publishing fraudulent data, editorial misconduct, and substantial undeclared conflicts of interest.
  • The articles in which Eysenck used fraudulent data are no longer cited much, but the influence of his claims which depended on these data remains profound.
  • Eysenck used fraudulent data to argue that CBT could prevent cancer and cardiovascular disease and extend the lives of persons with advanced cancer.
  • He similarly used fraudulent data to advance the claim that psychoanalysis is, unlike smoking, carcinogenic and has other adverse effects on health.
  • Ironically, Eysenck incorporated into his explanations for how CBT works elements of the psychoanalytic thinking that he seemingly detested.

If there is sufficient interest, a follow-up blog post will discuss:

  • Because of Eysenck’s influence, CBT in the UK exaggerates the role of early childhood adversity and much less to functional behavioral analysis than the American behavior therapy and cognitive behavior therapy.
  • Both CBT in the UK and some quack therapy approaches make assumptions about mechanism tied to Eysenck’s use of fraudulent data.
  • Consistent with Eysenck’s influence, CBT for physical problems in the UK largely focuses on self-report questionnaire assessments of mechanism of change and of outcome, rather than functional behavioral and objective physical health outcome variables.

8th-chocolate-happy-birthday-cake-for-HansHappy Birthday, Hans Eysenck

March 12, 2016 was the centenary of the birth of psychologist Hans Eysenck. The British Psychological Society’s  The Psychologist marked the occasion with release of a free app by which BPS members can access a collection of articles about Hans Eysenck from the archives.  Nonmembers can access the articles here.

The introduction to the collection, Philip Corr’s The centenary of a maverick states

Eysenck’s contributions were many, varied and significant, including: the professional development of clinical psychology; the slaying of the psychoanalytical dragon; pioneering behaviour therapy and, thus, helping to usher in the era of cognitive behavioural therapy…

Corr also wrote in the March 30 2016 Times Higher Education:

in defence corr

hans ensenck portraitThe articles collected in The Psychologist were written over many years. Together they present an unflattering picture of a controversial man who was shunned by his colleagues, blocked from getting awards, and who would humiliate those with whom he disagreed rather than acknowledge any contradictory evidence. Particularly revealing are Roderick Buchanan’s   Looking back: The controversial Hans Eysenck and a review of Buchanan’s book by Eysenck’s son Michael, Playing with fire: The controversial career of Hans J. Eysenck.

However, the collection stops short of acknowledging what was revealed in the early 90s in The BMJ: Eysenck knowingly published fraudulent data to back outrageous claims that CBT prevented cancer and extended the lives of patients with terminal cancer, whereas psychoanalysis was carcinogenic. He published his claims in journals he had founded, liberally self-plagiarizing and duplicate publishing with undeclared conflicts of interest. Eysenck received salary supplements and cash awards from German tobacco companies and from lawyers for the American tobacco companies for these activities.

slide 2 r smith should editors slide1 R Smith EysenckThe BMJ gave psychiatrists Anthony Pelosi and Louis Appleby a forum in the early nineties for criticizing Eysenck, even though the articles they attacked had been published elsewhere. The BMJ Editor Richard Smith followed up,  citing Eysenck as an example in raising the question whether editors should publish research articles in their own journal. Pelosi filed formal charges against Eysenck with the British Psychological Society. But, according to Buchanan’s book:

The BPS investigatory committee deemed it “inappropriate” to set up an investigatory panel to look into the material Pelosi had sent them, and henceforth considered the matter closed. Pelosi disagreed, of course, but was left with little recourse.

In an editorial in The Times Simon Wessely acknowledged Pelosi and Appleby’s criticism of Eysenck, but said “It would take more than a couple of psychiatrists to ruffle Eysenck.”

Simon on EysenckWessely suggested that the matter be dropped: the controversy was distracting everyone from the real progress being made in psychological approaches to cancer, like showing a fighting spirit extends the lives of cancer patients.  There was apparently no further mention in the UK press. Read more here.

Eysenck’s articles involving fraudulent data are seldom cited in the contemporary literature, but the claims the data were used to back remain quite influential. For instance, Eysenck claimed psychological factors presented more risk for cancer than many well-established biological factors. Including Eysenck’s data probably allowed one of the most cited meta-analyses of psychological factors in cancer to pass the threshold of hazard ratios strong enough for publication in the prestigious journal, Nature Clinical Practice: Oncology. Without the inclusion of Eysenck’s data, hazard ratios from methodologically weak studies cluster slightly higher than 1.0, suggesting little association that cannot be explained by confounds. A later blog post will document the broader influence of the Eysenck fraud on psychoneuroimmunology.

Eysenck’s claims concerning effects of CBT on physical health conditions now similarly go uncited.  However, the idiosyncratic definition he gave to CBT and his claims about the presumed mechanism by which it improved physical health pervade both CBT as defined in the UK and a number of quack treatments in the UK and elsewhere.

It is important to establish the connection between fraudulent data, distinctive features of CBT in the UK, and presumed mechanisms of action in order to open for re-examination the forms that CBT for physical health problems take in the UK and the way in which claims of efficacy are evaluated.

Fraudulent Data

Eysenck repeated tables and text in a number of places, but I will mainly draw on data as he presented them in the journal he founded, Behaviour Research and Therapy [1,   2], which correspond with what he presents elsewhere.

Eysenck’s Croatian collaborator Grossarth-Maticek conducted the therapy and collected the predictor and outcome data. A personality inventory  was used to classify participants receiving therapy into four types , a cancer-prone type (Type 1), a coronary heart disease (CHD)-prone type (Type 2), and 2 healthy types (Type 3 and Type 4). The typology was derived from quadrants in a 2×2 dichotomization of high versus low and rationality versus anti-emotionality, quite different from the dimensions and item content of the Eysenck Personality Questionnaire. Indeed, Roderick Buchanan noted in his biography that “Eysenck had struggled to banish typological concepts in favour of continuous dimensions for most of his career.” Grossarth-Maticekis questionnaire and typology has been sharply criticized later by Eysenck son Michael, among many others.

