By David Tuller, DrPH
David Tuller is academic coordinator of the concurrent masters degree program in public health and journalism at the University of California, Berkeley.
The past year has been a disaster for proponents of the PACE trial. They have faced growing international resistance to their exaggerated claims that cognitive behavior therapy and graded exercise therapy are effective treatments for chronic fatigue syndrome, also known as ME/CFS. The recent court-ordered release of key trial data has confirmed what was long self-evident: The PACE authors weakened their outcome criteria mid-stream in ways that allowed them to report dramatically better results for “improvement” (in The Lancet in 2011) and “recovery” (in Psychological Medicine in 2013). In refusing to provide the findings per the original protocol methods, or statistical analyses assessing the impact of the many mid-trial changes, or their actual trial data, they were able to hide their disastrous results for five years.
Yet the PACE authors and their allies continue, astonishingly, to defend the indefensible study, cite its findings approvingly, and push forward with ever more research into behavioral and cognitive interventions. The latest case in point: Esther Crawley, a British pediatrician and a highly controversial figure in the ME/CFS community because of her longtime promotion of the CBT/GET approach. On November 1st, the Science Media Centre in London held a press briefing to tout Dr. Crawley’s current venture–FITNET-NHS, a one-million-pound study of online CBT that is now recruiting and seeks to enroll more than 700 adolescents.
Dr. Crawley is a professor of child health at the University of Bristol. She is also currently recruiting for the MAGENTA study of graded exercise therapy for children with the illness. She is a lead player in the U.K. CFS/ME Research Collaborative, an umbrella organization that is sponsoring an ambitious Big Data effort called MEGA, now in the planning stages. While patients and advocates are desperate for the kind of top-notch biomedical and genetic research being proposed, many oppose MEGA precisely because of the involvement of Dr. Crawley and Peter White, the lead PACE investigator. (Dr. White is reportedly no longer involved in MEGA; Dr. Crawley still definitely is.)
The rationale for FITNET-NHS is that many ME/CFS patients live too far from specialists to obtain adequate care. Therefore, CBT delivered through an online portal, along with e-mail communication with a therapist, could potentially provide a convenient answer for those in such circumstances. The SMC press briefing generated widespread and enthusiastic news coverage. The BBC’s breathless online report about the “landmark” study noted that the online CBT “successfully treats two-thirds of children with chronic fatigue syndrome.” According to the BBC story, the intervention was designed “to change the way the children think of the disease.”
The BBC story and other news reports did not mention that the PACE trial–a foundational piece of evidence for the claim that changing people’s thoughts about the disease is the best way to treat it–has been publicly exposed as nonsense and is the subject of a roiling worldwide scientific debate. The stories also didn’t mention a more recent paper by the authors of the 2012 study from the Netherlands that was the source of the BBC’s claim of a “two-thirds” success rate.
The 2012 study, a Dutch version of FITNET, was published in The Lancet. (Why is The Lancet always involved?) In a subsequent paper published in Pediatrics in 2013, the Dutch team reported no differences among their trial participants at long-term follow-up. In other words, as with the PACE trial itself, any apparent advantages conferred by the investigators’ preferred treatment disappeared after the study was over. (More on the Dutch study below.)
The SMC, a purportedly neutral arbiter of science, actually functions as a cheerleader for research about cognitive and behavioral treatments for ME/CFS. Simon Wessely, a founder of the CBT/GET treatment paradigm and a close colleague of the PACE authors, is on the SMC’s board of trustees. The journalist who wrote the BBC story, James Gallagher, sits on the SMC’s advisory committee, so the reporting wasn’t exactly conducted at arm’s length. This reportorial conflict-of-interest was not disclosed in the BBC story itself.
(In fact, the Countess of Mar, a member of the House of Lords and a longtime advocate for ME/CFS patients, has filed a formal complaint with the BBC to protest its biased reporting on FITNET-NHS. In her complaint, she noted that “the BBC coverage was so hyperbolic and it afforded the FITNET trial so much publicity that it was clearly organised as a counter-punch to the anti-PACE evidence which is now gaining world-wide attention.”)
As a treatment for chronic fatigue syndrome, cognitive behavior therapy is grounded in an unproven hypothesis. According to the theory, the cause of patients’ continuing symptoms is a vicious downward spiral generated by false illness beliefs, a fear of engaging in activity, and progressive deconditioning. Whatever the initial viral or other illness that might have triggered the syndrome, patients are presumed to be currently free of any organic disease. Changing their beliefs through CBT, per the theory, will help encourage them to increase their levels of activity and resume their normal lives.
Here’s the rationale for the treatment from the PACE study itself: “CBT was done on the basis of the fear avoidance theory of chronic fatigue syndrome. This theory regards chronic fatigue syndrome as being reversible and that cognitive responses (fear of engaging in activity) and behavioural responses (avoidance of activity) are linked and interact with physiological processes to perpetuate fatigue. The aim of treatment was to change the behavioural and cognitive factors assumed to be responsible for perpetuation of the participant’s symptoms and disability. Therapeutic strategies guided participants to address unhelpful cognitions, including fears about symptoms or activity by testing them in behavioural experiments.”
The goal of this specific form of CBT, therefore, is to reverse the “reversible” illness by helping patients abandon their “unhelpful” beliefs of having a medical disease. This is definitely not the goal of CBT when it is used to help people cope with cancer, Parkinson’s, or other illnesses; no one claims those diseases are “reversible.” That the PACE authors, Dr. Crawley, and their Dutch colleagues promote CBT as a curative treatment and not simply a management or adaptive strategy is clear from their insistence on using the word “recovery”–a term that has no well-defined or universally understood meaning when it comes to this illness but has a very clear meaning to the general public.
While PACE so far remains in the literature, the study has been rejected by dozens of leading clinicians and academics, in the U.S. and elsewhere. Last February, an open letter to The Lancet signed by 42 experts and posted on Virology Blog condemned its egregious flaws, noting that they “have no place in published research.” The study has even been presented as a case study of bad science in graduate epidemiology seminars and at major scientific gatherings.
*****
Like the work of the PACE authors, Dr. Crawley’s research is fraught with misrepresentations and methodological problems. Like them, she routinely conflates the common symptom of chronic fatigue with the illness called chronic fatigue syndrome–a serious error with potentially harmful consequences. (I will mostly use chronic fatigue syndrome in describing the research because that is the term they use.)
