The science of craniocervical instability and other spinal issues and their possible connection with ME/CFS - discussion thread

[lunarainbows]

Because there is no science proving any particular cause of ME/CFS.

The starting point of science is observation. We have that, multiple people who’s clearly defined ‘ME’ (a disease of unknown causation) has markedly improved via surgery. Many of us feel better with our heads pulled on. (And by the way, whether or not it is something that can be fully fixed surgically is actually also a secondary question to the larger question, understanding our pathophysiology).

The end point of a scientific investigation not having arrived yet can’t be used as a justification for not investigating or as a rebuttal for a hypothesis.

We also, I’m sure, have people who haven’t markedly or dramatically improved following surgery, and we also have people who have got worse after having their heads pulled on. There are also people who probably have no reaction to it whatsoever.

No one as far as I can see, is using an end point not being arrived at, to justify not investigating or rebutting a hypothesis. People are rebutting the hypothesis based on what the hypothesis says, what the neurosurgeons are publishing in their materials, the lack of blinded clinical trials, lack of validity and testing of measurements, trying to understand what the “international consensus” means despite not having much (or any?) independent data to back it up or actually being picked up by most surgeons internationally which is why people are going to a handful of selected neurosurgeons.. (has anyone actually replied to @Michiel Tack ‘s question about the consensus criteria?)...etc

 

[ME/CFS Skeptic]

Still nobody has substantively engaged with any of the science posted by Jen and Debored and Hip...... Come on guys. You can't complain about lack of science and then ignore when you are shown the science.

With all the respect Sarah, but it seems that it's you who haven't kept up with what has been said in this thread.

There is basically nothing that links ME/CFS and CCI in the scientific literature. So we tried to look at the closest thing which was the 2019 Henderson study on 20 patients with hereditary hypermobility connective tissue disorders and CCI. At the time this study was cited with enthusiasm as if it supported the spectacular recovery stories in patients with ME/CFS. A closer look showed that it doesn't, as improvements were relatively small and not reported in comparison to any control group whatsoever. The paper suggests that comorbid conditions such as dysautonomia confounded the outcome while in a video Henderson said that "Even at 5 years ... 60 percent of patients had not returned to work." That study isn't cited that often anymore and I haven't seen much debate about why it doesn't more clearly show spectacular improvements as in the anecdotal reports that some are promoting online. All this was pointed out months ago.

Most of the links that have now been shared are about the Clivo-axial angle (CXA) and cervical traction and their ability to indicate CCI or the need for fusion surgery. Some studies help to indicate which CXA values are uncommon but it seems that we don't really know what that means or how it relates to symptoms and disability. Henderson has published two studies trying to argue that CXA is relevant to brainstem deformity but his studies are preliminary (one with 5 and one with 10 patients) and do not provide reliable evidence for its use yet. The consensus statement only speaks of values that are "potentially pathological" and according to Henderson it was not intended to describe surgical indication.

It is sometimes claimed that surgeons have been doing CCI surgery in EDS patients for a long time and that it's normal there etc. But there haven't been any good scientific studies on EDS patients either. Even Henderson acknowledges that it is controversial. In his 2016 review, he writes that "opponents argue ... that the radiological diagnosis of pathological instability at the cranio-cervical junction has not been clearly established in the literature for the hypermobility population." The consensus statement that is often cited was never published in a scientific journal and it seems that we don't even know who the exact authors were. So, again it would be up to the persons who have claimed that these are reliable and objective measurements for CCI diagnosis and treatment to come up with an explanation or some evidence for that. Thus far the pattern seems to be that each time we point out weaknesses or inconsistency, the discussion stops and things are moved on to something else.

Currently, that seems to be invasive cervical traction which some describe as an amazing, objective or reliable test to help determine if fusion surgery is indicated. But there doesn't seem to be a scientific study that tested this procedure and provide us with some figures of specificity or sensitivity. It seems that it's mostly based on the assumptions of surgeons and patients reporting dramatic and instantaneous improvements. That doesn't seem objective or reliable at all. So it would be interesting to hear why some people have presented it as such or whether they can explain why this procedure isn't used by other CCI experts.

 

[Guest 102]

I mean there is zero evidence. But that doesn’t really mean anything at this stage.

And obviously, this surgery is relevant to CCI. The question is how many people who meet ME/CFS criteria have CCI? And if it is a significant number, how they hell did they get CCI?

Another way of framing question. And how many who *meet* ME criteria were actually misdiagnosed and had CCI all along, resolved by surgery.

 

[ME/CFS Skeptic]

I couldn’t help but laugh reading this as I am not sure what surgeon would ever do surgery based on a patient’s DIY radiology analysis!