Eysenck and Grossarth-Maticek reported results of individually delivered “creative novation behaviour therapy”:

… Effects of prophylactic behaviour therapy on the cancer-prone and the CHD-prone probands respectively after 13 yr. It will be clear that treatment by means of creative novation behaviour therapy has had a highly significant prophylactic effect, preventing deaths from cancer in probands of Type 1, and death from coronary heart disease in probands of Type 2.

table 3 prophylactic effectsFor creative novation behaviour therapy delivered in a group format:

It will be seen that both cancer and CHD mortality are very significantly higher in the control group, as is death from other causes. Incidence rates are also very significantly higher in the control group for cancer, but with a difference below our selected P = 0.01 level of significance for CHD. Most telling is the difference regarding those ‘still living’-79.9% in the therapy group, 23.9% in the control group. The results of the group therapy study support those of the individual therapy group in demonstrating the value of behaviour therapy in preventing death from cancer and CHD, and in lowering the incidence from cancer and possibly from CHD.

table 4 group therapyStrong effects were reported even when the treatment was delivered as a discussion of a brief pamphlet. The companion paper  described this bibliotherapy and provided the pamphlet as an appendix,  which is reproduced here.

This statement is given to the proband, who also receives an introductory 1-hr treatment in which the meaning of the statement is explained, application considered, and likely advantages discussed. After the patient has been given time to consider the statement, and apply it to his/her own problems, the therapist spends a further 3-5 hr with the patient, suggesting specific applications of the principles in the statement to the needs of the patient, and his/her particular circumstances.

Six hundred probands received the bibliotherapy and a control group of 500 matched for personality type, smoking, age and sex received no treatment. Another 100 matched patients received a placebo condition in which they met with interviewers to discuss a pamphlet with “psychoanalytic explanation and suggestions.”

I encourage readers to take a look at the pamphlet, which is less than a page long. It ends with:

The most important aims of autonomous self-activation: your aim should always be to produce conditions would make it possible for you to lead a happy and contented life.

The results were:

There are no statistically significant differences between the control group and the placebo group, which may therefore be combined and considered a single control group. Compared with this control group, the treatment group fared significantly better. In the control group, 128 died of cancer, 176 of CHD; in the treatment group only 27 died of cancer, and 47 of CHD. For ‘death from other causes’, the figures are 192 and 115. Clearly the bibliographic method had a very strong prophylactic effect.

table 5 group and biblioEysenck and Grossarth-Maticek reported numerous other studies, including one in which 24 matched pairs of patients with inoperable cancer were assigned to either creative novation behaviour therapy or a control group. The patients receiving the behaviour therapy lived five years versus the three years of those in the control group, a difference which was highly significant.

Keep in mind that in these studies that all of the creative novation behaviour therapy sessions were solely provided by Grossarth-Maticek.

But let’s jump to a final in a series of tables constructed to make the argument that psychoanalysis was harmful to physical health.

We are here dealing with three groups. Group I is constituted of patients who terminated their  psychoanalytical treatment after 2 yr or less, and were then treated with behaviour therapy.

Group 2 is a control group matched with the members of group I on age, sex, smoking and personality type. Group 3 is a control group which discontinued psychoanalysis, like Group I, but did not receive behaviour therapy. Members of Group I and 2 do not differ significantly in mortality, but Group 3 has significantly greater mortality than either. Looking again at the percentage of patients still living, we find for Group 1 92, 95 and 95%, for Group 2 96, 89 and 95%, for Group 3 the figures are: 72, 63 and 61%. Clearly behaviour therapy can reverse the negative impact psychoanalysis has on survival.

table 15 psychoanalysisIn a number of places, this is explained in identical words:

Theoretically, this conclusion is not unreasonable. We have shown that stress is a powerful factor in causing cancer and CRD, and it is widely agreed, even among psychoanalysts, that their treatment imposes a considerable strain on patients. The hope is often expressed that finally the treatment will resolve these strains, but there is no evidence to suggest that this is true (Rachman & Wilson, 1980; Eysenk & Martin, 1987). Indeed, there is good evidence that even in cases of mental disorder psychoanalysis often does considerable harm (Mays & Franks, 1985). A theoretical model to account for these negative outcomes of psychoanalysis and psychotherapy generally has been presented elsewhere (Eysenck, 1985); it would apply equally well in the psychosomatic as in the purely psychiatric field.

dog breakfastCBT for physical health problems: a dog’s breakfast approach

Grossarth-Maticek had already formulated his approach and delivered all psychotherapy before Eysenck began co-authored papers and promoting him. In a 1982 article without Eysenck as an author, Grossarth-Maticek is quite explicit about the psychoanalytic theory behind his approach:

A central proposition of our research program is that cancer patients are either preoccupied with traumatic events of early childhood or with excessive expectations of the parents during their whole life. They are characterized by intensive internal inhibitions toward expressing feelings and desires. Therefore, we speak of a chronic blockade of expression of feelings and desires. We assume that parents of cancer patients did not respond adequately to the child’s cries for help and these children were obliged very early to do non-conforming daily task. Cancer patients have never learned to express persistent cries for help…

The specific family dynamics in the special educational pattern which block hysterical reactions determine the behavior, which in turn is characterized by excessive persistence of performance of the daily task, disregard of symptoms and lack of aggressiveness in behavior. Through the currents of negative life events (i.e., death of closely connected persons) expressions of loneliness and reactive depression can appear intensively and chronically.

