IS – Ian Sample
MS – Michael Sharpe
KG – Kim Goldsmith
In May activists rallied in New York to call on governments worldwide to take action for people living with ME or myalgic encephalomyelitis.
Their placards bore messages like ‘You can’t ignore ME now’ and ‘ME/CFS hidden suffering’. CFS stands for chronic fatigue syndrome and you often see ME and CFS mentioned together in the media.
MS: we don’t know whether they have a single cause or multiple causes so I think we should not necessarily assume that these are the same conditions although they do seem to have overlap.
The fact that we don’t know what causes the disease or diseases, has long been a source of contention.
Despite the discovery of biological abnormalities in patients, what is happening in people affected is far from clear.
KG: the disease is clearly really unpleasant and I do really feel it must be a really difficult thing to have happen to you..
Without an identified cause interventions have focussed on strategies like CBT or GET.
They aim to help sufferers cope with their symptoms.
But some see the focus on the mind as a dismissal of it being a physical disease. They say it suggests that the illness is ‘all in the mind’. It’s an argument that researchers we spoke to reject.
KG: It doesn’t mean that the disease might not have a physical cause it may very well have, have, such a thing.
For researchers in the field it can be a challenging environment to work in. Some face regular abuse, and campaigns that target their funders and institutions.
MS: any one who works in this field is familiar with receiving a fair bit of criticism and sometimes some very frank hostility.
IS: were you attacked personally?
MS: Any of your listeners wants to go on Google or Twitter and have a look they will see some not very pleasant comments to me.
But now the fraud debate may have reached another level. The Reuters News agency reports that a major journal is about to withdraw a much cited review of interventions for CFS amid what it calls fierce criticism and pressure from activists and patients.
KG: If its being withdrawn without there being any serious scientific fraud or major problems with how the review is done, and its being withdrawn anyway, I do find that a worrying precedent.
I’m Ian Sample and on science weekly we are looking at what role the public should have in research.
We are not looking at the cause of CFS or its potential treatment but asking whether boundaries should exist to protect the integrity of science.
MS: So my name’s Michael Sharpe. I’m Professor of Psychological medicine which is psychiatry and psychology applied to medical illnesses and I work at the University of Oxford.
IS: What is CFS?
MS: CFS is pretty much what it says on the tin. It’s a illness, eh, which is characterised by the symptom of severe fatigue, that’s usually made worse by activity, and it’s defined as having lasted at least 6 months and being disabling, that’s preventing the people, that’s preventing the sufferers doing the things that they would normally do.
IS: and how does that relate to ME we often hear these two terms together.
MS: the term ME stands for myalgic encephalomyelitis. This term was coined in the 1950’s to describe an epidemic of symptoms in the staff of the Royal Free Hospital which occurred during a polio epidemic. And in modern usage is used to describe complex symptoms without a clear medical cause which is often but not always characterised by severe fatigue. So they are often conflated as CFS/ME or ME/CFS. In fact they may not be identical. And the problem is where you have illnesses that are essentially defined on symptoms, we don’t know whether they have a single cause or multiple causes. So I think we should not necessarily assume that these are the same conditions although they do seem to have overlap.
IS: lets talk about CFS. Do we know what the cause of it is?
MS: Well CFS stands for Chronic Fatigue SYNDROME, and a syndrome defines a collection of symptoms, its not the same as a disease where we know the cause, so in simple terms we don’t have a single cause. We do know that it can start following infections. We do know that people who have suffered depression in the past are more likely to get it, we do know there may be a small genetic loading. But n terms of a specific clearly identifiable cause you are right we don’t know.
IS: and presumably not knowing the cause makes it that much harder to treat right?
MS: Well that is true to some extent. But a lot of treatments in medicine probably an embarrassingly large number are being designed and tested and are used without knowing the cause.
It would help to know the cause but that doesn’t mean we shouldn’t try treatments.
IS: Michael and a fellow scientist Kim Goldsmith were part of a team that looked at the efficacy of treatments for these patients. Kim is a Biostatistician at Kings College London.
She worked with Michael on the PACE trial. A large scale five year study, that compared a number of interventions aimed at helping patients.
KG: So, one of those was CBT, that addresses sort of how people might think about their symptoms and behave towards their symptoms. Another was GET, which a physiotherapist delivers, and with the person with the illness to gradually increase their exercise. The third one was called Adaptive Pacing therapy, and the idea of that is that you do things when you feel you have energy and you rest when you don’t. And those three treatments were compared against a control er, a treatment which was given to everyone on the trial.
IS: How effective do those treatments seem to be?
KG: We see those as moderately effective treatments. Erm so, as we also indicated in the paper, it probably means we need, we do need other treatments because these ones are moderately effective.
[she seems to have been cut off at this point]
IS: The paper received some criticism; one study published in the journal BMC Psychology reanalysed the raw data from the PACE trial and found that CBT and GET had quote ‘no long term benefits at all’.