Dr. Crawley favors subjective over objective outcomes. In PACE, of course, the objective measures–like a walking test, a step-test for fitness, and employment status–all failed to demonstrate “recovery” or reflect the reported improvements in the two primary subjective outcomes of physical function and fatigue. FITNET-NHS doesn’t even bother with such measures. The primary outcome is a self-report questionnaire assessing physical function, and almost all the secondary outcomes are also subjective.
This is particularly troubling because FITNET-NHS, like PACE, is non-blinded; that is, both participants and investigators know which intervention they are receiving. Non-blinded studies with subjective outcomes are notoriously vulnerable to bias–even more when the intervention itself involves telling participants that the treatment will make them better, as is the case with the kind of cognitive behavior therapy provided for ME/CFS patients.
The FITNET-NHS study protocol states that participants will be identified using the guidelines developed by NICE–the U.K.’s National Institute for Health and Care Excellence. The protocol describes the NICE guidelines as requiring three months of fatigue, plus one or more of nine additional symptoms: post-exertional malaise, difficulty sleeping, cognitive dysfunction, muscle and/or joint pain, headaches, painful lymph nodes, general malaise, dizziness and/or nausea, or palpitations. In other words, according to the protocol, post-exertional malaise is not required to participate in FITNET-NHS; it is clearly identified as an optional symptom. (In the U.K., the illness can be diagnosed at three months in children, rather than at six months.)
But the proposal’s claim to be following the NICE guidelines does not appear to be true. In the NICE guidelines, post-exertional malaise is not an optional symptom. It is required, as an essential element of the fatigue itself. (In addition, one or more of ten other symptoms must also be present.) To repeat: post-exertional malaise is required in the NICE guidelines, but is not required in the description of the NICE guidelines provided in the FITNET-NHS protocol.
By making this subtle but significant shift–a sleight-of-guideline, so to speak–Dr. Crawley and her colleagues have quietly transformed their prospective cohort from one in which post-exertional malaise is a cardinal characteristic of the illness to one in which it might or might not be present. And they have done this while still claiming–inaccurately–to follow NICE guidelines. As currently described, however, Dr. Crawley’s new study is NOT a study of chronic fatigue syndrome, as she maintains, but of chronic fatigue.
As a result, the actual study participants, like the PACE cohort, will likely be a heterogeneous grab bag of kids suffering from fatigue for any number of reasons, including depression–a common cause of exhaustion and a condition that often responds to psychotherapeutic interventions like CBT. Some or even many participants–an unknown number–will likely be genuine ME/CFS patients. Yet the results will be applied to ALL adolescents identified as having that illness. Since those who actually have it suffer from the required symptom of post-exertional malaise, an intervention that encourages them to increase their activity levels, like CBT, could potentially cause harm.
(I suppose it’s possible the FITNET-NHS protocol’s inaccurate description of the role of post-exertional malaise in the NICE guidelines was inadvertent, a case of sloppiness. If so, it would be an extraordinary oversight, given the number of people involved in the study and the enormous implications of the switch. It is curious that this obvious and jarring discrepancy between the NICE guidelines and the FITNESS-NHS description of them was not flagged during the review process, since it is easy to check whether the protocol language accurately reflects the recommendations.)
Yet Dr. Crawley is experienced at this blurring of categories–she did the same in a study she co-authored in the journal Pediatrics, in January of this year. In the study, “Chronic Fatigue Syndrome at Age 16 Years,” she and colleagues reported that almost one in fifty adolescents suffered from the illness–an extremely high rate that attracted widespread media attention. The main conclusion was described like this: “CFS affected 1.9% of 16-year-olds in a UK birth cohort and was positively associated with higher family adversity.”
However, the Pediatrics study is unreliable as a measure of “chronic fatigue syndrome.” It is of note that this paper, like the FITNET-NHS protocol, also appears to have inaccurately presented the NICE guidelines. According to the Pediatrics paper, NICE calls for a CFS diagnosis after three months of persistent or recurrent fatigue “that is not the result of ongoing exertion, not substantially alleviated by rest, has resulted in a substantial reduction of activities, and has no known cause.” But this description is incomplete–it omits the NICE requirement that the fatigue must include the specific characteristic of post-exertional malaise in order to render a diagnosis of chronic fatigue syndrome.
In the Pediatrics paper, the determination of illness was based not on clinical examination but on parental reports of children’s unexplained fatigue. In a previous study of 13-year-olds that relied on the same U.K. database, Dr. Crawley and her co-authors referred to the endpointappropriately–as “disabling chronic fatigue.” But in this study, they justified changing the endpoint to “chronic fatigue syndrome” by noting that they cross-referenced the parental reports with children’s self-reports of their own fatigue.
Here’s how they explained this shift in nomenclature: ‘In the earlier study, we were unable to confirm a diagnosis of CFS because we had only parental report of fatigue; hence, chronic disabling fatigue was defined as the study outcome. In the present study, parental and child report of fatigue were combined to identify adolescents with CFS.”
This reasoning is incoherent. A child’s confirmation of a parental report of fatigue cannot be taken to indicate the presence of chronic fatigue syndrome–especially without a clinical examination to rule out other possible conditions. Moreover, neither the parental nor child reports appear to have included information about post-exertional malaise, which is required for a diagnosis of chronic fatigue syndrome–even though the Pediatrics study did not mention this requirement in its description of the NICE guidelines. In fact, the authors provided no evidence or data to support their assumption that a double-report of fatigue equaled a case of chronic fatigue syndrome. (How’d that assumption ever pass peer review?)
Moreover, the study itself acknowledged that, when those found to be suffering from high levels of depression were removed, the prevalence of what the investigators called chronic fatigue syndrome was only 0.6 %. And since depression is likely to be highly correlated with chronic fatigue as well as with family adversity, it is not surprising that the study found the apparent association between family adversity and chronic fatigue syndrome that the investigators highlighted in their conclusion. That misinterpretation of their data has likely lent support to the widespread but inaccurate belief that the illness is largely or even partly psychiatric in nature.
In any event, the figure of 0.6 % should have been identified as the prevalence of “chronic disabling fatigue, not attributable to high levels of depression.” Without any further clinical data, to identify either 1.9 % or 0.6 % as the prevalence of chronic fatigue syndrome was unwarranted and irresponsible. Although the authors cited the lack of clinical diagnosis as a limitation, this acknowledgement does not excuse their interpretive leap. To call this a study of chronic fatigue syndrome is really misleading–a serious over-interpretation of the data.