I thought there was agreement that manual traction, neck collars, and upright scans can cause significant harm as well. So I don't see what's funny about this.

The main argument for CCI surgery seems to be an argument from authority:

Neurosurgeons must know what they are talking about.

I agree.

Basically, if a lot of ME/CFS patients say they have been diagnosed with CCI in a short period of time and mostly by the same surgeons, it's normal to ask whether those diagnoses are reliable or not. CCI and ME/CFS have different clinical presentations and the scientific literature does not suggest a link between them. The two neurosurgeons who have been most involved in making these diagnoses know little about ME/CFS, work in private practice and don't publish much about their findings in the scientific literature. One is controversial and has been accused by former patients for performing unnecessary surgery. Its seems that most of their colleagues do not share their assumptions and methods.

So even though we can't yet know the answer, it doesn't seem unreasonable to consider the possibility that these neurosurgeons are overdiagnosis patients with CCI. If so, that would make the whole enterprise of guiding ME/CFS patients to get tested and treated for CCI with these neurosurgeons highly problematic. Unfortunately, the only response to this possibility has been to either ignore it or say that these neurosurgeons are really great and can be trusted.

It has also been claimed that we are "trying to protect people who are consenting adults engaged in a lawful exchange under the conditions of completely well-informed consent." But if that wasn't the case it would be illegal and possibly criminal. It's not a very persuasive argument to say that raised concerns aren't relevant because everything is legal.

It has also been claimed that patients who pursue this CCI-path really read up on the available information and that they get second opinions etc. But if they have been reading the things that we have debunked here or what Jeffs "indispensible" website promotes they might have been misled rather than informed. I also wonder who the second opinions are given that there are so few neurosurgeons who are willing to go that route. Who's the second or third opinion for people in Europe where everybody seems to go to Barcelona to get diagnosed and treated for CCI?

 

[Sly Saint]

@JenB have you or any other member of your CCI etc FB groups done Ron Davis nanoneedle test?

@Ben H will RD be publishing or releasing information re the nanoneedle testing he was proposing to do on ME/CFS patients going to have CCI surgery?

 

[Hip]

And as far as we know he has no evidence for that claim.

"As far as we know" being the operative phrase. We don't actually know what evidence there is. So we cannot claim that there is none.

We would need to do some further investigation into the subject matter.

How would he have got evidence for the claim?

We don't know, but it may simply be by observing during his long career that fusion surgeries given to patients who satisfied the cervical medullary syndrome symptom criteria and the radiological measurement criteria, but who did not show prior major symptomatic improvements via cervical traction, were the ones who also did not show symptomatic improvements after surgery. Whereas the ones who improved under traction also improved under surgery.

This is a rapidly evolving field. Indeed, even in the short time I have been reading about CCI, clinical practice has changed in the case of some of these doctors. Thus things may have be learnt by doctors that have not yet been tested in formal studies.

You don't actually need to do any modelling fo r ti to be obvious that a high CXA angle would be associated with stretching of brainstem - it is obvious it would.

Didn't you just earlier criticize doctors who intuit what is going to work without actually testing it in formal studies. So isn't it a good thing that this has been examined in a formal study, rather than just going on what is "obvious"?

He is either unaware of, or conveniently forgets, the psychology of this sort of situation.

Not at all, he addresses the placebo effect, and explains how he accounts for it at timecode 59:40 of his 2018 video, which you might like to listen to. He uses repeated traction tests the patient does at home and/or with a physiotherapist to verify that the improvement in symptoms was not just a fluke in the doctor's office, but is a reproducible phenomenon.

Also, if you are claiming placebo, would you like to explain why pushing down on the head (axial loading) makes symptoms dramatically worse? According to your placebo theory, anything the doctor does should improve symptoms, but in fact axial loading makes symptoms worse, whereas cervical traction makes the symptoms better.

You have to tell me what piece of scientific evidence that tests an idea we haven't addressed.I

@Sarah94 did not refer to "scientific evidence" but to "science".

I am sure everybody knows that there are two fundamental pillars of science: empirical evidence gathering and theory/hypothesis creation.

By focusing on scientific evidence only, it's only doing half the job, and creates a lopsided science.

In the area I studied, physics, researchers by nature gather into two distinct groups: the experimentalists (who with the aid of lab equipment gather empirical evidence) and the theoreticians (who with nothing more than pencil and paper and their own minds and imaginations devise theories that can explain the evidence). Most physicists fall into one or the other of the groups, and it is said that the two groups never mix, because of their different interests.

All the analysis I have seen on these CCI threads on S4ME has been solely from the empirical evidence perspective, thus creating a bit of lopsided science.

I think this lopsided analysis is in part the reason why those interested in the possible CCI connection to ME/CFS discuss their interests elsewhere.

 

[butter.]