If this is not clear enough:

In our approach we try not to deny the psycho analytic propositions but to integrate the psychoanalytic research program with social psychological and sociological factors, hereby assuming that they have interactive effects on carcinogenesis.

Strangely, Grossarth-Maticek suggests in this article, that the psychoanalytic factors interact with “organic risk factors such as cigarette smoking in the case of lung cancer.” Grossarth-Maticek and Eysenck would soon be receiving tens of thousands of dollars in support from the German tobacco companies and lawyers from the American tobacco companies to promote the idea that personality caused smoking and lung cancer, but any connection between smoking and lung cancer was spurious. Product liability suits against tobacco companies should therefore be dismissed.

In the articles co-authored by Grossarth-Maticek and Eysenck, these roots of what Eysenck repackaged as creative novation behaviour therapy are only hinted at, but are noticeable to the observant reader in references to the role of dependency and autonomy. Fraudulent data are mustered to show the powerful positive effects of this behaviour therapy versus the toxicity of psychoanalysis.

On page 8 of this article, ten  explicitly labeled behavioural techniques are identified as occurring across individual, group, and bibliotherapy:

  • Training for reduction of the planned behaviors initiation of autonomous behavior.
  • Training for cognitive alteration under conditions of relaxation
  • Training for alternative reactions.
  • Training for the integration of cognition, emotionality and intuition.
  • Training to achieve stable expression of feelings.
  • Training for potentiating social behavioral control
  • Training to suppress stress-creating ideas
  • Training to achieve a behavior-directing hierarchic value structure
  • Training in the suppression of stress-creating thought.
  • Abolition of dependence reactions.

This approach has only superficial resemblance to American behavioral therapy and CBT. The emphasis on expression of emotional feelings and abolition of dependent reactions is incomprehensible when it is detached from its psychoanalytic roots. The paper refers to behavioral analysis, but interviews about the past, including childhood experiences are emphasized, rather than applied behavioral analysis. The hierarchies of behavior do not correspond to operant approaches, but to a value structure of autonomy versus dependence.

There is also considerable reference to the use of hypnosis to achieve these goals.

In short, neither the goals nor the methods have much relationship to learning theory at the time that Eysenck was writing nor to contemporary developments in operant conditioning. His approach is a tortured extension of classical conditioning. Outside of the fraudulent data that Grossarth-Maticek developed and that he published with Eysenck, there is little basis for assuming that psychological factors were related to physical health in the way the treatment approach postulated.

It should be kept in mind that Eysenck was not a psychotherapist. He actually detested psychotherapy and generated considerable controversy earlier by arguing that any apparent effects of psychotherapy were due to natural remission. It should also be noted that Eysenck was claiming creation novation behaviour therapy modified personality traits, even when delivered in a brief pamphlet, in ways that could not be anticipated by his other writings about personality. Finally, the particular personality characteristics that Eysenck was talking about modifying were very different than what he assessed with the Eysenck Personality Inventory.

Only “controversial” and “too good to be true” or fraud?

 Before Eysenck began collaborating with Grossarth-Maticek, there was widespread doubts about the validity of Grossarth-Maticek’s work.  In 1973, Grossarth-Maticek’s work had been submitted to the University of Heidelberg as a Habilitation, a second doctoral degree required for a full professorship. It was rejected. One member of the committee, Manfred Amelung, declared the results “too good to be true.” He retained a copy and would later put his knowledge of its details into a devastating critique. According to Buchanan’s biography, Eysenck demanded of Grossarth-Maticek “you must let me check your data, for if you deceive me I will never forgive you.”

Eysenck gained access to the data set, sometimes directing reanalyses by Grossarth-Maticek and his statistician. Other analyses were done by Eysenck’s statisticians in London. Eysenck’s biographer Buchanan noted “there were ample opportunities to select, tease out, or redirect attention – given a data set that was apparently sprawling chaotic but rich and ambitious….From the mid-1980s, Eysenck did virtually all of the writing for publication in English and presumably exerted a strong editorial control.” Buchanan also notes that tobacco companies became skeptical of the strength of findings that were reported, but also their inconsistency. They refused to continue to support Eysenck unless an independent team was set up to check analyses and the conclusions that Eysenck was drawing from them.

Eysenck single-authored a target article for Psychological Inquiry that reproduced many of the tables that we have been discussing. More than a dozen commentators included the members of the independent team, but also others who did not have access to the data, but who examined the tables with forensic attention. The commentary started off with Manfred Manfred Amelung who made use of what he had learned from Grossarth-Maticek’s doctoral work.

Many of the commentators suggested that the intervention studies presented conclusions that were “too good to be true,” not only in terms of the efficacy claim for the intervention, but for the negative outcomes claimed for the control group. But other commentators pointed to gross inconsistencies across different reports in terms of methods and results, clear evidence of manipulation of data, including some patients being counted a number of times, other patients dying twice, Eysenck and Grossarth-Maticek’s improbable ability to obtain matching of intervention patients and controls, and too perfect predictions. In the end, even Grossarth-Maticek’s Heidelberg statistician expressed concerns that there had been tampering with the data.

Both Grossarth-Maticek and Eysenck got opportunities to respond and were defensive and dismissive of the overwhelming evidence of exaggeration of the results and even fraud.

The exchanges in Psychological Inquiry occurred over two issues. Taken together, the critical commentaries are devastating, but the criticisms became diffuse because commentators focused on different problems. It took a more succinct, pithy critique by Anthony Pelosi and Louis Appleby in The BMJ to bring the crisis of credibility to a head.

