By David Tuller, DrPH
In a new paper, a team of investigators from the University of South Australia in Adelaide, Australia, describes a “pilot feasibility” trial for an ME/CFS intervention focused on physical activity. The trial fell dramatically short on recruitment efforts—a failure that the investigators appear to explicitly blame on the patient community rather than any possible shortcomings in their approach. The paper, in the journal Translational Exercise Biomedicine, is called “Pacing, conventional physical activity and active video gaming to increase physical activity levels for adults with myalgic encephalomyelitis/chronic fatigue syndrome: a pilot feasibility study.”
A major issue seems to be the basic assumption behind the study–that the question of whether physical activity is rehabilitative for ME/CFS remains unsettled. That assumption is false. The question has been asked and satisfactorily answered: Interventions focused on graded exercise and boosting physical activity do not lead to improvement or recovery. As the UK’s National Institute for Health and Care Excellence (NICE) found in its assessment of the literature for its 2021 guidance on ME/CFS, the quality of evidence in favor of such interventions is either “very low” or merely “low.”
And yet, the Australian investigators write this:
“Physical activity-based (e.g. exercise therapy) interventions are not well understood in this population, with the effectiveness, safety, optimal intervention design, and method of delivery yet to be properly elucidated. A review of eight physical activity-based interventions concluded that whilst definitive conclusions were difficult to draw, it was likely that physical activity had a positive effect on fatigue experienced by adults living with ME/CFS compared to usual care or passive therapies.”
The “review of eight physical activity-based interventions,” cited authoritatively to support the claim that the science is unsettled, is a discredited Cochrane review last revised in 2019. The investigators do not acknowledge Cochrane’s own admission, at the time the 2019 version was published, that the review was not fit for purpose and required an update. In December, Cochrane did an abrupt about-face and cancelled the planned update. The largest study in the Cochrane review was the fraudulent PACE trial, with the overall body of research in this domain marked by a range of serious flaws–as noted by NICE.
Based on their apparent misunderstanding of the state of play, the investigators decided that a gentler form of physical activity might be the answer. They settled on active video-gaming (AVG). What is that? (I didn’t know either.) From the study: “Active video gaming can be broadly summarised as any video game that requires participant trunk and limb movements for game play. Games can be played at home, standing or sitting, and for short periods (e.g., 1–2 min).”
The plan was to divide participants into three arms: pacing alone, pacing combined with conventional physical activity, and pacing combined with AVG. Both the conventional and AVG interventions included incremental increases in activity every two weeks, but these increases were framed as “symptom-contingent.” In other words, participants could decide not to increase their activity, or even reduce it, depending on their response to the intervention. The study included a range ot subjective and objective outcomes.
(As a side issue, the investigators seem to regard pacing as a theory-driven practice rather than a self-help strategy used by patients to avoid relapses. “Pacing draws on elements of self-determination theory by allowing participants to self-regulate their activity within their perceived boundaries, promoting intrinsic motivation and sense of control,” they write. Ok, I guess so. But there’s no need to engage in elaborate theorizing to explain why people with ME choose to pace.)
Unfortunately, recruitment was dismal. Despite an estimated 11,000 people with ME/CFS in South Australia, only a few dozen expressed interest during the 17-month recruitment period. Ultimately, 15 participants enrolled, 12 completed the assigned intervention, and 11 provided follow-up data six months after the intervention. As a result, the investigators concluded that “the feasibility of the intervention was deemed to be low.”
In seeking to explain the recruitment problems, the investigators have successfully found others to blame:
“Recruitment may have been negatively impacted by a small but vocal number of people in the ME/CFS community spreading misinformation about the study via social media and patient support groups. The motive of these individuals was not known although their messaging insisted that (1) physical activity is harmful, and (2) research funds should only be allocated to projects that directly seek to understand the pathological mechanisms and lead to an eventual cure. Although the latter point is entirely understandable, the researchers and stakeholder advisory group members believed the desire to maximise the quality of life for those already suffering from such a debilitating disease is also important, and worthy of research investment.”
