The Concept of ME/CFS

[Jonathan Edwards]

I am uploading here a draft of a review I am working on, on the concept of ME/CFS. I will probably send this to Qeios like the piece on feeding support. There is a lot of overlap but this one is designed for a different purpose.

The citations have not been filled in yet.

Thoughts very welcome.

 

[Jaybee00]

“People with ME/CFS say they do not have fatigue in its usual sense(s). “

I have fatigue/tiredness/exhaustion/lack of energy in what I believe is the usual sense.

 

[duncan]

Lots of good stuff here.

I'm a bit leery of referring to ME/CFS as a "concept" Feels uncomfortably close to idea or belief, and can illness belief be far behind?

 

[InitialConditions]

Hi Jonathan. Can you—or perhaps someone else—please upload a PDF version? Cheers.

 

[Andy]

Hi Jonathan. Can you—or perhaps someone else—please upload a PDF version? Cheers.

Try this, any issues let me know.

 

[InitialConditions]

Try this, any issues let me know.

Thanks Andy

 

[Trish]

Paragraph 1

UK National Institute for Clinical Care and Excellence (NICE)

should be
UK National Institute for Health and Care Excellence (NICE)
https://www.nice.org.uk/
______________

Secondly, symptoms relapse or worsen with a time relation to exertion that is delayed and prolonged but also unpredictable, in a way not explained by immediate physiologic change, or local tissue effects (in contrast to post-exercise muscle soreness). This is known as post-exertional malaise (PEM). The clearest illustration of PEM is popularly known as a ‘crash’.

A good summary of the effect on symptoms. Do you think you need to expand this to include that the exertion that triggers it is slight, and that PEM includes loss of function as well as increased symptoms?
I'm not sure what the last sentence means.
______________

I have not mentioned fatigue. People with ME/CFS say they do not have fatigue in its usual sense(s). People with renal failure or rheumatoid disease with fatigue do not ‘crash’. Moreover, fatigue associated with most conditions is often best relieved by sitting in a comfortable, but upright, chair.
....
More importantly, it is very uncertain that the core symptom of ME/CFS is even subjectively the same.

I think some pwME do say they have fatigue, though different from healthy fatigue or tiredness.
The last sentence seems to imply 'the core symptom of ME/CFS' to mean fatigue, not PEM. Which do you mean?
_________________

These 2 paragraphs stand out to me as central, perhaps worth reiterating in a conclusion:

Importantly, where ME and CFS have tended to be labels for what people guess the process might be (neurological or psychosocial), and may be empty, ME/CFS covers a concept that is undeniable – that there are a number of people in the community seriously disabled by a particular pattern of symptoms, a small number of whom die as a result.

There is more work to do to get the concept of ME/CFS as clear as it could be. More precise delineation of symptom patterns, perhaps backed up by detailed posture-sensitive actimetry studies, could be very useful. Nevertheless, a huge amount of progress has been made in the last decade. Clear clinical definition has allowed unproductive theories such as deconditioning cycles to been shown to be off target. To a considerable extent progress has been driven by citizen scientist patients, or carers and friends with biomedical science skills. The science community must swallow the unpalatable truth that it is the patient community that has brought methodological rigour to the subject, not the academics. For more than one reason, physicians should listen to patients.

__________________

Worryingly, not only has the psychological medicine approach failed but its proponents have shown no recognition of their lack of understanding of either the illness or research methodology (White, Anomalies paper). This is hard to square with the fact that there is no evidence for any reduction in the prevalence of the illness and people are still dying of avoidable dehydration.

I'm not sure the 'reduction in prevalence' is a good argument for psych factors being wrong.
And should it be 'dehydration and starvation'?
__________________

ME/CFS has been flying under physicians’ radar for some years. I see an important driver for this as the fashion for ‘multidisciplinary teams’. Where we have complex technical knowledge in several disciplines relevant to the care of someone with renal lupus or head and neck malignancy, multidisciplinary team meetings are essential, but that has little to do with the diffusion of personal responsibility and decision-making that allows someone with ME/CFS to be sent off to three different therapists, none of whom have any idea what modalities are of clinical value – because nobody knows.