In subsequent correspondence, three professors of pediatrics–Marvin Medow and Julian Stewart from New York Medical College, and Peter Rowe from Johns Hopkins–scolded the study authors for identifying the participants as having chronic fatigue syndrome rather than chronic fatigue. They cited this misclassification as the likely source of the reported link between chronic fatigue syndrome and family adversity. In particular, they challenged diagnoses made without benefit of clinical evaluations.
“An important component of the diagnosis is a physician’s history and physical examination to exclude conditions that could explain the fatigue, including hypothyroidism, heart disease, cancer, liver failure, covert drug abuse, medication side effects, gastrointestinal/nutritional, infectious and psychiatric conditions,” they wrote. The Pediatrics paper, concluded the three pediatricians, “should be titled ‘Chronic Fatigue but not Chronic Fatigue Syndrome at Age 16 Years.'”
In response, the study authors agreed that clinical diagnoses would be more accurate. But they did not address the critical issue of why they decided that two reports of chronic fatigue could be used to identify chronic fatigue syndrome.
*****
The conflation of chronic fatigue and chronic fatigue syndrome is a huge problem in ME/CFS research. That’s why a major report last year from the National Institutes of Health declared that the case definition used in PACE–which required only six months of unexplained fatigue but no other symptoms–could “impair progress and cause harm,” and should be “retired” from use. But Dr. Crawley and her colleagues do not seem to have gotten the message.
At the SMC press briefing presenting FITNET-NHS, one of the experts appearing with Dr. Crawley was Dr. Stephen Holgate, the leader of the CFS/ME Research Collaborative and a professor of immunopharmacology at the University of Southampton. According to the BBC report, he praised the new trial as “high-quality research.” This endorsement suggests that Dr. Holgate, like Dr. Crawley, does not appreciate the significance of the distinction between the symptom of chronic fatigue and the illness called chronic fatigue syndrome–a troubling blind spot. It also suggests that Dr. Holgate is unaware or unconcerned that the main support for the use of CBT in this illness, the PACE trial, has been discredited.
Also at the SMC briefing was Paul McCrone, a professor of health economics from King’s College London and a PACE co-author. Dr. McCrone is serving as the chair of FITNET-NHS’ independent steering committee–another unsettling sign. As I have documented on Virology Blog, Dr. McCrone made false claims as lead author in a 2012 PLoS One paper–and those false claims allowed the PACE authors to declare that CBT and GET were cost-effective. They have routinely cited this fraudulent finding in promoting the therapies.
Beyond the problem of conflating “chronic fatigue” and “chronic fatigue syndrome,” Dr. Crawley’s reliance on the Dutch trial suggests that this previous FITNET study warrants a closer look–especially since the BBC and other news outlets cited its robust claims of success in extolling the U.K. version.
The approach to CBT in the Dutch FITNET trial reflects that in the U.K. Of the online intervention’s 21 modules, according to the protocol for the Dutch study, fourteen “focus on cognitive behavioural strategies and include instructions and exercises on how to identify, challenge and change cognitive processes that contribute to CFS.” Of course, experts outside the CBT/GET/PACE bubble understand that ME/CFS is a physiological disease and that faulty “cognitive processes” have nothing to do with perpetuating or contributing to it.
The Dutch study found that those assigned to FITNET reported less fatigue, greater physical function, and greater school attendance than those in the comparison group, who received standard treatment–referred to as “usual care.” And using a composite definition of “recovery,” the study reported that 63% of those in the FITNET group–just shy of two-thirds–“recovered” at six months, compared to just eight percent in the comparison group. But this apparent success masks a much more complicated reality and cannot be taken at face value, for multiple reasons.
First, the subsequent 2013 paper from the Dutch team found no differences in “recovery” between participants in the two groups at long-term follow-up (on average, 2.7 years after starting). Those in the comparison group improved after the trial and had caught up to the intervention group, so the online CBT conferred no extended advantages or benefits. The researchers argued that the therapy was nonetheless useful because patients achieved gains more quickly. But they failed to consider another reasonable explanation for their results.
Those in usual care were attending in-person sessions at clinics or doctors’ offices. Depending on how often they went, how far they had to travel and how sick they were, the transportation demands could easily have triggered relapses and harmed their health. In contrast, those in the FITNET group could be treated at home. Perhaps they improved not from the treatment itself but from an unintended side effect–the sedentary nature of the intervention allowed them more time to rest. The investigators did not control for this aspect of the online CBT.
Second, the “recovery” figure in the Dutch FITNET study was a post-hoc calculation, as the authors acknowledged. The protocol for the trial included the outcomes to be measured, of course, but the authors did not identify before the trial what thresholds participants would need to meet to be considered “recovered.” The entire definition was constructed only after they saw the results–and the thresholds they selected were extremely lenient. Even two of the PACE authors, in a Lancet commentary praising the Dutch study, referred to the “recovery” criteria as “liberal” and “not stringent.” (In fact, only 36% “recovered” under a more modest definition of “recovery,” but the FITNET authors tucked this finding into an appendix and Dr. Crawley’s FITNET-NHS protocol didn’t mention it.)
Now, the fact that “recovery” was a post-hoc measure doesn’t mean it isn’t valid. But anyone citing this “recovery” rate should do so with caveats and some measure of caution. Dr. Crawley has exhibited no such reticence; in a recent radio interview, she declared flatly that the Dutch participants had made a “full recovery.” (In the same interview, she called PACE “a great, great study.” Then she completely misrepresented the results of the recent reanalyses of the PACE trial data. So, you know, take her words for what they’re worth.)
Given the hyperbole about “recovery,” the public is understandably likely to assume that Dr. Crawley’s new “landmark” study will result in similar success. A corollary of that assumption is that anyone who opposes the study’s approach, like so many in the patient and advocacy communities, could be accused of acting in ways that harm children by depriving them of needed treatment. This would be an unfair charge, since the online CBT being offered is based on the questionable premise that the children harbor untrue cognitions about their illness.