Note that the passage does not share the “misinformation” purportedly being spread by these “vocal” patient advocates, although it highlights the critics’ positions on the dangers of physical activity for ME/CFS patients and the need for biomedical research. It is hard to take issue with either of those stances. When those whining about “misinformation” do not back up their statements with examples, it suggests that the issue might not be “misinformation” but differing interpretations of the existing data. The investigators write that “the motive” of these critics was unknown, but their motive appears to have been very clear and eminently reasonable–to prevent harm to patients and to ensure that research funding isn’t wasted.
Participants who completed the AVG intervention reported finding it helpful in some ways; the investigators therefore reported that it was “viewed as acceptable and potentially useful.” However, no one increased their physical activity levels—which was the point of the intervention in the first place. Oh, and did I mention that they had a huge recruitment problem?
We can all agree that it would be great, as the investigators write, “to maximise the quality of life for those already suffering from such a debilitating disease.” But misrepresenting the medical literature in order to pursue research that patients obviously reject does nothing to improve anyone’s quality of life. The critics who challenged the rationale for this research were right.
Same old – blame others when things don’t go well or to plan. If the authors wanted to alienate patients and patient advocates, then I’d say that they probably succeeded in doing that. Could they believe that they’re talking behind closed doors here? Maybe they haven’t yet grasped that these days there will be patients and patient advocates who take an active interest in the medical literature, conference presentations etc of their diseases/conditions and that badmouthing them or making insinuations against them in those places is likely to be found out. I think they’d do well to reflect on what they both wrote and omitted to write about people in the ME/CFS community. Was it fair or helpful to attempt to apportion blame without providing any evidence of wrongdoing? Would they treat their friends like that? Could they perhaps, during their careers, have learned resentment of patients and their advocates? I suggest that they read this -https://bjgplife.com/unlearning-resentment/ and consider a less belligerent approach.
“Active video gaming can be broadly summarised as any video game that requires participant trunk and limb movements for game play. Games can be played at home, standing or sitting, and for short periods (e.g., 1–2 min).”
Sounds like Wii Tennis or a similar game. Which I found to be great … when used in conjunction with pacing.
Going by the adverse events information in the Supplementary Material, it seems that two participants experienced increased post-exertional malaise – 1 (of 9, so around 11%) in the AVG + pacing group and 1 (of 10, so 10%) in the conventional physical activity + pacing group. The former apparently stopped the AVG exercise but continued in the study. The paper’s section on adverse events indicates that one patient in the AVG group had PEM due to exercising for longer (rather than experiencing increased PEM from the intervention). So was this new PEM or an increase in the duration or severity of what they usually suffered at the start of the study? If the participants had been selected at the start as those diagnosed by the CCC criteria (so known to experience PEM) there would be little problem with this discrepancy, as all participants in the trial would have presumably had PEM from the outset and it would be clear that the participant in question had indeed experienced an increase in severity or duration PEM due to the intervention. But with the CCC criteria being added later to the protocol, isn’t there a slim chance (perhaps 1 in 11, going by the cardiopulmonary exercise testing) that a participant who hadn’t previously experienced PEM could have gained a new problem as a result of taking part in the intervention? Can we know for sure that this didn’t happen?
I’ve been wondering what happened to this study which was being designed c 2016, quite a long time ago. There was considerable discussion among patients at the start about the design and assumptions of this study and the authors were told of these. Also mentioned were doubts about the ability of patients to tolerate the AVG due to sensory issues. All to no avail. I’d begun to think the whole thing had collapsed and the result is not far from it!
I have ME/CFS. I use every ounce of physical capacity that I have just trying to live through a day. I do have periods of part- remission and as soon as I have those I am more physically active because I am ABLE to be. I would NEVER join a trial that suggested more physical activity….that risks worsening the illness. Believe me….I would be up running if I could be. You cannot imagine the pain and ‘hit by a truck’ experience that comes in post-exertional malaise.
Pacing allows “participants to self-regulate their activity within their perceived boundaries, promoting intrinsic motivation and sense of control.”