I put this to Michael.
MS: Well we’ve been very carefully through that paper and of course it makes some points about the limitations of these kind of trials which are actually described in the paper. But we don’t agree with their reanalysis and we don’t think it actually changes the results of the trial.
IS: But the criticisms didn’t stop there.
MS: Any one who works in this field and that’s not just the authors of the PACE trial, anyone who does this kind of research into CFS is familiar in receiving a fair bit of criticism and sometimes some very frank hostility.
IS: were you attacked personally?
MS: Any of your listeners wants to go on Google or twitter and have a look they will see some not very pleasant comments to me.
IS; Have you in some sense become a lightning rod for this kind of reaction?
MS: er no I don’t think that’s true, in fact I know it isn’t true, because I am in touch with colleagues in a number of other countries and I’m sorry to say they pretty much all receive the same kind of treatment.
Erm so colleagues here, in Norway, in the Netherlands, in Denmark, so some extent in the US, so this is a general phenomenon.
IS: Why are these campaigners so opposed to things like GET and CBT?
MS: well I can only answer that in terms of what they say when they are criticising. I think there are very strongly and sincerely held views about the nature of the illness. That it is in their mind a neurological illness that has no psychological or psychiatric aspect to it, and it could not be changed by such a simple thing as changing activity. Therefore, evidence which is contrary to that very strongly held view must be false and actually they feel its challenging the narrative that they favour, not only must it be false but it must be removed. So I think this is a clash of very strongly held views about the illness between what the science is telling us and what people passionately believe about the illness they often have themselves.
IS: Critics argue that the focus on psychological treatments implies that the illness is all in the mind, that it does not have a physiological component.
IS: there seems to be something going on here could be where some patients may see that the suggestion that a sort of psychological intervention could work for them as dismissing it as a mental condition as not a physical disease. Is that the case?
KG: no that’s not the case at all. That’s not how I see it, and I know its not how my colleagues see it either. These treatments, I think the way that we all see them is that they can help people cope with their symptoms, you know, and we are in a situation where those are the only evidence based treatments and that is kind of where we are at present.
And it doesn’t mean that the disease might not have a physical cause, it may very well have such a thing.
KG: You know I work at the Institute of Psychiatry at Kings and many many of my colleagues also talk about the, you know this distinction between mental disorders or illnesses and physical ones, they don’t really see why theres a distinction there because you know it’s the brain and so on is part of the body as well of course.
IS: But Michael and Kim decided not to work in this field anymore they’ve moved on to research other areas. I asked them why.
KG: I think I would be concerned about taking up more research in this area and this actually I did feel concerned about this in general because again, I don’t know for sure, but I think some scientists have probably stepped away from working in this area because of some of the backlash
that can sometimes happen.
IS (to MS): do you still do research in this area?
MS: um, actually I’m not, no. I mean there are a number of reasons for that I’m doing other types of research. But I certainly wasn’t particularly
encouraged to carry on with it. The reason is, not because of the hostility, but because the complaints that you get have to be responded to by the institutions and then they ask you to respond. And now seven years on, after publishing this trial I’m still spending a half a day a week responding to these kind of complaints and criticisms, and that’s not really sustainable and it gets in the way of research.
IS: As you have said this study is something like seven years old now, erm are you happy to be still coming in to talk about it after all that time?
MS: Well its very nice of you to invite me to talk about my research scientists always enjoy that, but you are right that it is old news and to be honest one would like to just let go of it. But I think it is very important to defend the work of myself and a very large number of colleagues who worked on this trial, and other researchers. And I also think it is important to speak up about the harassment that journals and funders and other institutions get, so that these discussions are in the public domain.
IS: Before the break I spoke to Michael Sharpe and Kim Goldsmith about their experience of doing research into Chronic fatigue syndrome. Michael spoke about the abuse he received Kim mentioned her concerns about continuing to research the illness. They wanted to speak to us because they worried about how attacks on science could harm research.
I quoted a Reuters report at the top of the podcast. They reported that a highly respected science journal is considering withdrawing a much cited review of interventions for Chronic Fatigue Syndrome amid quote ‘fierce criticism and pressure from activists and patients’.
We wrote to the journal in question, the Cochrane Review. We asked them what the nature of the complaint was. Was it from a member of the public? Or a scientist? And finally whether they were withdrawing the paper? They wrote back to say:
“The Cochrane review of Exercise therapy for Chronic fatigue Syndrome was last updated in 2014 and is subject to an ongoing process of review and revision following the submission of a formal complaint to the Editor in Chief. Cochrane carefully considers all feedback and complaints and revises when appropriate to do so. The review author team have advised us that a resubmission of this review is imminent. A decision on the status of this review will be made once this resubmission has been through editorial process, which we anticipate will be towards the end of November of 2018.”
IS: When we enquired about the nature of the complaint and what it pertained to they didn’t comment. We also contacted the lead author.
She also preferred not to comment at this stage.