Third, the standard treatments received by the usual care group were described like this: “individual/group based rehabilitation programs, psychological support including CBT face-to-face, graded exercise therapy by a physiotherapist, etc.” In other words, pretty much the kinds of “evidence-based” strategies these Dutch experts and their U.K. colleagues had promoted for years as being effective for chronic fatigue syndrome. In the end, two-thirds of those in usual care received in-person CBT, and half received graded exercise therapy. (Many participants in this arm received more than one form of usual care.)
And yet less than one in ten of the usual care participants were found to have “recovered” at six months, according to the 2012 study. So what does that say about the effectiveness of these kinds of rehabilitative approaches in the first place? In light of the superlative findings for online CBT, why haven’t all chronic fatigue syndrome patients in the Netherlands now been removed from in-person treatments and offered this more convenient option? (Dr. Crawley’s FITNET-NHS proposal tried to explain away this embarrassing finding of the Dutch study by suggesting that those providing usual care were not trained to work with this kind of population.)
Finally, the Dutch study did not report any objective measures of physical performance. Although the study included assessments using an actometer–an ankle bracelet that monitors distance moved–the Lancet paper did not mention those results. In previous studies of cognitive and behavioral treatments for ME/CFS, reported improvements on subjective measures for fatigue or physical function were not accompanied by increases in physical movement, as measured by actometer. And in PACE, of course, the investigators dismissed their own objective measures as irrelevant or non-objective–but only after these outcomes failed to provide the desired results.
In response to correspondence calling for publication of the actometer data, the Dutch investigators refused, noting that “the goal of our treatment was reduction of fatigue and increase in school attendance, not increase in physical activity per se.” This is an inadequate explanation for the decision to withhold data that would shed light on whether participants actually improved in their physical performance as well as in their subjective impressions of their condition. If the actometer data demonstrated remarkable increases in activity levels in the online CBT group, is there any doubt they would have reported it?
In short, the Dutch FITNET study leaves a lot of questions unanswered. So does its U.K. version, the proposed FITNET-NHS. And Dr. Crawley’s recent media blitz–which included a “can’t-we-all-get-along” essay in The New Scientist–did little to quell any of the reasonable qualms observers might have about this latest effort to bolster the sagging fortunes of the CBT/GET/PACE paradigm.
“Patients are desperate for this trial, yet some people are still trying to stop us,” wrote Dr. Crawley in The New Scientist. “The fighting needs to end.”
However, those mysterious and sinister-sounding “some people” cited by Dr. Crawley have very thoughtful and legitimate reasons for questioning the quality of her research. The fighting, as she calls it, is likely to end when Dr. Crawley and her colleagues stop conflating chronic fatigue and chronic fatigue syndrome through the use of loose diagnostic criteria. And when they acknowledge what scientists in the U.S. and around the world now understand: The claim that cognitive and behavioral approaches are effective treatments that lead to “recovery” is based on deeply flawed research.
A Short Postscript:
Several Dutch colleagues have joined Dr. Crawley as part of the FITNET-NHS study. Two of them, Dr. Gijs Bleijenberg from the Radboud University Medical Centre in Nijmegen, and Dr. Hans Knoop from the University of Amsterdam, are among the leaders of the CBT/GET movement in the Netherlands and have collaborated with their U.K. counterparts. Not surprisingly, their work is similarly dodgy.
In a post last year, I dissected a 2011 commentary in The Lancet on the PACE trial, co-authored by Dr. Bleijenberg and Dr. Knoop, in which they argued that 30 percent of the participants in the CBT and GET groups had met “a strict criterion for recovery.” This statement was absurd, since these “strict” thresholds for “recovery” were in fact so lax that participants could get worse during the study and still meet them. Although the problematic nature of the thresholds has been pointed out to Dr. Bleijenberg and Dr. Knoop, they have stood by their nonsensical claim.
Earlier this year, the Dutch parliament asked the Health Council–an independent scientific advisory body–to review the state of evidence related to the illness, including the evidence on treatments like CBT and GET. The Health Council appointed a committee to conduct the review. Among the committee members are Dr. Knoop and colleagues who share his perspective. It remains unclear whether the committee is taking sufficient account of the methodological flaws underpinning the evidence for the CBT/GET paradigm and of the ongoing condemnations of the PACE trial from well-respected scientists. I plan to blog about this situation soon.
Thank you for telling this important story, David. Dr Crawley and colleagues are continuing trucking along, promoting harm and delaying biomedical research for patients with ME using tactics which would be deemed ridiuclous if the studied field was HIV or oncology or cardiology.
You talk a lot in this article about who is in bed with whom. It is very curious at the same name come back and come back again, but now we hear about british media, notably BBC who is going along with the same stories which ignore the real science and ignore how PACE trial has been debunked. Of course the common denominator that BBC has is Science Media Center.
The game they play, in the name of the science, has been very harmful, and has been allowed to go on for decades now, because of who is in bed with whom. Like how Putin and Trump, if you will.
Thank you for your work. Will be sharing.
Thanks to David Tuller for his continuing work.
Not only does Crawley continue to assert that PACE is “a great, great studyâ€, but she has also claimed that it showed a recovery rate of 30-40% for CBT and GET. The most indefensible claim to come from this embarrassing debacle?
All of Crawley’s work seems focused on exaggerating the prevalence of CFS and the efficacy of the treatments available for it. This approach serves the interests of those who run and work at the NHS’s ‘specialist’ CFS centres, but not patients.
It does not surprise me at all that so many patients now seem opposed to more funding being given to any research that involves Crawley.
Thank you for your continued work on this (and thanks also to the Countess of Mar for her work). The extent to which the individuals behind the PACE studies have continued to promote their premise in the face of all the emerging evidence is disgraceful.
Lisa Petrison, Ph.D.
Executive Director
Paradigm Change
Thank you David for continuing to cover the absurdities in our dusty, forgotten little corner of medicine. It’s ridiculous that such obvious flaws need to be pointed out to researchers who receive millions of dollars for research, but here we are again.
To add to the discussion of Esther Crawley’s false illness beliefs, it would appear that she doesn’t believe that severe ME/CFS occurs in children, when a child patient doesn’t respond to CBT/GET in the way that, according to EC, they should, they are then diagnosed with Pervasive Refusal Syndrome. Stories also exist of parents, concerned at the damage being caused to their children’s health, are reported to social services if they seek to stop the ‘treatment’ which is harming their child.
