Were historic 'ME' outbreaks really ME?

[chrisb]

This thread has been split from https://www.s4me.info/threads/bmj-pressure-grows-on-lancet-to-review-“flawed”-pace-trial.5444/page-6

Nothing Wessely and co have done targets ME, that is the whole crux. Their conflation with fatigue has caused chaos and untold harm. The PACE architects would often say in the 'small print' that ME and CFS may not be the same thing (or words to that effect) but then would go ahead and conflate anyway. We were fighting this conflation long before PACE. PACE is just the disastrous outcome of conflation.

They seem to play these word games all the time. I have been trying to work out the attitude to "hysteria". In their book Wessely, Hotopf and Sharpe say @p232 10.5 CFS and hysteria:

Thus hysteria, used properly as meaning conversion disorder, is rarely a tenable explanation for CFS. Three caveats are necessary. First in the context of epidemic forms of ME, explanations involving transmitted emotional distress ("mass hysteria"), merit attention. However, as we and others have also shown, such examples have little to do with CFS. Second...…"

My interpretation of this is that they confirm their apparent approval of the views of McEvedy and Beard for the epidemic forms, but clear themselves by saying that such cases are not CFS. I would be happy to have alternative views should others think me wrong. It seems like a case of Humpty Dumpty Syndrome-words mean what I want them to mean.

 

[Jonathan Edwards]

My interpretation of this is that they confirm their apparent approval of the views of McEvedy and Beard for the epidemic forms, but clear themselves by saying that such cases are not CFS. I would be happy to have alternative views should others think me wrong. It seems like a case of Humpty Dumpty Syndrome-words mean what I want them to mean.

I have recently gone through McEvedy and Beard in detail. I am not clear what 'hysteria' is intended to mean. I certainly do not know what the scientific basis of the 'conversion disorder' concept is or why mass hysteria should be considered 'transmitted emotional distress'. However, I think McE and B are correct to suggest that in the few 'severe' cases in the Royal Free outbreak of putative meningoencephalitis with 'neurological signs' recorded in the notes, these signs do not indicate any specific local neurological lesions. They correspond to self-image patterns and their similarity strongly indicates that they are largely due to suggestibility. Apparent neurological signs like this are common enough and I do not know what one should call their cause but they indicate that you will not find any pathology.

The quoted paragraph from Wessely et al. therefore seems very fair. These cases of acute illness, on which the original concept of myalgic encephalomyelitis was built by people like Acheson, have nothing to do with the chronic illness that is now called either ME, CFS or SEID. It is very unfortunate that the term ME, originally intended to cover an acute illness due to some supposed new virus that turned out not to have any local neurological lesions after all, also got used to cover Ramsay's concept of a prolonged illness with PEM.

The only thing that I think is unhelpful in Wessely's account is the implication that ME is not to be used for the illness that affects about 0.2% of the population and which they prefer to call CFS. Things are further confused by Wessely saying that ME is just the unhelpful belief that you have ME and has virtually no overlap with CFS, but then the PACE trial was supposed to study people with CFS and treat their unhelpful beliefs!

So I agree that they make a muddle of things but I think it is right to say that the cases looked at by McE and B were not CFS. They were not cases of a chronic illness.

 

[chrisb]

They correspond to self-image patterns and their similarity strongly indicates that they are largely due to suggestibility

At the moment I don't have the energy to refer to the source but my recollection is that one part of Acheson's four part rebuttal of this suggestion was the apparent similarity of symptoms in cases where neither patient or doctor had knowledge of other outbreaks. The Royal Free outbreak had similarities to the Dalston outbreak which occurred earlier in the year but which was only reported later.

There is a further difficulty. Acheson acknowledged that he was not covering the full spectrum of cases reported and was restricting his description to those showing apparent neurological symptoms. It is possible that this led to false impressions.

 

[Jonathan Edwards]

one part of Acheson's four part rebuttal of this suggestion was the apparent similarity of symptoms in cases where neither patient or doctor had knowledge of other outbreaks. The Royal Free outbreak had similarities to the Dalston outbreak which occurred earlier in the year but which was only reported later.