Anthony Pelosi and Louis Appleby in The BMJ

 In the first round of their two-part attack, Pelosi and Appleby centered on Eysenck and Grossarth-Maticek’s  two articles in Behaviour Research and Therapy, but referenced the critiques in Psychological Inquiry. The incredible effectiveness of these two psychiatrists depended largely on their pointing  out what was hiding in plain sight in the two Behaviour Research and Therapy articles. For instance:

After 13 years, 16 of 50 untreated type 1 subjects had died of a carcinoma. Not one of the 50 cancer prone subjects receiving the psychotherapy died of cancer. The therapy was a genuine panacea, giving equivalent results for type 2 subjects and heart disease. The all cause mortality was over 60% in untreated and 15% in treated subjects. The death rate in the untreated subjects was truly alarming as they began the trial healthy and most were between 40 and 60 years of age.

I encourage readers to compare the Pelosi and Appleby paper to the tables I presented here and see what they missed.

Pelosi and Appleby calculated the effort required by Grossarth-Maticek if he had – as Eysenck insisted- single-handedly carried out all of the treatment.

It is striking that all the individual and group therapy was given by Professor Grossarth-Maticek. The trials were undertaken between 1972 and 1974 and involved 96 subjects (or perhaps 192 subjects, see below) in at least 20 hours of individual work, and at least 10 groups (245 subjects with 20-25 in each) for six to 15 sessions each. Add to this Grossarth-Maticek’s explanatory introduction to bibliotherapy for 600 people, and it can be seen that the amount of time spent by this single senior academic on his experimental psychotherapies is huge and certainly unprecedented.

They summarized inconsistencies and contradictions reported in the Psychological Inquiry, but then added their own observation that a matching of 192 pairs of intervention and control patients had only produced a sample of 192! They suggested that in the two Behaviour Research and Therapy articles there were at least  “10 elaborate misprints or misstatements in the description of the methods” that the editor or reviewers should have caught.

At no point, does the word “fraud” or “fraudulent” appear in Pelosi and Appleby’s first article. Rather, they suggest that  “Eysenck and Grossarth-Maticek… are:

making claims which, if correct, would make creative novation therapy a vital part of public health policy throughout the world.”

They conclude with

For these reasons there should be a total reexamination and proper analysis of the original data from this research in an attempt to answer the questions listed above. The authors give their address as the Institute of Psychiatry in London, which must be concerned about protecting its reputation. Therefore the institute should, in our view, assist in this clarification of the meaning of the various studies. There should also be some stern questions asked of the editors of the various journals involved, especially those concerned among the editorial staff of Behaviour Research and Therapy who, in our opinion, have done a disservice to their scientific disciplines, and indeed to Professors Eysenck and Grossarth-Maticek, in allowing this ill considered presentation of research on such a serious topic.

Eysenck’s reply and Pelosi and Appleby’s response

 Readers can consult Eysenck’s reply  for themselves, but it strikes me as evasive and dismissive. Specific criticisms are not directly answered, but Eysenck points to consistency between his results and those of David Spiegel, who had claimed to get even stronger effects in his small study of supportive expressive therapy for women with metastatic breast cancer. Rather than demolishing the credibility of his work with Grossarth-Maticek, Eysenck argues that Pelosi and Appleby only point to the need for funding of a replication. Eysenck closes with:

Their critical review, however incorrect, full of errors and misunderstandings, and lacking in objectivity, may have been useful in drawing attention to a large body of work, of both scientific and social relevance, that has been overlooked for too long.

Pelosi and Appleby took Eysenck’s reply as an opportunity to get even more specific in the criticisms:

We are accused of being vague in mentioning many errors, inappropriate analyses, and missing details in the publications on this research programme. We value this opportunity to be more specific, to clarify just a few of the questions raised by ourselves and others, which Eysenck has failed to answer, and to outline additional findings from these authors’ investigations.

After a detailed reply, they wrap up with references to the criticisms that Eysenck received in Psychological Inquiry, in an ironic note, turning Eysenck’s attacks on proponents of the link between smoking and lung cancer on to Eysenck himself:

Our concern has been to clarify the methods and analyses of a body of research which, if accurate, would profoundly influence public health policies on cancer and heart disease. Other critics have been more challenging in what they have alleged, and in our opinion the controversy which now surrounds one of academic psychology’s most influential figures constitutes a crisis for the subject itself. The seriousness of the detailed allegations by van der Ploeg, although refuted by Eysenck and Grossarth-Maticek, should in themselves prompt these authors to reexamine their own findings after appropriate further training in the methodology of medical research. Perhaps the most skilfully worded criticism on this subject was made not about Eysenck but by him in a debate on the relation between smoking and cancer. In disputing the findings of Doll and Hill’s epidemiological studies on this association he comments: “What we have found are serious methodological weaknesses in the design of the studies quoted in favour of these theories, statistical errors, and unsubstantiated extrapolations from dubious data to unconfirmed conclusions.” Eysenck owes it to himself and to his discipline to reconsider critically his own work on this subject.

In the over 20 years since this exchange, Pelosi and Appleby and their ally editor Richard Smith of The BMJ failed to get an appropriate response from the British Psychological Society, King’s College London or the Institute of Psychiatry, the journal Behaviour Research and Therapy, or the Committee on Publication Ethics (COPE). This situation demonstrates the inability of British academia to correct bad and even fraudulent science. It stands as a cautionary note to those of us now attempting to correct what we perceive as bad science. Efforts are likely to be futile. On the other hand, the editorship of Behaviour Research and Therapy has passed to an American, Michelle Craske, a professor at UCLA. Perhaps she can be persuaded to make a long overdue correction to the scientific record and remove a serious blemish on the credibility of that Journal.

If there is sufficient interest, I will survey the profound influence of the fraudulent work of Eysenck and Grossarth-Maticek in a future blog post.