So it is unclear whether Cochrane will withdraw the review or not.
It’s also unclear precisely what the complaint was about.
MS: The Cochrane Collaboration, as your listeners might know, is regarded as a gold standard of evidence in the medical field and what Cochrane do is very rigorous systematic reviews and metanalyses of existing clinical trials to provide the best answer that we can to questions of clinical treatments.
So what the Cochrane does in terms of evidence is very important. What I am told is that my colleagues who did a systematic review of graded exercise and Chronic fatigue syndrome, they were told that their reviews were going to be retracted specifically because of complaints that they’d had. So if that were to be the case, that would clearly be very alarming because that would be an example of the evidence that is available to doctors and the public being changed not by a change in science but by campaigns.
IS: It was a concern echoed by Kim too.
KG: If the review is being withdrawn because of concerns that people have about the treatments that may, may or may not have a strong scientific basis, so be so, you know, bad as warrant withdrawal of the review, and then this is, becomes a common practice in other areas, then I feel that this is, you know, influencing science in an unscientific way potentially. That’s really, I think, that’s really my concern.
IS: If it is campaigners that have lead Cochrane to consider pulling this paper I wondered why do they hold so much sway?
KG: Um, yeah I guess in some ways I’m not sure. I s’pose we have to ask the, for example people at the Cochrane Collaboration, but I do think the disease is clearly, its really unpleasant, and I do really feel, it must be a really difficult thing to have happen to you, and also I think because we don’t know much about what causes the disease, I think maybe that’s why because it’s not clear what causes the disease and these treatments ehm, are more trying to help people cope with symptoms rather than getting at the cause, perhaps that’s why these criticisms do have sway.
IS: What do you think of the consequences of this where you have editors
looking at withdrawing a review of research, do you have scientists stepping back from doing the research itself?
KG: Well I think, I think its er, it’s really dangerous, I mean, the thing about this particular situation is, these are, as far as I know, the only two evidence based treatments that exist. So my concern is that if these treatments were not available anymore that there would be no treatments for people with Chronic Fatigue Syndrome, and I think that given that there is an evidence base for moderate effectiveness I think that would be potentially quite serious.
IS: But this is not just happening in the field of Chronic Fatigue Syndrome.
MS: Um, but as a phenomena, this is happening across a number of fields of science, and very important areas like vaccination.
I think it is a challenge for how we communicate science, because if our usual systems of peer review and dissemination are to be altered by essentially pressure groups, that will mean that the information available to the public will not reflect the science, it will reflect the interest of pressure groups. And if one’s passion is about science I think that is a cause for concern.
IS: A lot of this work is publicly funded and presumably that means that the public have some input at some point along the way. Where do they come in? Where does the public voice, which presumably includes campaigners, where do they have a like a legitimate entry point for discussing this, and steering this work? And where do they maybe not?
MS: I think that most people that do health research are used to and indeed welcome and, are required to in many cases, involve members of the public and patients in their research and that’s in formulating the questions, it’s in designing the studies and making sure they’re suitable for the participants, and its interpreting the result, their results, and it’s indeed in helping disseminate the results. So that’s really er good practice now and I think most people would do that.
Clearly it’s also quite reasonable for the public and campaigners to criticise research when it’s published. And the standard way we have of doing that is to write a letter to the journal that is sounding a little antiquated now but that is our system, or they can write blogs, and so on.
I think where the line is crossed for me is when they campaign to have research removed from the public record because it offends them in some way. And I think, that for me is the line that’s too far.
IS: What are scientists to do, do you think, in situations like this?
MS: I think it’s difficult. I hope that scientists would on the one hand listen carefully to criticisms made because sometimes they might be good ones, um but at the same time stand up for the conclusions of the data, and not allow themselves to be silenced or swayed just because someone’s shouting. I think it’s a challenge across science and it’s one we’re gonna have to work out how we deal with it.
IS: And what about patients in this? Because, for a patient going online and looking at CFS, ME, things come up on the PACE trial things come up on the Cochrane review, I don’t know how a patient is supposed to understand what is right and what is wrong.
MS: Well I think you’ve put your finger on the nub of this whole issue.
Is, if the information becomes so confused that patients and indeed physicians don’t know what the right treatment is, then patients can’t benefit from treatment that has been found in research. I think it is very important just to reiterate the point that my colleagues that run clinics for people with Chronic Fatigue Syndrome tell me that most people they see are quite happy to undertake these treatments and that the people campaigning do not represent all patients. They represent a smaller group of people, and it’s really important that the campaigners, to my view, don’t stop people who would like to have these treatments, from being able to have them.
IS: So how much say should the public have in science? I think we can all agree that scientists can do more to engage with the public. And, as the public we need to take more interest and discuss the research we want.
The question is how?
Chronic Fatigue Syndrome is a debilitating condition that has an enormous effect on peoples lives. Clearly a better understanding of the illness is needed to better help those affected, and the only way that will happen is with more research.