There is a further difficulty. Acheson acknowledged that he was not covering the full spectrum of cases reported and was restricting his description to those showing apparent neurological symptoms. It is possible that this led to false impressions.

The similarity of pattern does not need an explanation. 'Glove and stocking' patterns and unilateral 'arm and leg' patterns are standard patterns for apparent neurological signs that turn out not to be due to any structural lesions. Every medical trainee is taught about them and familiar with them. They are based on self-image elements like hand, foot, arm, leg. True neurological signs do not follow those patterns because the nerves distribute to skin and muscle in unexpected ways.

I think McE and B may also have been suggesting that limited medical knowledge by nurses might have reinforced the patterns - and these outbreaks seem to recur in hospital staff. Nurses will have heard of 'glove and stocking' neuropathy. What they are in general unaware of is that it is actually very rare for true signs of neuropathy to fit with gloves and stockings. In most cases leg anaesthesia spreads up to a very vague cut off well up towards knee or thigh while hand symptoms are mild without any complete anaesthesia.

I strongly suspect that in the 1950s there was a general fear of polio and other neurological illnesses amongst hospital staff and these patterns would be exactly the sort of patterns staff might think the symptoms would be like.

Acheson limited himself to the neurological picture, yes. That was because he was interested in the idea that there was some new virus a bit like polio that was producing a neurological illness. The other features would just be the usual features of acute viral infection like fever and of no real interest, and certainly of no interest to the chronic condition we call ME. So I am not sure what you mean by a difficulty there?

 

[chrisb]

So I am not sure what you mean by a difficulty there?

I think that what I am suggesting is that within the broader spectrum of illnesses unreported by Acheson, but referred to in the literature, it would be necessary to rule out the possibility that some matched the current view of ME.

 

[Jonathan Edwards]

I think that what I am suggesting is that within the broader spectrum of illnesses unreported by Acheson, but referred to in the literature, it would be necessary to rule out the possibility that some matched the current view of ME.

I don't think they can, really, because he was discussing an acute epidemic illness. As far as I understand a relatively small number of people who were in the Royal Free outbreak of an acute illness went on to have a chronic disabling illness. Ramsay was obviously interested in them and we accept that ME often follows an infection. If you are in the middle of an acute viral illness, then things like PEM don't even come on the radar - you feel dreadful all the time. With EBV it is common to be delirious, and unable to get out of bed or even eat and drink because of the throat symptoms. Maybe everyone with acute glandular fever ticks the boxes for the symptoms of ME as now understood but that does not make them cases of ME.

 

[Guest 102]

Ellen Goudsmit’s 1987 paper on McEvedy and Beard:

http://www.axfordsabode.org.uk/me/articl02.htm

Edit: I know that Behan and his late wife studied some of the original Royal Free patients decades later and found mitochondrial abnormalities

 

[adambeyoncelowe]

However, I think McE and B are correct to suggest that in the few 'severe' cases in the Royal Free outbreak of putative meningoencephalitis with 'neurological signs' recorded in the notes, these signs do not indicate any specific local neurological lesions.

I see you mention unilateral glove and stocking signs, but that doesn't match Ramsay's descriptions either. I'm going to copy a few from his white book (paraphrasing rather than quoting, because I don't have a digital edition to hand).

He mentions quite a lot of recorded neurological symptoms: all the typical symptoms of brain fog, word-finding problems, emotional lability, sleep dysfunction, frequency of micturition, hyperacusis and tinnitus, episodic sweating, OI and vertigo, blurring of near vision, diplopia, unequal pupils, lack or weak reaction of the pupils to light, ptosis, paralysis of the face on one or both sides, nystagmus, myoclonic jerks, motor weakness (more common than sensory disturbance and more likely to occur in the lower muscles), muscle spasms, ataxia, Babinski's sign, cranial nerve palsies, cold extremities and paralysis. ANS problems were most common, but CNS symptoms were present in 74% of those at Royal Free, with similar prevalence at other outbreaks.

The muscle problems could include one or both sides, but always were worse on the side of the body used most often (so right-handed people would have worse weakness in their right arm than their left). Neuropathic pain was noted in limbs and was 'associated with marked muscle tenderness'. Bands of 'exquisite tenderness' were discovered in the trapezii and gastrocnemii by separate doctors in the UK and Australia who had never met.