  • Because of their influence, CBT in the UK gives an exaggerated emphasis to early childhood adversity and much less to functional behavioural analysis than the American behavior therapy and CBT.
  • Consistent with Eysenck’s influence, CBT for physical problems in the UK largely focuses on self-report questionnaire assessments of mechanism of change and of outcome, rather than functional behavioral and objective physical health outcome variables.

Influences can also be seen in:

Contemporary CBT for physical conditions as practiced in UK, including CBT for irritable bowel syndrome (IBS), fibromyalgia, and other “all in the head” conditions that are deemed Medically Unexplained Symptoms (MUS) in the UK, as in PRINCE trial of Trudie Chalder and Simon Wessely.

The “psychosomatic” approach as seen in neurologist Suzanne O’Sullivan’s  recent editorial in The Lancet and her “It’s All in Your Head”, which won the 2016 Wellcome Book Award and her.

Quack treatments, such as Phil Parker’s Lightning Process, which the UK’s Advertising Standards Authority (ASA) ruled against advertising its effectiveness in treatment of chronic fatigue syndrome/ myalgic Encephalopathy,  multiple sclerosis, and irritable bowel syndrome/digestive issues. The Lightning Process is nonetheless implemented in the UK NHS under the direction of University of Bristol Professor Esther Crawley 

Quack cancer treatments such as Simonton visualization method.

More mainstream, but unproven psychological treatments for cancer including David Spiegel’s supportive expressive therapy. Neither Spiegel –nor anyone else– has ever been able to replicate the finding praised by Eysenck, but repeats his claims in a recent non-peer reviewed article in the UK-based Psycho-Oncology and with a closely related article in BPS’ British Journal of Health Psychology.

More mainstream, but unproven psychological approaches to cancer that claim to improve immune functioning by reducing stress.

Some Scottish readers will understand this message concerning Eysenck’s fraud: The ice cream man cometh.

My usual disclaimer: All views that I express are my own and do not necessarily reflect those of PLOS or other institutional affiliations.

“Understanding Psychosis and Schizophrenia” and mental health service users

understand coverDoes Understanding Psychosis and Schizophrenia exploit, disrespect, and marginalize service users?

Genre confusion.

The 180-page Understanding Psychosis and Schizophrenia produced by the British Psychological Society Division of Clinical Psychology is a puzzling document. We need to know its genre to decide what standards we apply in evaluating it. The authors tell us:

The report is intended as a resource for people who work in mental health services, people who use them and their friends and relatives, to help ensure that their conversations are as well informed and as useful as possible. It also contains vital information for those responsible for commissioning and designing both services and professional training, as well as for journalists and policy-makers. We hope that it will help to change the way that we as a society think about not only psychosis but also the other kinds of distress that are sometimes called mental illness.

“Well-informed” by what or whom? How is the information “vital”? Does “vital” assume “trustworthy” and “credible”?

As I will cover in a later blog issue, the document strikingly lacks the transparency that it would need to be taken seriously.  Understanding Psychosis conforms to none of the well-defined processes and standards – checks and balances – expected to be met by professional organizations producing a report aimed at policy-makers and the general public.

mental ellf1For now, note these psychologists did not engage other professionals with complementary viewpoints and expertise. And the writing  was closed to anyone not already expressing strongly held particular opinions. When critics nonetheless provided a detailed analysis of some crucial points at the popular blog, Mental Elf, the authors of Understanding Psychosis retweeted and favorited a denunciation of them as a “circle jerk,” i.e., mutually masturbating.

circle jerk
Please click to enlarge

how vulgar

Key stakeholders were simply excluded – primary care physicians, social workers, psychiatrists, police and corrections personnel who must make decisions about how to deal with disturbed behavior, and –most importantly- the family members of persons with severe disturbance. There was no check on the psychologists simply slanting the document to conform to their own narrow professional self-interests, which we are asked to accept as “expertise.”

Is Understanding Psychosis evidence-based?

Understanding Psychosis occasionally cites some empirical findings, but can’t be seen as evidenced-based. That would require transparent, systematic strategies for gathering, interpreting, and integrating evidence that are simply not there.

Indeed, I think it is an excellent document for PhD students and trainees to practice debunking the creation of false authority by selective citation and miscitation and ignoring of contradictory studies. I suggest that they arm themselves with Google Scholar and tools provided in

Greenberg, S. A. (2009). How citation distortions create unfounded authority: analysis of a citation network. BMJ, 339.

Then start checking the citations provided for seemingly evidence-based statements in Understanding Psychosis. Ask questions like “What relevant studies are not cited? What studies are misinterpreted or simply cited for findings they did not contain?” Go to Google Scholar or Web of Science and find out.

For instance, take the opinion

In view of the problems with diagnoses, many researchers and clinicians are moving away from using them, and recent high-profile reports have recommended this. 55 56.

Check the references and see that the authors of Understanding Psychosis are the “many researchers and clinicians.” They are praising their own opinion pieces as “high-profile.”

55. British Psychological Society (2013). Division of Clinical Psychology position statement on the classification of behaviour and experience in relation to functional psychiatric diagnoses: time for a paradigm shift. Leicester: British Psychological Society.

56. Division of Clinical Psychology (2011). Good practice guidelines on the use of psychological formulation. Leicester: British Psychological Society.

The authors of Understanding Psychosis would have embarrassed themselves if they stated outright “It is our opinion that…and we consider our opinion high-profile and you should be duly impressed.” They depend on readers not checking references.

Argument from cherry picked quotes.cherrypicking

Understanding Psychosis is a collection of quotes. We might be inclined to interpret this as a strength, a sign of collaborative  participatory research.