I think risk of short and long term harm specifically from prescription of exertion and sensory challenges should be mentioned.
___________________

The names used go in a never-ending progression, each with a rather different ‘meme’ attached: neurasthenia, ME, fibromyalgia, functional neurological disorder, and so on.

Does FM belong in that list, and why not CFS?

 

[Sasha]

This is fascinating! Thanks so much for writing it, @Jonathan Edwards, and for staying with us and listening and working with us all these years, which has enabled you to produce this synthesis.

Now I see why you were so interested in us having to lie flat. I'd thought that there were PwME who didn't consider themselves to have OI. Do you think it matters that not all have OI?

I think it's a superb piece of writing but that both the title and the first para undersells it by not flagging up what the payoff will be for reading it. I wonder if we can come up with a better hook for this important work?

I'm curious about Qeios, and couldn't really grasp what it is from its website - is it essentially a journal with open peer review? Is a publication in it likely to get attention? Is the idea of publishing there to avoid weird BPS-sodden gatekeepers at the other journals?

 

[Trish]

Summary: Why ‘ME/CFS’ is a worthwhile concept.

It is time for physicians to take ME/CFS seriously as an unsolved disabling illness, with no presuppositions about causal mechanisms. The time course of deterioration following exertion is not explained by any currently understood type of mechanism. Neo-Freudian psychodynamics or ‘predictive coding’ theories, as proposed for notional ‘functional neurological’ problems (which themselves appear to give the wrong predictions), have nothing coherent to offer. Metabolic deficits do not fit well either. Failures in immunological or autonomic control might explain things but even there, no simple story fits.
Some humility is in order. ME/CFS appears to be based on a process at the interface between central nervous and immune systems at a level that currently we have no way of modelling. Popular quips about ‘mind-body interaction’ replacing Cartesian Dualism simply reveal as deep an ignorance of great philosophers as of human biology. It might turn out to be as simple as another error of acetyl choline metabolism, with complicated secondary effects. It might involve misdirection of tissue-bound complement regulatory proteins. Who knows? The history of medicine is full of situations where illness is entirely misunderstood until some empirical finding makes sense of it. Until then there is a need to get as clear as possible the clinical concept we are trying to explain, so that research studies have the very best chance of finding clues to what is going on.

I don't think the summary should include concepts not already covered. I think these paragraphs should be earlier in the article.

A summary would, I think, draw out the key points from the whole article.

_______________-

A very worthwhile article, thank you.

 

[Sasha]

I might even suggest that we need people who have not been trained in disciplines where uncritical acceptance of theory-based practice is the norm. Physiotherapy and clinical psychology come to mind.

This wording seems to suggest that physiotherapy and clinical psychology are the people who haven't been badly trained. How about:

I might even suggest that we need people who have not been trained in disciplines such as physiotherapy and clinical psychology where uncritical acceptance of theory-based practice is the norm.​

 

[Nightsong]

Yes, a lot of very good material here.

Ramsay must have thought that 'chronic ME' was not just post-viral fatigue

His views seemed to evolve over time. In his earlier "Infectious Diseases" textbook (where he covered both glandular fever and "benign myalgic encephalomyelitis" in separate chapters) he said of glandular fever that "[p]ersistent fatigue and inability to concentrate are common after infectious mononucleosis and may persist for several weeks or months but ultimate recovery is assured". Later he seemed influenced by the "chronic mono" literature which mainly came out of the US (in his well-known book "Myalgic encephalomyelitis and postviral fatigue states" he draws attention to it, p55). So in his later works he's talking about post-viral fatigue states as well but doesn't abandon the idea of a discrete chronic viral infection that characterised his earlier work.