The quoted paragraph from Wessely et al. therefore seems very fair. These cases of acute illness, on which the original concept of myalgic encephalomyelitis was built by people like Acheson, have nothing to do with the chronic illness that is now called either ME, CFS or SEID. It is very unfortunate that the term ME, originally intended to cover an acute illness due to some supposed new virus that turned out not to have any local neurological lesions after all, also got used to cover Ramsay's concept of a prolonged illness with PEM.

This also doesn't fit Ramsay's description. Importantly, he notes loss of muscle power after exertion that took 3-5 days to return even in the acute cases (i.e., the PEM was there from the first few weeks of illness).

Apparent neurological signs like this are common enough and I do not know what one should call their cause but they indicate that you will not find any pathology.

Are the signs above common, or just the ones mentioned by McEveady and Beard? I suspect they include only the information that fits their thesis, as Wessely and co do.

So I agree that they make a muddle of things but I think it is right to say that the cases looked at by McE and B were not CFS. They were not cases of a chronic illness.

He specifically said it was an illness 'with an alarming tendency for chronicity', but it was recognisable within the first few weeks as the same illness. I.e., it didn't need to be chronic.

All the above is from Myalgic Encephalomyelitis & Postviral Fatigue States: The saga of the Royal Free Disease (Second Edition).

 

[Jonathan Edwards]

I see you mention unilateral glove and stocking signs, but that doesn't match Ramsay's descriptions either.

I don't think I mentioned unilateral glove and stocking. I think I said glove and stocking and unilateral arm and leg. These were from the original case notes according to McE and B.

He mentions quite a lot of recorded neurological symptoms: all the typical symptoms of brain fog, word-finding problems, emotional lability, sleep dysfunction, frequency of micturition, hyperacusis and tinnitus, episodic sweating, OI and vertigo, blurring of near vision, diplopia,

All of those would with any severe acute viral illness in some cases.

unequal pupils, lack or weak reaction of the pupils to light, ptosis, paralysis of the face on one or both sides, nystagmus, myoclonic jerks, motor weakness (more common than sensory disturbance and more likely to occur in the lower muscles), muscle spasms, ataxia, Babinski's sign, cranial nerve palsies, cold extremities and paralysis. ANS problems were most common, but CNS symptoms were present in 74% of those at Royal Free, with similar prevalence at other outbreaks.

The trouble is that this is just a random mixture of just about any neurological sign you can think of. That makes it hard to take very seriously. And I am pretty sure none of these people continued to have neurological signs long term. So one has to separate the acute situation where there may or may not have been reliable neurological signs, from a chronic illness that has no neurological signs.

I think the problem with Ramsay is that he thought of the acute illness and the chronic problem as the same thing but it really wasn't. The most one could say is that it seems that a particular type of viral illness we no longer see involved short term neurological signs and in some cases, like other infections, led on to ME.

The muscle problems could include one or both sides, but always were worse on the side of the body used most often (so right-handed people would have worse weakness in their right arm than their left). Neuropathic pain was noted in limbs and was 'associated with marked muscle tenderness'. Bands of 'exquisite tenderness' were discovered in the trapezii and gastrocnemii by separate doctors in the UK and Australia who had never met.

Again, those features don't make any particular neurological or myological sense. Bands of exquisite tenderness might have been venous thromboses or pressure sites. They don't make any neurological sense.

I think one has to accept that an awful lot of physicians who are not neurologists are not that good at eliciting neurological signs. A neurologist would not produce a list like this because he would know his colleagues would regard it as amateur, sloppy and unsystematic. A proper neurological examination is a bit like a print out of a robotic diagnostic check of a piece of complex machinery. And the neurologist does not leave the bedside without knowing what the anatomy of the nerve lesion is. If he did his assistant would spread the word that the boss was lazy and he would never live it down.

This also doesn't fit Ramsay's description. Importantly, he notes loss of muscle power after exertion that took 3-5 days to return even in the acute cases (i.e., the PEM was there from the first few weeks of illness).