Link is to a paper EC co-authored on Pervasive Refusal Syndrome back in 2014 https://www.researchgate.net/publication/273874213_G160P_Case_series_of_Pervasive_Refusal_Syndrome_presenting_with_Chronic_Fatigue_Syndrome_avoiding_the_pitfall_of_a_wrong_diagnosis
Patients in the UK, and elsewhere, are desperate for good quality biomedical research into ME, it would seem, sadly, that Crawley, Holgate et al are only intent on riding the biopsychosocial CBT/GET gravy train for as long as it keeps running.
You may be interested to this blogpost, David. https://opposingmega.wordpress.com/2016/11/19/dr-keith-geraghty-comments-on-opposing-mega/
It ties in nicely to what you are saying. Same group of people involved.
I suspect we see relatively little opposition to PACE, FITNET, etc. from within psychiatry and psychology because bad methodology is widespread and nobody is willing to acknowledge the extent of the problem. The bad methodology is not accidental. It serves a purpose. That of generating (false) positive results to advance careers and promote treatments.
I’m not sure if I missed it in the body but there does seem to be some confusion floating around about the follow study on FITNET. The non FITNET arm were offered the intervention at six months so the follow up study is no longer a comparison of intervention vs control. It does appear that FITNET did help but that is most likely (IMO) because it was given to people with chronic fatigue rather than CFS/ME.
I am grateful this story continues to be told. I just hope it reaches those who are unwittingly being drawn into the FITNET and MAGENTA trials, which show every reason to prove harmful.
Thank you, David, for this excellent analysis. In any other field, researchers responsible for fraudulent studies like PACE etc would have been drummed out of their profession, but it seems that the pseudoscience of psychiatry has far more lax standards.
I think this trial shows that there is a lack of quality control on trials in the UK. The ethics committee should have picked on the lack of independence of their McCrone in their steering committee. Peer review should have picked up that the protocol will yield meaningless results given all the outcomes are subjective and the trial is aiming to change patients views on symptoms.
The publicity campaign is concerning as it is offering a cure prior to the trial being run. In it they are misleading the public.
This trial should be stopped because it will not lead to meaningful results and is therefore unethical.
A great article. Its so so appreciated to have you calling out the FITNET problems. The flaws stand out like dogs balls and yet Bristol University, Oxford University, King’s College of London, Queen Mary of University of London all turn a blind eye?? Why?? If a student handed in this crap they would be failed, wouldn’t they? Or has UK science sunk this low???? Has Ester Crawley been assessed???
I have never understood why Professor Crawley has been involved with ME or CFS, as it would appear she never diagnoses any illnesses, and gets easily confused with the NICE guidelines as you so expertly pointed out.
In the NICE guidelines, it states that heart monitors should be worn if exercise is to be undertaken, no one ever takes any notice of this either. On the PoTS NHS choses it states that PoTS is also present in CFS, Professor Crawley never checks as a matter of routine for this or any OI? She trains many hospitals in the condition including Great Ormand Street hospital? It would seem no hospital is free from her influence, so we have nowhere to go as parents.
The heart monitor matters to children with ME, so does the fact that they have connective tissue problems. Connective tissues are not just in joints but internal organs too, yet she does not recognise EDS.
My son at the age of 9 was taken from the psychologists room where we having were having a consultation, and without my knowledge or consent, he was taken to another room and made to stand up from a chair 20 times and catch a heavy ball many times, by a physio. I had advised them of him not being able to see my face at times before this consultation took place. He has never recovered from that day. His heart rate from a sitting to stand position can go up to 240 BPM his blood pressure is all over the place.
In that consultation I was questioned from every angle on what I said to my child, and was advised never to speak of my sons symptoms, they were “superficial� My son has never had a scan, so I could not understand why she would use this assumption.
Being questioned in such a way made me re-valuate my understanding of parenting, it is made to do that! So I looked for a second opinion on my son’s condition. I have much to thank Doctor Speight for. He understood what I had gone through and knew I would not be satisfied with just reassurance that I was doing the right thing. He methodically took me through my son’s medical history and symptoms. He pointed out my actions and reasoning was correct. When I questioned him, he gave me time and space to process the information. We worked together on a plan to go forward with my son’s care and now his is improving, it is a very slow but steady progression.
Through understanding this condition, my son has now been diagnosed as Coeliac, which is also an autoimmune disease. PoTS and hypermobile joints, most if not all of the medical staff I have encountered first view CFS as insignificant and I have to point out the Coeliac and PoTS to get any credence to his condition. This is an absurd situation as ME is a very significant disease.
I cannot fight NHS, NICE, PACE or FitnetNHS, I can’t even speak in consultations or ask questions due to the idea mothers perpetuate this illness. I know and talk to many mothers who feel the same. So it with a humble heart David that I thank you for your involvement in this tangled web. Your level reasoning; like Doctor Speight, gives me an understanding of the condition and the politics that play a big role in this life changing illness. More importantly you give us parents a voice and hope.
Thanks very much for continuing to cover PACE and its fallout. What’s disturbing now is that trials such as MAGENTA (also headed up by Esther Crawley) explicitly base themselves on claims that PACE showed that CBT/GET were effective – whereas arguably, PACE showed, via objective measures, that they weren’t.
In a sane world – which excludes the UK medical funding bodies, apparently – no further trials would be conducted on CBT or GET.
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Another point worth making is Dr Crawleys Lightning Process trial on children (SMILE) the results of which remain a mystery.
Thanks so much David for bringing reason and scientific scrutiny to this area. It is difficult to believe how much power and support Crawley et al must have that they can get away with poorly designed studies based on false premises.
Thank you again David for pulling all the threads of this cumbersome and deliberately tangled web into some sort of order that everyone can understand. It would seem the Wessely School is getting ever more desperate to hang on to the gravy train that has provided good incomes, chairs, Knighthoods and ego-grooming for many decades but even those within their own discipline are realising that this ‘little Napoleon’ is pretty much stark naked. Although this link (below) deals with psychiatric disease, and might not be as clear as I personally would prefer in making the distinction that ME is unequivocally a biomedical problem, it’s helpful for sufferers to know that a movement does exist to expose the abuses meted out by the psychiatric profession in general and specifically to people with ME (http://blogs.lse.ac.uk/businessreview/2016/11/21/healing-the-rifts-between-mental-health-workers-and-psychiatric-survivors/)
“The poor treatment of service users is not only historical with the current mental health system still having a lot to answer for. This involves not only the use of coercive ‘treatment’ and confinement, including psychosurgery, ECT and often harmful psychoactive drugs, but also various forms of invalidation, or what has been called ‘epistemic injustices,’ where people’s self-knowledge and experiences are disbelieved and dismissed.