A couple of very minor things to tweak: "people ME/CFS"; "UK National Institute for Clinical Care and Excellence" (think it's ..for Health and Care Excellence); "to been shown".

 

[Jonathan Edwards]

I have fatigue/tiredness/exhaustion/lack of energy in what I believe is the usual sense.

OK, but a lot of people with ME/CFS make this distinction. Maybe I should add 'Many people' which would imply that there is a difference from healthy fatigue at least but also that there are differences in the way people describe it.

 

[Jonathan Edwards]

I'm a bit leery of referring to ME/CFS as a "concept" Feels uncomfortably close to idea or belief, and can illness belief be far behind?

I think that may be stretching things. Diabetes is a concept, disabled people is a concept. I am trying to make the point that ME/CFS is useful as a syndrome concept - the nature of which I go on to describe. GPs may believe anything but I am saying quite clearly that what they should be believing is that there are hundreds of thousands of people with significant disabling symptoms out there who need help. Which there are.

 

[Kitty]

Really interesting read, thank you!

First thoughts:

ME/CFS appears to stand for myalgic encephalomyelitis/chronic fatigue syndrome but in an important sense it stands for neither. It is a newer concept, differing from both.

This seems to me so important that I'd put it in its own paragraph. Possibly even starting "First, ME/CFS appears to stand for...".

I have not mentioned fatigue. People with ME/CFS say they do not have fatigue in its usual sense(s).

This may need clarifying a bit. People do talk about fatigue, but the difference is that they describe feeling exhausted and ill, the way people do with 'flu.

It reminds me of the game of snakes and ladders in that a person coping quite well may, at any time, suddenly crash and return to bed (a snake) and, sadly much less often, may also, after struggling for months or even years, suddenly experience relief (a ladder). If that follows a therapy, it is often touted as a success, but so far there is no reliable evidence that any specific modality helps.

Would be useful to say plainly in the final sentence here that the timing of the ladder and the treatment could be a coincidence? (Though as the intended audience is probably medics, it might already be plain enough.)

I might even suggest that we need people who have not been trained in disciplines where uncritical acceptance of theory-based practice is the norm. Physiotherapy and clinical psychology come to mind. (Although there are individual physiotherapists and psychologists who, perhaps despite training, see the wood for the trees.)

This came across as a bit unclear to me. At first read it seemed to be recommending physiotherapy and psychology as useful disciplines, rather than suggesting they tend to be uncritical.

Maybe recasting it slightly would help. "I might even suggest that we need people who have not been trained in disciplines where uncritical acceptance of theory-based practice is the norm (physiotherapy and clinical psychology come to mind). Although there are individual physiotherapists and psychologists who, perhaps despite their training, see the wood for the trees."

 

[InitialConditions]

"I have not mentioned fatigue. People with ME/CFS say they do not have fatigue in
its usual sense(s). People with renal failure or rheumatoid disease with fatigue do not
‘crash’. Moreover, fatigue associated with most conditions is often best relieved by sitting in
a comfortable, but upright, chair. Interest in fatigue in systemic disease has grown recently
and there has been a tendency to see mechanisms as generic. The use of pathway specific
drugs such as TNF inhibitors has, however, shown us that even for other systemic illnesses
mechanisms are heterogeneous. More importantly, it is very uncertain that the core
symptom of ME/CFS is even subjectively the same."

I am with Trish on this. The last sentence is unclear and seems to imply fatigue as the main symptom. I would delete this sentence and expand the first part of this paragraph, because it is a key concept: many of us feel that our experience cannot be reduced to even a more severe version of 'normal fatigue'

 

[EndME]

Thank you for this piece and for spending time to listening to patients for many years.

Paragraph 1
I'm not sure the 'reduction in prevalence' is a good argument for psych factors being wrong.

I agree.

Unpleasant symptoms in ME/CFS include nausea, orthostatic intolerance, pain, (hence ‘malaise’) and sensitivity to environmental stimuli such as light, sound, physical contact and odours.