I really don't see how Ramsay could be sure of this. If you have an acute viral illness and you exert yourself it is very likely to be a few days before you feel back to being able to do anything.

Are the signs above common, or just the ones mentioned by McEvedy and Beard? I suspect they include only the information that fits their thesis, as Wessely and co do.

All the 'signs' Ramsay mentions are common enough. The average 'healthy' person might well show one or two. What would be very uncommon would be to have all these signs together, elicited by a neurologist, without there being some reasonable anatomical interpretation. I strongly suspect that this is just the some total of all the various signs Ramsay thought he could pick up in a fairly large group of people.

He specifically said it was an illness 'with an alarming tendency for chronicity', but it was recognisable within the first few weeks as the same illness. I.e., it didn't need to be chronic.

But that does not seem to be the reality. The Dubbo study found a significant minority of people with symptoms at six months - I doubt there were more in the Royal Free situation. And it was not the same illness, because there were no long term neurological signs as far as we can tell.

I realise that we can never be sure what the reality was like. However, I think we can be sure that McEvedy and Beard were justified in suggesting that the neurological features that were sufficiently well documented to analyse did not suggest there was any real nerve deficit. We can also be sure that the acute illness Ramsay thought he was documenting has not been described for a long time now and was not involved in the genesis of ME for the vast majority of people with the illness. I do not see why one should call this particular acute illness ME, rather than calling glandular fever ME. It skews the perception of the illness in a way that distracts from a sensible scientific analysis to my mind.

 

[Jonathan Edwards]

Ellen Goudsmit’s 1987 paper on McEvedy and Beard:

http://www.axfordsabode.org.uk/me/articl02.htm

Edit: I know that Behan and his late wife studied some of the original Royal Free patients decades later and found mitochondrial abnormalities

Thanks. I will have a read. But I note this in the abstract:

Nevertheless, it is certainly possible that a number of the affected nurses at the Royal Free Hospital may not have suffered from encephalomyelitis, but from an anxiety-related disorder.

 

[chrisb]

I worry about this apparent endorsement of hysteria as being a causal factor in any but a tiny minority of the epidemic cases. It might be viewed as controversial.

 

[Jonathan Edwards]

Having looked at Ellen Goudsmit's account it seems that her main point is that the outbreak did not fit well with the standard description of hysteria. I have no problem with that because I am very wary of using the term hysteria in any context - I am very unclear what it is supposed to mean. I tend to agree with her that a number of the nurses may have had an anxiety-related state. She also makes the point that there is evidence for ME, as we now understand it, having demonstrable biological abnormalities. I think the studies she quotes are questionable now but the key point is that there is a confusion between an acute illness shrouded in the mists of time and a chronic one we see now.

 

[Jonathan Edwards]

I worry about this apparent endorsement of hysteria as being a causal factor in any but a tiny minority of the epidemic cases. It might be viewed as controversial.

I don't think I have at any point said I endorse hysteria, have I? I have said that apparent neurological signs of this sort are common and turn out not to be signs of nerve damage. I have never understood what causes them.

And I don't think my view would be controversial. If I remember rightly it was the view expressed by my mother when mentioning the outbreak decades ago - she was a virologist at the central public health laboratory.

 

[chrisb]

I don't think I have at any point said I endorse hysteria, have I?

No, but McEvedy and Beard, whose views in some respects you appear to accept, did. It would help to distinguish carefully the points of difference.

 

[Jonathan Edwards]

No, but McEvedy and Beard, whose views in some respects you appear to accept, did. It would help to distinguish carefully the points of difference.

If you look at my posts I think you will see that I have been trying hard to make that distinction clear! Right from the start I said I had no idea what hysteria meant or what 'conversion' meant.

 

[adambeyoncelowe]

I don't think I mentioned unilateral glove and stocking. I think I said glove and stocking and unilateral arm and leg. These were from the original case notes according to McE and B.

You're right. That's my muddled brain.

All of those would with any severe acute viral illness in some cases.

Okay. That makes sense.

The trouble is that this is just a random mixture of just about any neurological sign you can think of. That makes it hard to take very seriously. And I am pretty sure none of these people continued to have neurological signs long term. So one has to separate the acute situation where there may or may not have been reliable neurological signs, from a chronic illness that has no neurological signs.