Just two examples will suffice……………Second, the psychiatrisation of conditions like Myalgic Encephalopathy/Chronic Fatigue Syndrome (ME/CFS) where organic and physical conditions become ‘all in the mind’. Here, whilst there is a complex inter-relationship between the mind and body, psychiatric reductionism has resulted in a catalogue of instances of maltreatment, neglect and abuse. ”
Interestingly the author continue thus : ” Most ME/CFS sufferers are female, many of whom have been referred to as ‘hysterical’ and ‘irrational’ by doctors and psychiatrists. The medical and psychiatric establishment has yet to come clean, admit to or apologise for these abuses.” ..but in the 24 hours since this blog was first posted has removed a comment in which she directly implicates Wessely. I wonder why this is so? I think many of us can make an educated guess.
Hi David,
Thank you so much again.
I would like to add one or two quick things. Firstly, the ‘other care’ group were receiving not just CBT. but also GET and physio. Your hypothesis that the travel could have exhausted kids in this group is entirely plausible, but just as reasonably we could expect the deleterious effects of exercising these kids to show up in the data. This could easily account for the difference in recovery rate between the two groups.
Secondly, for uninitiated readers, I don’t think even the anti-PACE scientific articles go far enough to stress just how damaging GET can be. 74% of respondents to the largest survey conducted reported that GET had made them worse. Although this did not appear in a peer reviewed journal, it had double the participants of PACE and had an extensive statistical analysis. We need to stress this point most of all- that the PACE techniques aren’t just ineffective and misleading, but can cause severe and lasting damage.
Thank you for your work and advocacy. We need more scientists like you. I wish your writing could reach all those people who are in a position to make decisions about research funding, especially those allocating funds in the U.K. I also wish that scientific news reporting could rely on sound investigative journalism rather than biased press releases. The status quo is unbelievably damaging to those of us who cannot fight back. Please continue your work on our behalf and on behalf of true science. We certainly need you.
Your story has driven me to tears. It is awful enough to be an adult with this disease. I can’t even begin to imagine what it must be like for your little boy. It must be truly heartbreaking to witness his suffering and horrific to have to deal with the likes of the CBT/GET proponents. Stay strong and have faith that a cure is on its way. Here in Norway thankfully they are doing promising research.
Very interesting read.
I think it’s worth pointing out that children are particularly exposed to the risk of bias in trials that use subjective measures. Children look up to adults, and especially doctors who they believe, with their limited life experience, can fix all problems just like Doc McStuffins etc.
The child/adult relationship alone pressures the child to give the “right” answer, just like in school.
How studies like this get approved I do not know.
I recently signed the stopGET petition, which may be of interest to people: http://www.meaction.net/suspend-all-trials-of-graded-exercise-therapy-in-children-and-adults-with-mecfs/
Absurd how these ‘researchers’ imagine a kind of synchronised somatising, that exists across decades, across countries… And even more absurd that they get away with it. Thirty years ago, aged 20, having been diagnosed with severe ME, I was in an NHS neurology ward in UK having a plasma exchange with immunosuppression – this was an experimental therapy, but I was, at least, being treated appropriately by a consultant neurologist for the poorly understood neuroimmune illness I had (and still have). It beggars belief that three decades on, this core of UK researchers are conflating fatigue with ME – and now potentially harming children. I was only 18 when I got the enterovirus that triggered ME – had anyone suggested GET or CBT for my wellbeing/recovery it would have been torture – and, as I was severely ill, impossible.
Thank you for highlighting Esther Crawley’s continued incompetence in the field of research into Chronic Fatique Syndrome. That she is allowed to continue with the MAGENTA trial given all the evidence of the harms of GET and flaws of the PACE trial is an indictment of the situation people and children with ME face in the UK. Your help in shining a light on this travesty will hopefully bring an end to it soon for the sake of children who are suffering. It is surely one of the worst things a person can do, to harm a child, how Crawley sleeps at night I do not know. If she doesn’t realise what she is doing then her incompetence is even greater than first thought.
Sorry that I made you cry Belen Fleck. It is heartbreaking at times but we are made of stern stuff and will make the best of what we have.
The suffering I think in the most part is avoidable is appropriately handled by well informed practitioners that can put over the correct way to accommodate a better life if not recovery. And you are so right in Norway they are making leaps and bounds.
Truth has a way of coming out and I looked at it as dripping tap – the more of us speak about our experiences the more people get to know and one day that dripping tap will fill the bucket and the truth will spill out for all to see.
I will keep strong and will think of you often.
Tina x
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A useful evaluation thank you.
One thing people seem not to have picked up on is Prof Crawley’s bizarre attempt to shift from a pysco-social model of ME to a biological whilst justifying CBT as a cure. I find this even more worrying as she is proposing another model that is totally without an evidence base for ME as a way of ignoring previous criticisms of the PACE influenced research:
‘I got ME and thought: ‘This is the end’ | The Times | 2 November 2016
From The Times, 2 November 2016. Story by Janet Eastham.
Crawley believes that CFS is a biological illness and that online CBT can be used to alter the young person’s biology. “In children we know that this is a very heritable condition: [some] children are genetically vulnerable,†she says. “And they get a virus or infection and that sets off a whole load of biology. Thirty per cent of children who come to our clinic do develop depression or anxiety and I am always constantly surprised that it’s not more.
“We use cognitive behavioural therapy to help children change their sleep, their exercise, their activity and so on, but what cognitive behavioural therapy does in this illness is change the biology. You change someone’s sleep so they are getting better sleep and they are waking up at the same time everyday, you change almost everything about them. You change the way their brain produces cortisol, you change what is called the hypothalamic-pituitary-adrenal axis . . . We don’t have a pill for this but this might as well be a pill in terms of the effect it actually has on the body.â€
Excellent. Thank you for all the work you’ve done on this, David.