I do NOT regulate my activity based primarily on my perceptions, but on the results of continuous heart rate monitoring – a technique I was trained in through a Workwell seminar. Successful pacing requires me to acknowledge that I am NOT in control of my exertional boundaries and must subordinate my motivations and desires to my physical limitations. Sadly, I was not introduced to this disease management strategy until I had been ill for over a decade and my ME/CFS worsened significantly as a result.
While it is understandable (and something I’d like too) to desire the details of the cited misinformation, as an academic I would just point out that it is common in every setting including research settings to not delineate every detail of every thing listed in any given writing. I have not read the whole study yet and I appreciate you bringing it to our attention so that I can do so, but I would disagree with your assessment that, based on the limited excerpts you provided here, your case was made. This situation mirrors the situation you are critiquing, in that you may have plenty of proof in other particulars of the study, just like the writers of the study may have plenty of proof of their claim in other piles of their research notes. It is worth remembering too that the writers of such study summaries are often supervised by editors who decide what stays in the published version and what doesn’t, so the more fruitful action if one wants to add rigor, is to contact the authors and ask for the citations, rather than criticizing them without due process. Personally, for me any type of video gaming at all would exacerbate every symptom I have, so it sounds like a nightmare to me, but I thought when I saw your original post with its title that you were going to be citing how the study overly relied on the free labor of patients who obviously have very little bandwidth to publicize their study and recruit participants. I can certainly vouch for that being the case since I would have participated in any study I saw and I’ve never seen this and I follow lots of related feeds. But instead, it seems you focused on one single detail and ripped into i without any of the same rigor you accuse the authors of ignoring. You might consider this for the future. You are a great writer and a persuasive voice, and I’d love to read your work when it can really analyze something that I can’t poke holes in immediately. I was surprised and honestly disappointed to not be able to agree with you, since you are making an important point…just without the requisite data we need in order to be credibly heard. Just something to consider.
Fool me once, shame on me.
Keep fooling me with the exercise slippery slope and thoughts – I’m not that stupid or misinformed any more.
We are connected. We read what David Tuller writes. We know what to ignore: anything that can make us worse.
We certainly don’t need the pressure and stress and authoritarianism.
To my mind, text that could besmirch the character of members of a patient community should be well-supported by evidence. I’m surprised that the peer reviewers and editor/s involved in the publication of this paper didn’t press for clear evidence to be supplied or for the removal of that section. Whatever the merits of the rest of the paper, to me it headed for the gutter at that point by encouraging hostility in a medical context.
More generally – surely mutual understanding must be the best way forward to achieve good patient care? If researchers are hoping for a good level of patient engagement and participation then I’d suggest that adopting a bunker mentality is probably not going to help with that.
Hi, June–Thanks for sharing your perspective. I understand, of course, as both a journalist and academic, that you can’t always fit everything into an article. In this case, the investigators went out of their way to blame patients/advocates for wrecking their recruitment prospects by disseminating “misinformation.” Leaving that accusation hanging out there without providing any evidence to support it is unacceptable, in my view–outrageous, really. And that’s especially so given that this patient community has been regularly defamed as purportedly harassing and threatening researchers and the like. They have an obligation to provide enough information for people to understand what they’re talking about. They failed to do so. Any decent peer-reviewer and/or journal editor should have raised this issue and asked them to provide evidence or remove the accusation.
I looked into doing this study and rejected doing it as the initial entry into the study was not restricted to people diagnosed with ME against diagnostic criteria that require the presence of post exertional symptom exacerbation. Given the broad entry criteria it is not certain that any participants would actually have ME (as per 2003 – CCC or later diagnostic criteria).
The authors blame the patients for not wanting to take part in their poorly designed study yet they ignored patient input and failed to address our concerns with their study. The authors talk about “perceived boundaries” which is galling. Our boundaries are real are physiological and NOT perceived. In preliminary discussions with the authors they appeared not to understand ME, rejected our physiological reality and didn’t appear to acknowledge the fact that OVEREXTERION triggers an exacerbation of symptoms for people with ME. The authors now appear to gas light the patient community and blame us for the poor take up rate of their flawed study. REST does reduce our symptom load and OVEREXERTION makes us worse.