Or maybe this represents qualitative research allowing  people to speak for themselves, rather than requiring that their experiences be processed through others’ filters and concepts.  But bona fide, credible qualitative research requires that biases of  investigators not intrude upon what they report.  Some controls must be visibly present preventing the investigators from doing so.

Quotes are carefully selected to support by the psychologists opinions expressed before the document was prepared – like 15 years ago in their Recent Advances in Understanding Mental Illness and Psychotic Experiences.

Many quotes are not from people suffering from schizophrenia. In most instances, we are not given sufficient information to determine this.  The authors systematically withhold information that would allow readers to determine who is and who is not a service user.

In this issue of Mind the Brain, I examine implications of this heavy dependence on these particular quotes. I will question whether Understanding Psychosis involves using and even exploiting service users, pitting more highly functioning ones against those who are functioning less well and their families who have to deal with them when they cannot take care of themselves.

Where do the quotes in Understanding Psychosis come from?

Some quotes were simply pasted in from the 2000 Recent Advances in Understanding Mental Illness and Psychotic Experiences.

Presumably people had relevant experience in the interim for our grasping the relevance to what it like living with schizophrenia and other psychoses – if that was actually their circumstances. Unfortunately, no follow-up is provided. The authors did not respond to repeated inquiries to asking whether they even obtained permission to use these quotes.

The quotes also have been trimmed of most details about their context that are available in original sources. Going to the original sources, we find the sources deliberately sampled people who were not service users.

Yup, people stripped of their identities are paraded out without the benefit of information that would render their experiences meaningful. Readers can’t independently assessment the uses to which the psychologist authors of Understanding Psychosis put these quotes.

What is not at issue is whether people with unusual experiences can get our attention when they talk about them. What is at issue is that a group of professionals take these quotes out of context and insist that they be accepted as the primary basis for – as their title states – our understanding of psychosis and schizophrenia.

Some of the quotes come from sources like

Jackson, L., Hayward, M. & Cooke, A. (2011). Developing positive relationships withvoices: A preliminary grounded theory. International Journal of Social Psychiatry, 57(5), 487–495.

Freeman, D., Garety, P.A., Bebbington, P.E., Smith, B., Rollinson, R., Fowler, D. et al. (2005). Psychological investigation of the structure of paranoia in a non-clinical population. British Journal of Psychiatry, 186, 427–435.

Heriot-Maitland, C., Knight, M. & Peters, E. (2012). A qualitative comparison of psychotic-like phenomena in clinical and non-clinical populations. British Journal of Clinical Psychology, 51(1), 37–53.

Jackson et al report

Five men and seven women were recruited through local NHS services, community advertisement and the local branch of the Hearing Voices Network.

Freeman  et al report

An anonymous internet survey [was]… e-mailed the address of a website where they could take part in a survey of ‘everyday worries about others’.

Heriot-Maitland et report interviewing 12 participants, who reported “psychotic-like ‘out-of-the-ordinary’ experience (OOE) in the past five years.”

The quotes come from persons who are lucid enough to be recruited for small studies of , highly selected articulate persons. They certainly don’t display the distorted thought and behavior disorder and simple incoherence of many people with acute and chronic schizophrenia.

I agree with the Understanding Psychosis authors that few people who have ‘psychotic-like’ experiences meet criteria for a diagnosis of schizophrenia. But should we accept a carefully cherry-picked and edited group of quotes as the basis for revising our understanding of people who do meet criteria?

A number of quotes sound like people who are high functioning and showing an unusual degree of  fantasy-proneness:

P 29 I work four days a week in a professional job; I own my own house and live happily with my partner and pets. Occasionally I hear voices – for example when I have been particularly stressed or tired, or I have seen visions after a bereavement. Knowing that many people hear voices and live well, and that some cultures see these experiences as a gift, helps me to never catastrophise or to worry that it may be the start of a breakdown. Although I am lucky that the experiences have never been as upsetting as some people’s, if someone had told me it was madness I could have got into a vicious cycle and struggled to get out.

Some of the quotes seem to represent clinically significant distress, but probably not psychosis or schizophrenia.

p 53 One thing that you might hear a lot about is that anxiety is a trigger of suspicious thoughts. I have never been that good at recognising my own anxiety. Quite a high level of anxiety is pretty normal for me. So normal that I wouldn’t normally do anything about it, but I now recognise that it sets the background for the expected potential threats in any situation, and so the suspicious thoughts and ideas of reference can pop right in there. I find people as having the most potential as a source of threat and because of that I am prone to suspicious thoughts about others. So now what I do is try to address the level of anxiety I feel in these situations. Adam

We’re not provided any information suggesting this suspiciousness is the psychotic symptom, paranoia.

P 43 After being almost killed by my ex-boyfriend when I was 16 I have had OCD. I have also developed paranoia about someone trying to kill me. If I have conflict with someone over anything I worry they are going to kill me or have someone come and kill me. I wake up worried someone is in my bedroom. I think about trying to be ready to protect myself if someone comes at me. I don’t think I would have this if I had not been traumatised half my life ago.  Josephine

Yale Professor Joan Cook and other colleagues and I recently published  a mixed method study of a national sample of psychotherapists providing residential treatment to veterans for posttraumatic stress disorder.  A number reported difficulties deciding whether the “voices” that some veterans describe represented schizophrenia or vivid re-experiencing symptoms consistent with posttraumatic stress syndrome, for which exposure therapy is indicated.

The authors of Understanding Psychosis express a clear disdain for making diagnostic distinctions. But,  it is important for clinicians to decide about the nature of clients’ distress in order to decide how to treat it. They best do so by formulating a hypothesis based on evidence tied to diagnoses, and then sympathetically probing.  Gradual exposure to past trauma would likely tame the distress of someone meeting criteria for PTSD. But this could prove absolutely terrifying and decompensating for someone whom additional information suggested a diagnosis of psychosis. So clinicians have to have some evidence-based ideas to probe and make decisions or proceed blindly.