I wonder whether "neurocognitive deficits" or whatever the right wording might be for what is often also described as "brain fog" shouldn't be mentioned here as well. It is part of most clinical criteria and my impression is that patients often describe it as very disabling, if they aren't bedbound it may be one of the reasons why they cannot resume their careers and lives. I mention this specifically because this is an area nobody has ever seemed to listen to patients. Different researchers have used tools from other diseases to quantify cognitive deficits, sometimes yielding positive results, sometimes yielding negative results, but no one has asked themselves what is it exactly that patients are describing, what kinds of patterns exists and how should we go about quantifiying it?

 

[Jonathan Edwards]

UK National Institute for Health and Care Excellence (NICE)

Thanks

I'm not sure what the last sentence means.

I shortened it and it may not quite make sense. I will review it.

I think some pwME do say they have fatigue, though different from healthy fatigue or tiredness.

The last sentence seems to imply 'the core symptom of ME/CFS' to mean fatigue, not PEM. Which do you mean?

I guess I mean the core symptoms, which has to be plural. The ones I have listed earlier.

I'm not sure the 'reduction in prevalence' is a good argument for psych factors being wrong.
And should it be 'dehydration and starvation'?

Yes, that needs tidying.

I think risk of short and long term harm specifically from prescription of exertion and sensory challenges should be mentioned.

It is mentioned briefly somewhere else. I am deliberately not making any claims for which we have no solid evidence, even if it seems likely to be a real risk.

Does FM belong in that list, and why not CFS?

I think FM belongs because it is another of these theory-drive concepts - in this case based on 'central sensitisation'. CFS probably isn't. It is all the people who go on being fatigued. The problem with it is different - that it suggests that you can bundle all these people together. Some BPS people may have theories of CFS but if anything they tend to prefer CF.

 

[Jonathan Edwards]

Do you think it matters that not all have OI?

I think OI is often used as a more specific term for not being able to tolerate being upright or even half upright. What I am suggesting is that this may be the sharp end of a more general set of symptoms that make people seek relief in lying flat at least some of the time during bad patches. I don't claim that everybody even feels that but my sense is that this has something to do with what is different about ME/CFS.

I know that Alastair Miller has been criticised for his views on causation and treatments and what he says justifies that criticism. However, in his 2015 YouTube video he describes the clinical pattern of ME/CFS in almost exactly the same way as Amok Bansal has and I have here. As I say later, there is still room for getting the description clearer but I sense that physicians who talk to people with ME/CFS over a long period tend to get this same picture.

Suggestions for a better title are welcome.

I'm curious about Qeios, is it essentially a journal with open peer review? Is a publication in it likely to get attention? Is the idea of publishing there to avoid weird BPS-sodden gatekeepers at the other journals?

It is an online journal set up by a UCL staff member with also Italian input. I use it partly because I think it is the way we should publish - with no kudos for going through hoops therefore no point in wasting energy on writing hack stuff. The open, signed, peer review system is excellent and allows good dialogue. Papers on Qeios will get attention if they are worth reading. Nobody really cares where something is published if they think it is interesting. I notify relevant people about these papers going up and I have had some comments indicating that word has got through to physicians. And yes, the gatekeepers at places like BMJ are to be avoided at all costs.

 

[Jonathan Edwards]

I don't think the summary should include concepts not already covered. I think these paragraphs should be earlier in the article.

In this case I disagree, Trish. I think you are being editorish. When Ludwig Wittgenstein wrote the Blue Book everything was all over the place but it was perfectly clear what it was about and an intriguing read. Surprising people at the end seems to me something almost imperative.

 

[Jonathan Edwards]

This wording seems to suggest that physiotherapy and clinical psychology are the people who haven't been badly trained. How about:

I might even suggest that we need people who have not been trained in disciplines such as physiotherapy and clinical psychology where uncritical acceptance of theory-based practice is the norm.

Yes that was another edit down that lost traction.
Well worded.