A person that is involved in ME/CFS research at the NIH agreed that the evidence for craniocervical surgery being relevant to ME/CFS is very weak.

who?

 

[Hip]

Wow. I mean this is bizarre on so many different levels. I’m just imagining a doctor I know saying something like this and I just can’t.

Are you not familiar with what is known as a doctor's sense of humour? Dr B has a great doctor's sense of humour, which has made me laugh on a number of occasions in his videos.

 

[Hip]

As far as I’m aware these patients aren’t being fused in traction positions, and they’re not becoming ill from being fused in the “normal position”, from what I understand? This is what is confusing me so much.

The problem is that no information seems to be available online regarding the procedures and fusion hardware the NHS use in their CCI fusion surgeries. Therefore we have no idea what you would be letting yourself in for if you went to the NHS.

 

[Hoopoe]

who?

I do not want to reveal the name. If they want, in time, they will make a statement on their own. I am hoping that NIH will investigate these claims in their own intramural study.

 

[JenB]

Do you actually mean this? That the fact that there is no evidence is not important? Because I'm having a hard time understanding that stance, on many levels.

That there is no evidence that CCF is relevant to ME. Someone unnamed source at the NIH saying so doesn’t mean anything. It is self-evident. There have been no studies. There is no literature. No one has even thought about this much at all until a few months ago. (The conversation around cervical issues, on the other hand, goes back many decades.)

CCF is a treatment for craniocervical instability, which a number of people who meet ME criteria are turning out to have. They are also turning out to have many other things that are related to connective tissue disorders. The also have connective tissue disorders. This should intrigue us. It has intrigued us—this is why people are starting to study this. Soon we’ll have more evidence.

That there is none yet tell us very little about the relevance of this line of inquiry to our patient population.

 

[JenB]

@JenB have you or any other member of your CCI etc FB groups done Ron Davis nanoneedle test?

@Ben H will RD be publishing or releasing information re the nanoneedle testing he was proposing to do on ME/CFS patients going to have CCI surgery?

I did but something went wrong with my test so I need to redo it. I’ll only have a “post” test, so it doesn’t tell us much. There is at least one person who had done a “pre” test and has since had surgery. She is doing well and is planning to go back to work in January. I have no idea what her results have been, obviously. I don’t know if any other people have or not. Of course they’ll share the data at some point in the future, when they have some.

 

[JenB]

I thought there was agreement that manual traction, neck collars, and upright scans can cause significant harm as well. So I don't see what's funny about this.

I don’t think that was the topic of Hip’s post...

It depends on the context, Michiel. The context. Doctors recommend these things to patients who need them all the time. I’ve seen some people who have craniocervical junction problems have worsened symptoms after an upright scan. I have seen people by in large recover. I see no reason flexing/extending should be harmful to someone with a normal craniocervical junction, but I am not a doctor.

My personal experience of getting worse was not after a single upright scan (which was fine) but after multiple repeated provocative exams and imaging by no less than six different doctors at UCLA, Stanford, Penn State, etc. My cervical collar was prescribed to me by a neurosurgeon at Stanford. Sometimes, doctors need to do these things to find out what is wrong with you or to manage severe symptoms.

 

[Dx Revision Watch]

Free full text:

https://www.thespinejournalonline.com/article/S1529-9430(16)31093-2/fulltext

Variability in diagnostic error rates of 10 MRI centers performing lumbar spine MRI examinations on the same patient within a 3-week period Herzog, Richard et al. The Spine Journal, Volume 17, Issue 4, 554 - 561

Abstract
BACKGROUND CONTEXT:
In today's health-care climate, magnetic resonance imaging (MRI) is often perceived as a commodity-a service where there are no meaningful differences in quality and thus an area in which patients can be advised to select a provider based on price and convenience alone. If this prevailing view is correct, then a patient should expect to receive the same radiological diagnosis regardless of which imaging center he or she visits, or which radiologist reviews the examination. Based on their extensive clinical experience, the authors believe that this assumption is not correct and that it can negatively impact patient care, outcomes, and costs.

(...)

CONCLUSIONS:
This study found marked variability in the reported interpretive findings and a high prevalence of interpretive errors in radiologists' reports of an MRI examination of the lumbar spine performed on the same patient at 10 different MRI centers over a short time period. As a result, the authors conclude that where a patient obtains his or her MRI examination and which radiologist interprets the examination may have a direct impact on radiological diagnosis, subsequent choice of treatment, and clinical outcome.

 

[JenB]

Another way of framing question. And how many who *meet* ME criteria were actually misdiagnosed and had CCI all along, resolved by surgery.

Totally! I think that’s something we need to find out.

 

[JenB]

With all the respect Sarah, but it seems that it's you who haven't kept up with what has been said in this thread.