Some quotes probably refer to brief psychotic reactions. Responding to Understanding Psychosis, Allan Frances noted

Brief psychosis is considered a mental disorder, but it is just a transient one with excellent prognosis and no reason to expect long-term impairment. The symptoms emerge suddenly in response to stress and usually disappear just as suddenly (especially if the stress is removed), often never to reappear. This is common in many cultures, and I have seen it fairly often in college students away from home for the first time, in travelers in strange lands, and in people who have had something terrible happen to them. Antipsychotic medicine is needed only briefly, if at all.

Quotes were selected to fit the authors’ conviction that what other professionals call psychosis or schizophrenia is an understandable reaction to life events. But if we go to the larger literature, the associations between adverse experiences and psychosis, even in a meta-analysis of one of the authors of Understanding Psychosis, are not large enough that would suggest such strong causality.  Adverse experiences are linked to lots of negative outcomes, but generally do not lead to psychosis or schizophrenia, even if there is a significant, but not overwhelming correlation.

Understanding Psychosis is not a transparent, systematic review of available evidence. Authors are mustering quotes to fit their preconceived notions. And leaving out quotes and details that don’t fit.

American psychiatrist Bernard “Barney” Carroll slammed the arrogant response of President of the American Psychiatric Association President  Jeffrey Lieberman to media coverage of Understanding Psychosis. Barney called it over-the-top” and a “disservice to psychiatry.” Yet, this was not before he nailed the report for its “domesticating psychosis”:

Hallucinations become the experience of hearing voices; delusions become the experience of unusual beliefs; paranoid thinking becomes the experience of anxiety – never mind that the great majority of patients with clinical anxiety disorders are not at all paranoid in the way that psychotic patients are. They also make much of the fact that milder forms of these “experiences” are common in the general population – as are milder forms of many clearly medical symptoms. In short, they fail to acknowledge the state transition that demarcates mild or prodromal symptoms from outright psychotic illness.

… The BPS document fails adequately to convey the range of symptoms and associated behaviors in psychosis/schizophrenia. Even when these are mentioned, they are not addressed in a way that matches their clinical salience. Thus, decompensating psychotic crises are discussed unhelpfully in the framework of poor sleep habits. Acute inpatient psychiatric units are discussed in a patronizing way and are faulted as being unhelpful for some patients – never mind their rescue function. Catatonia as a common feature is not acknowledged. Psychotic terror and panic are not acknowledged. Formal thought disorder with truly crazy speech is not acknowledged.

A disclosure of my past.

I’m struck by the huge gap between the clear, articulate statements in the quotes provided in Understanding Psychosis and the incoherent mumbling and sometimes raging of people who are acutely psychotic.  I wonder how many of the authors have ever tried to conduct an interview with someone in that state.

cowboy entering belgium-1-page-001My clinical training involved six years of live supervision at the Mental Health Institute (MRI)provided by professionals widely recognized for their innovative work in analyzing the communication of persons considered as having schizophrenia –  Paul Watzlawick, John Weakland, and Richard Fisch – although they would have objected to that diagnostic label.

At the time, I probably was more anti-diagnosis than many of the authors of Understanding Psychosis are today. But then as Director of Research at Mental Research Institute, I witnessed the disaster of its Soteria Project. I’ll leave that for another time, but Wikipedia states

The Soteria project was admired by many professionals around the world who aspired to create mental health services based on a social, as opposed to a medical, model. It was also heavily criticized as irresponsible or ineffective. The US Soteria Project closed as a clinical program in 1983 due to lack of financial support, although it became the subject of research evaluation with competing claims and analysis. Second generation US successors to the original Soteria house called Crossing Place is still active, although more focused on medication management.

While Paul, John, and Dick were widely recognized for their work analyzing communication with severely disturbed persons, they operated with a sense that at some point the disturbance of thought and behavior could became too much to carry on a discussion. And talking to highly disturbed persons, they knew not to take what was being said literally.

Who was selected for inclusion in Understanding Psychosis and who was excluded and left silent?

Many patients with acute and chronic psychosis are essentially nonverbal and cannot communicate their distress. Sure, they can’t provide coherent quotes for the psychologists who assembled Understanding Psychosis, but it is irresponsible for those psychologists to pretend these people don’t exist or that the quotes they assembled represent their best interest.

Many patients who meet criteria for schizophrenia will times be unable to take care of themselves or to make basic decisions.  The burden of caring and decision-making will fall on family members if they are available. The alternative for persons with schizophrenia is to become homeless or go to jail or prisons because more appropriate beds and hospitals are not available. Nowhere in Understanding Psychosis are we reminded that persons with schizophrenia sometimes need sanctuary in hospitals.

Nowhere are we reminded that 10% of persons with schizophrenia will die by suicide. There is recent evidence that psychotic people may account for nearly 1/3 of suicide attempts with intent to die.

If I were a family member of someone with schizophrenia, I would be damn angry at the gap between the quotes in Understanding Psychosis what I knew about the person for whom I had to provide care. I’d also be angry that no one in my situation had been invited to participate as a stakeholder.

Psychologists in search of opportunities to work with YAVIS clients

purchase of friendshipThe carefully selected quotes suggest people who would be more satisfying to work with than many persons with psychosis and schizophrenia. Reading them, I was immediately reminded of William Schofield[‘s  50-year-old book Psychotherapy: The Purchase of Friendship in which he lamented the strong tendency of mental health professionals wanting to work with the YAVIS: clients who are young, attractive, verbal, intelligent, and successful.  One of the authors of Understanding Psychosis also co-authored the widely misrepresented Lancet study of cognitive behavioral therapy for psychosis and could tell us how difficult and ineffective  it was doing  therapy in  that study with the older patients who had more psychotic  episodes.