The lack of controls (patients who are not given CBT or GET) in the Dutch FITNET and proposed FITNET-NHS trials is staggering. It’s like doing a drug trial with half the patients given the drug at home and the other half being given the same drug at a clinic. And then, when those given the drug at home do better, concluding that this is evidence that the drug is highly effective, wherever it is given. These aren’t clinical trials, they are show trials. Again and again we are told by the MRC (and other funding agencies) that the reason that so little money has been given to biomedical ME research studies is because of the poor quality of research proposals they receive. And yet they continue to throw money at this pseudoscience.
As you point out, one of the obstacles to progress and understanding is the UK Science Media Centre. I wonder if you and others could set up an international ME/CFS media centre to counter the SMC propaganda.
Myalgic Encephalomyelitis patients, one of the most medically undermined and neglected groups of the 21st century, are once again being let down by the UK psych lobby. FITNET, a potential UK trial on ME, one of the most debilitating and misunderstood illnesses today, is once again perpetuating the notion that psych therapies such as CBT and GET are potential easy quick-fix remedies for the extremely physical autoimmune illness ME.
Ignoring the recent debunking of PACE and heading into another similar trial, with the strong possibility of wasting so much money and time, and with the potential of harming very ill young patients, as well as perpetuating the notion that CBT and GET will treat ME effectively, is just very very very absurd.
The elephant in the room is actually beginning to feel ignored! The whole notion of FITNET is so completely preposterous with its unreliable and inappropriate criteria for patient selection – its use of patients with Chronic Fatigue instead of Chronic Fatigue Syndrome; with its use of the internet as an actual medium for dispensing treatment; with its constant ignoring of the failures of PACE; with its false truths about the Dutch study; with its unwillingness to set up trials to include appropriate scientific studies by scientists who’d look at clinical issues and malfunctioning whole bodily systems; with its persistence to identify ME as an ‘it’s-all-in-the-head’ illness; etc etc etc etc.
This has all been said before over and over again but lands on biased ears. Why can’t you listen with intent to hear the truth? Why do you defy the truth? Why do you ignore millions of patients worldwide, including 250,000 in the UK? Why do you falsely claim to be harassed and threatened by ME patients, activists and support groups; why do you see ME patients as a threat to the survival of real science, your professional standing, and your massive ego; why do you make false claims over and over again without concrete proof and use the BBC and other platforms like the Science Media Centre to spread your inaccuracies???
Thank you David for standing in our neglected corner once again and exposing the nightmares that are PACE, FITNET, MAGENTA, etc. You are bringing a vile injustice to the attention of a wider audience and exposing the horrors of a wicked side of the med world, as well as giving a voice to very frustrated, very ill, very angry patients who will not put up with this PACE-type crap any longer.
It’s time for Esther Crawley and her ilk to get real and do the right thing for ME patients, young and old. To you Esther Crawley and your cronies I want to say that your country is currently sitting in the naughty corner of the science/med world for faking and falsifying science, as well as ignoring real science, while your counterparts in the US etc are pushing boundaries and setting the trail for a better outcome for ME patients worldwide.
For every positive step forward towards a better life for people living with ME you cause a backward motion and you delay potentially better outcomes. Please stand back and let real scientists do their work!
Thank you David and kindest regards from a ME patient now heading into her 10th year with this awful illness, 7th year without her health, career, hobbies and friends; and way way too many years listening to UK psycho-babble claptrap!
Thank you David for exposing the harm they are attempting to perpetrate on children.
Just amazing. Thank you so much. Also for us as Dutch patients. The claims Knoop and Blijenberg are making are just as off the wall as the ones of Crawley. Scary part is that the medical establishment just believes it. Assumes without checking up on it.
All these trial results mean nothing with these variations of the Oxford criteria. Waste of money and health.
Wanted to add this nice picture.
Anyways David you are truly amazing. Thank you so so much. https://uploads.disquscdn.com/images/8b41b1a3c0f9a7ec96f77ef89daa88fa93638f671afbf79694b3abf5872a11bf.jpg
I inadvertently joined Esther Crawley’s Patient Advisory Group last year, not knowing anything about her or PACE, after my teenage daughter collapsed with ME. I had ME myself twenty years ago, and her father has Ankylosing Spondylitis, a rheumatic auto-immune disease. It has always struck me how very similar many of the symptoms are, including the fatigue, which has kept him off work for several years at a time during his working life, and the up-and-down nature of the illness. Although I’m not a medic, I am an academic, and I was interested to meet Esther; when she claimed that most children and young people got better within 6 months, I asked her if she checked how well they were in the months and years after that. She swept away my question by saying that she clearly didn’t have the funding to contact people after the end of the study. I thought this odd. My daughter felt better after 6 months, but much, much worse after 8. She has now been housebound for 18 months, and too ill to continue her education. I also asked Esther about the rituximab trial in Norway which I had read about in the New Scientist with real excitement – as my husband is on a similar immune-suppressant which has made a huge difference to his health, I could completely understand that this might be a breakthrough. In response Esther was completely dismissive of the trial itself, making very strong statements about the dodgy nature of the methodology of the investigators involved, and suggesting that this was the general view of the scientific community. How interesting to discover later, then, that very similar aspersions had been made about her own work. As an academic under increasing pressures to get funding and grow my international reputation and evidence of ‘impact’, I can understand Esther’s hard work to convince society of the effectiveness of her research through the media. I don’t, however, forgive her for her ability to suspend her morals by dismissing all evidence which suggests that she is wrong. As Karl Popper pointed out, we should all be trying as hard as we can to disprove our hypotheses, as only then can we hope to prove them.
Many thanks for another great article David (and Vincent for hosting it) and for hyperlinking to our petition Opposing MEGA. We’ve included this in today’s update on the Opposing MEGA petition and hope that’s ok – https://www.change.org/p/opposing-mega-a-vote-of-no-confidence-in-mega-research-for-me-cfs/u/18554906 Thanks again for all your amazing work.
This was our update following the media coverage of FITNET – https://www.change.org/p/opposing-mega-a-vote-of-no-confidence-in-mega-research-for-me-cfs/u/18346814
Dr. Nigel Speight is an internationally renowned UK expert in paediatric myalgic encephalomyelitis (ME). His knowledge, experience and expertise has been invaluable over many years to patients and families, particularly in the increasing number of cases of severe ME, diagnosed or re-diagnosed with labels such as factitious disorder (assuming the child is feigning their symptoms) or factitious disorder by proxy (suspecting parents/caregivers of inducing or maintaining the child’s illness, commonly known as Munchausen’s syndrome by proxy) or pervasive refusal syndrome (severe ME can cause an inability to speak or swallow) or pervasive arousal withdrawal syndrome (severe ME can cause extreme sensitivity to sensory stimuli such as sound, light, touch).