Yes, that is the case. In each outbreak he summarises the key neurological features (usually five or so from each site), and he attributes variations to different viral triggers. However, in general, he claimed the neurological damage was 'diffuse', hence not necessarily located in one site. He does report EEGs and electromyograms that gave abnormal results but I'm not sure how many had these results. The ANS symptoms were the most common across the board, as is the case now.

I think the problem with Ramsay is that he thought of the acute illness and the chronic problem as the same thing but it really wasn't. The most one could say is that it seems that a particular type of viral illness we no longer see involved short term neurological signs and in some cases, like other infections, led on to ME.

This may prove correct. He points out that 25% of his patients became chronic, and compares this to EBV, where this doesn't usually happen. Except, of course, Americans are very big on EBV, where he was big on enteroviruses. The likelihood is that both could be involved.

He does, as well, suggest the problem is the host's abnormal immune reaction, rather than a persistent viral infection.

I realise that we can never be sure what the reality was like. However, I think we can be sure that McEvedy and Beard were justified in suggesting that the neurological features that were sufficiently well documented to analyse did not suggest there was any real nerve deficit. We can also be sure that the acute illness Ramsay thought he was documenting has not been described for a long time now and was not involved in the genesis of ME for the vast majority of people with the illness. I do not see why one should call this particular acute illness ME, rather than calling glandular fever ME. It skews the perception of the illness in a way that distracts from a sensible scientific analysis to my mind.

Would the brainstem signalling issues that have been suggested recently perhaps account for any of these symptoms?

 

[Snow Leopard]

I think McE and B may also have been suggesting that limited medical knowledge by nurses might have reinforced the patterns - and these outbreaks seem to recur in hospital staff. Nurses will have heard of 'glove and stocking' neuropathy. What they are in general unaware of is that it is actually very rare for true signs of neuropathy to fit with gloves and stockings. In most cases leg anaesthesia spreads up to a very vague cut off well up towards knee or thigh while hand symptoms are mild without any complete anaesthesia.

I strongly suspect that in the 1950s there was a general fear of polio and other neurological illnesses amongst hospital staff and these patterns would be exactly the sort of patterns staff might think the symptoms would be like.

What you are suggesting is a double edged sword - the presentation and recording of the symptoms are not the symptoms themselves and may reflect those same biases you mention, while the underlying illnesses were genuine.

 

[arewenearlythereyet]

Whilst I pretty much agree with the logic of the arguments regarding the Ramsay outbreak I’m a bit confused about the following @Jonathan Edwards ?

They correspond to self-image patterns and their similarity strongly indicates that they are largely due to suggestibility. Apparent neurological signs like this are common enough and I do not know what one should call their cause but they indicate that you will not find any pathology.

Does this mean it’s suggestive of some form of hypochondria or just that it doesn’t make sense for a viral attack on nerves, or that hand and sock neuropathy is bogus full stop?

 

[Guest 102]

Thanks. I will have a read. But I note this in the abstract:

Nevertheless, it is certainly possible that a number of the affected nurses at the Royal Free Hospital may not have suffered from encephalomyelitis, but from an anxiety-related disorder.

Yes, absolutely, I noted that too - and maybe not all actually had what was then being called ME. I just don't have faith in McEvedy and Beard's analysis - they didn't ever see any patients.

 

[Guest 102]

Having looked at Ellen Goudsmit's account it seems that her main point is that the outbreak did not fit well with the standard description of hysteria. I have no problem with that because I am very wary of using the term hysteria in any context - I am very unclear what it is supposed to mean. I tend to agree with her that a number of the nurses may have had an anxiety-related state. She also makes the point that there is evidence for ME, as we now understand it, having demonstrable biological abnormalities. I think the studies she quotes are questionable now but the key point is that there is a confusion between an acute illness shrouded in the mists of time and a chronic one we see now.

Here is Melvin Ramsay’s own chapter on McEvedy and Beard if it is of any interest. Hope the files are readable! I think the main concern is the influence their opinion had on perception of ME thereafter. Edit: there are seven pages, not sure why one has displayed above my text.