Despite the authors of the Lancet study having distanced themselves from earlier claims showed cognitive therapy had effects equivalent to antipsychotic medication, authors of Understanding Psychosis persist in making the claim to service users:

It would also appear that CBT can bring comparable benefits even when people choose not to take medication.

As we would expect from recommendations produced by tightly knit groups representing single professions, Understanding Psychosis is a bid for more resources for its authors to work with clients with whom they want to work.  But like any policy recommendations, we need to examine the evidence and look at where those resources would come.

Please click to enlarge
Please click to enlarge

I’ll leave that discussion to another blog post, but take a look at the graph on the left. It represents the dramatic shift in resources from inpatient beds to outpatient treatment settings. The profoundly disturbed persons who need those beds would undoubtedly be less suitable for the conversations that the Understanding Psychosis psychologists want to be having. The long term reduction in inpatent services represents not so much deinstitutionalization as transinstutionalization.  A lack of those beds means that persons in need of them are being relegated to jails and prisons. In the United States, the Los Angeles jails represent the largest mental health treatment facility in the United States and the conditions for the severely disturbed are abominable. Similar situations hold in the UK.

An inpatient psychiatrist recently wrote in the New York Times:

We also need to rethink how we care for another group of vulnerable patients who have been just as disastrously disserved by policies meant to empower and protect them: the severely mentally disabled.

He went on:

We have worked to minimize the use of restraint and seclusion on my unit, but have seen the frequency of both skyrocket. Nearly every week staff members are struck or scratched by largely nonverbal patients who have no other way to communicate their distress. Attempting to soothe these patients monopolizes the efforts of a staff whose mission is to treat acute psychiatric emergencies, not chronic neurological conditions. Everyone loses.

Professor-Simon-Wessely-007Somebody in the UK should be speaking up for the inarticulate vulnerable persons with schizophrenia needing inpatient beds who are silenced and marginalized by the authors of Understanding Psychosis.  Where the hell is Simon Wessely when they need him?

Promoting an unrealistic view of schizophrenia?

If the authors of Understanding Psychosis were truly interested in providing authoritative information for persons with schizophrenia or psychosis, their family members, and professionals who come into contact with them, they would’ve provided the latest evidence about long-term course and outcome.

For instance, a key English study provides a 10 year follow-up individuals with a first episode of psychosis initially identified in either southeast London or Nottingham.

Morgan, C., Lappin, J., Heslin, M., Donoghue, K., Lomas, B., Reininghaus, U., … & Dazzan, P. (2014). Reappraising the long-term course and outcome of psychotic disorders: the AESOP-10 study. Psychological medicine, 44(13), 2713-2726.

At follow-up, of 532 incident cases identified, at baseline 37 (7%) had died, 29 (6%) had emigrated and eight (2%) were excluded. Of the remaining 458, 412 (90%) were traced and some information on follow-up was collated for 387 (85%). Most cases (265, 77%) experienced at least one period of sustained remission; at follow-up, 141 (46%) had been symptom free for at least 2 years. A majority (208, 72%) of cases had been employed for less than 25% of the follow-up period. The median number of hospital admissions, including at first presentation, was 2 [interquartile range (IQR) 1–4]; a majority (299, 88%) were admitted a least once and a minority (21, 6%) had 10 or more admissions. Overall, outcomes were worse for those with a non-affective diagnosis, for men and for those from South East London.Conclusions Sustained periods of symptom remission are usual following first presentation to mental health services for psychosis, including for those with a non-affective disorder; almost half recover.

Put differently, overall

12% (9% for non-affective) of our sample recovered within 6 months of contact with services and did not have a further episode, 20% (14% for non-affective) never had an episode lasting more than 6 months, and around 50% (40% for non-affective) had not experienced symptoms in the 2 years prior to follow-up.

And then there is the most recent comprehensive systematic review and meta-analysis.

Jääskeläinen, E., Juola, P., Hirvonen, N., McGrath, J. J., Saha, S., Isohanni, M., … & Miettunen, J. (2012). A systematic review and meta-analysis of recovery in schizophrenia. Schizophrenia Bulletin, sbs130.

We identified 50 studies with data suitable for inclusion. The median proportion (25%–75% quantiles) who met our recovery criteria was 13.5% (8.1%–20.0%). Studies from sites in countries with poorer economic status had higher recovery proportions. However, there were no statistically significant differences when the estimates were stratified according to sex, midpoint of intake period, strictness of the diagnostic criteria, duration of follow-up, or other design features. Conclusions: Based on the best available data, approximately, 1 in 7 individuals with schizophrenia met our criteria for recovery. Despite major changes in treatment options in recent decades, the proportion of recovered cases has not increased.

One in 7 people with schizophrenia meet criteria for recovery, and the portion has not increased in recent decades. Compare that with the unrealistically cheery assessment offered in Understanding Psychosis:

Even if people continue to hear voices or hold unusual beliefs, they may nevertheless lead very happy and successful lives. Sometimes a tendency to ‘psychosis’ can be associated with particular talents or abilities.

And

p 30 People who continue to have severe and distressing experiences may lead happy and successful lives in all other respects, such as work and relationships.

Sure, the authors of Understanding Psychosis keep reminding us of the cliché that everybody is different. But they are asking us to make clinical and policy decisions that are life altering for some people and could be life-ending for others. We can’t afford to ignore a larger body of relevant data.

In light of the data from long term follow-up studies, Understanding Psychosis should be seen as a cruel hoax perpetrated against more typical severely disturbed mental health service users, their family, and policymakers