Dr. Nigel Speight commented that he signed Opposing MEGA,
Because I have long and bitter experience of the injustices done to ME sufferers by those doctors who hold to the psychological view of this very real illness. Nigel Speight, DURHAM, United Kingdom. (Link to comment – change.org/p/opposing-mega-a-vote-of-no-confidence-in-mega-research-for-me-cfs/c/527889662)
Incredibly to anyone who knows him, in April 2016, the UK General Medical Council (GMC) imposed conditions on Dr. Nigel Speight’s licence to practise, stating that he must not carry out any work in relation to ME/CFS in either a paid or unpaid capacity and must refer any patient with a possible diagnosis of ME/CFS to another consultant paediatrician, keeping a log detailing every such case. These restrictions were provisional until 19th July 2017. Fortunately all were removed unconditionally by a review panel at the Medical Practitioners Tribunal Service (MPTS) in October 2016 and the GMC continues its investigation. As the conditions included public speaking about ME/CFS, it is especially heartwarming to read the comment by Dr. Nigel Speight on a public petition site. We thank him for taking the time to consider and support Opposing MEGA.
(The above is from Quoting from – https://opposingmega.wordpress.com/2016/10/28/dr-nigel-speight-signs-opposing-mega/)
Many thanks.
Hi Peter, it’s the bio-psycho-social model, which has been a growing movement in recent years, but now that they are actually saying out loud that psycho-behavioural therapies treat the biology in ME/CFS, they hopefully begin to sound more ridiculous to more people. This is the thrust of the Opposing MEGA counter-petition that David kindly hylerlinked to in his article – https://www.change.org/p/opposing-mega-a-vote-of-no-confidence-in-mega-research-for-me-cfs
This document provides useful background to the BPS movement in UK, updated Sept. 2016 – http://www.margaretwilliams.me/2016/proof-positive-revisited.pdf
From this catalogue of articles –
http://www.margaretwilliams.me/
Hi Alice, firstly, sorry about your daughter’s illness and wishing her well. That’s quite incredible (though sadly not surprising) to hear that Esther Crawley was casting such aspersions on the work of the Norwegian rituximab researchers, as they are among the most highly esteemed researchers in the world for their work on ME/CFS. With regard to UK, a rituximab trial is planned to take place in Norwich (the base for the Invest in ME Research Centre of Excellence for ME) and Dr. Fluge is visiting Norwich in January to discuss and will also give a lecture http://www.cofeforme.eu/
It is so bizarre that supposedly respectable academics have been allowed to pursue therapeutic interventions based on completely unsubstantiated theories. In the PACE published literature they presented no evidence for their eminently testable psycho-social model of ME and completely ignored the overwhelming evidence for biological involvement.
Again in the FITNET study Prof Crawley is presenting a model of ME without any evidence and a complete failure to evaluate it. Certainly, given her use of the same therapeutic interventions as the PACE trials, it feels as though her theory is a post hoc attempt to justify continuing with those interventions.
It so feels as though what we are seeing with the spin and politician manoeuvring, what we are seeing here in the UK, is what might be termed ‘post-truth science’. Really this story would be better acted out in a soap opera or a medical thriller.
Again a very thorough article on the ongoing misleading actions by this kind of scientists in the UK and in the Netherlands. It is appalling that they still go on spreading this nonsense under the umbrella of scientific research.
Thank you for another clear and thorough analysis. I’m very glad to see you comment on the role of the Science Media Centre; there’s a lot to be said about their influence on the public view of ME in the UK.
Brilliant, rational, thoughtful article David. Absolutely on point. Patients just want proper science-Davis, Naviaux, Hanson, Fluge, Mella etc. The distinction between these real scientists, and Dr Crawley is remarkable. When will the UK wake up. As you noted, patients have legitimate concerns, because to be frank, this is an abomination of a study in all sense, especially methodology and criteria, let alone lack of objective measures (as always). We are not a ‘vexatious’ lot (as has now been dismissed in court), we are just desperate patients yearning for real science. Its really not that complicated from our side.
Hear hear. Well said man.
Thank you for this analysis and commentary. The absence of objective outcomes in studies of therapies designed to alter cognitions is disturbing. SMILE, Dr Crawley’s earlier study which compared the Lightning Process with specialised medical care, used the SF-36 physical function subscale at 6 months post-randomisation as the primary outcome, but also included school attendance as one of several secondary outcomes.
I contacted Dr Crawley recently about SMILE as it finished recruiting 3 years ago, and publication was planned for the end of the 2015. It’s still not been published but Dr Crawley replied that she hopes to publish in the next year. It will be interesting to compare questionnaire results with time in school.
http://www.bristol.ac.uk/ccah/research/childdevelopmentdisability/chronic-fatigue/smile.html
Yes, we can: the same reason the massively popular petition opposing someone’s knighthood was taken down even though it wasn’t based in the UK.
Excellent coverage again. My understanding of the role of the Science Media Centre is that they are not only cheerleaders, but also orchestrators of one-sided media blitzes for the psychosocial cabal, to the extent of creating news out of nothing like with the recent coverage of Crawley about to start a study. And also they have advised against or prevented coverage of most biomedical research.
Snap!
Oddly enough I was watching All the President’s Men as i was reading this. All I can think of is “Follow the Money.” Meaning, who benefits from this disease being portrayed as insignificant and psychiatric, and CBT/GET portrayed as “cures.”
May I add to your list of real scientists (as these are in UK) the Invest in ME Research team and are renowned scientists – Simon Carding and Tom Wileman (gut microbiome) Jo Cambridge (B-cells/rituximab) Angela Vincent (neuroimmunology) with Jonathan Edwards also advising on research strategy. Just because they don’t get mainstream media coverage (courtesy of the UK Science Media Centre) doesn’t mean they aren’t doing the work in UK as well as in collaboration with the researchers you mention in USA, Europe (and Australia). This is all initiated, organised, and currently funded by patients and parents/carers. Invest in ME Research Advisory Board also includes Ian Gibson – http://investinme.org/IIMER-ab.shtml