There is basically nothing that links ME/CFS and CCI in the scientific literature. So we tried to look at the closest thing which was the 2019 Henderson study on 20 patients with hereditary hypermobility connective tissue disorders and CCI. At the time this study was cited with enthusiasm as if it supported the spectacular recovery stories in patients with ME/CFS. A closer look showed that it doesn't, as improvements were relatively small and not reported in comparison to any control group whatsoever. The paper suggests that comorbid conditions such as dysautonomia confounded the outcome while in a video Henderson said that "Even at 5 years ... 60 percent of patients had not returned to work." That study isn't cited that often anymore and I haven't seen much debate about why it doesn't more clearly show spectacular improvements as in the anecdotal reports that some are promoting online. All this was pointed out months ago.

Most of the links that have now been shared are about the Clivo-axial angle (CXA) and cervical traction and their ability to indicate CCI or the need for fusion surgery. Some studies help to indicate which CXA values are uncommon but it seems that we don't really know what that means or how it relates to symptoms and disability. Henderson has published two studies trying to argue that CXA is relevant to brainstem deformity but his studies are preliminary (one with 5 and one with 10 patients) and do not provide reliable evidence for its use yet. The consensus statement only speaks of values that are "potentially pathological" and according to Henderson it was not intended to describe surgical indication.

It is sometimes claimed that surgeons have been doing CCI surgery in EDS patients for a long time and that it's normal there etc. But there haven't been any good scientific studies on EDS patients either. Even Henderson acknowledges that it is controversial. In his 2016 review, he writes that "opponents argue ... that the radiological diagnosis of pathological instability at the cranio-cervical junction has not been clearly established in the literature for the hypermobility population." The consensus statement that is often cited was never published in a scientific journal and it seems that we don't even know who the exact authors were. So, again it would be up to the persons who have claimed that these are reliable and objective measurements for CCI diagnosis and treatment to come up with an explanation or some evidence for that. Thus far the pattern seems to be that each time we point out weaknesses or inconsistency, the discussion stops and things are moved on to something else.

Currently, that seems to be invasive cervical traction which some describe as an amazing, objective or reliable test to help determine if fusion surgery is indicated. But there doesn't seem to be a scientific study that tested this procedure and provide us with some figures of specificity or sensitivity. It seems that it's mostly based on the assumptions of surgeons and patients reporting dramatic and instantaneous improvements. That doesn't seem objective or reliable at all. So it would be interesting to hear why some people have presented it as such or whether they can explain why this procedure isn't used by other CCI experts.

Yeah, but a lot more has been said and shared than this, both here and in my Medium posts. I think this gets back to @Hip ’s earlier point. Is this all only worth discussing when we have a clinical trial or systematic evidence in our patient population? Or can we also discuss the literature on CCI, tethered cord, EDS, MCAS, infection and cervical problems, symptom overlaps, etc, etc that I’ve been sharing? As they relate to ME/CFS literature and other disease models? Can we make our own observations and generate our own hypotheses? Is this a space for the kind of scientific discussion you would see in an academic setting, which is heavily weighted toward hypothesis-generating and thinking about study design? Or is it only a place for discussing research that has already been published in ME/CFS?

If the latter, then I suggest we all hit pause and regroup in a couple of years.

We’ve already spun the same circles so many times, there is not much left to say.

 

[Esther12]

Currently, that seems to be invasive cervical traction which some describe as an amazing, objective or reliable test to help determine if fusion surgery is indicated. It seems that it's mostly based on the assumptions of surgeons and patients reporting dramatic and instantaneous improvements. That doesn't seem objective or reliable at all. So it would be interesting to hear why some people have presented it as such or whether they can explain why this procedure isn't used by other CCI experts.

I've not been able to keep up with all the discussions here, but the claims about the test for determining if fusion surgery is indicated did stand out to me as important (particularly as I'd previously assumed this judgement was being made on the basis of mri scans that could have been assessed under blinded conditions). If we have a test which has been shown to provide good evidence that certain individuals will benefit from this treatment, then that's important. Equally, if this test is being used to recommend people for surgery but there isn't good evidence that it's of any value then I'd have thought everyone would agree that is reason for concern.

How has this testing been assessed, and what is the best evidence we have for its value? Has there been any sort of single-blind assessment of how different groups respond to it (or would that be unethical if the process itself brings some risk)? Do the results for this test correlate well with any of the other ways in which patients are being assessed for surgery? Has there been any assessment of whether different practitioners carrying out this test get the same results on the same individuals? Sorry if all this has been posted somewhere and I missed it.

 

[Adrian]

Because there is no science proving any particular cause of ME/CFS.

The starting point of science is observation. We have that, multiple people who’s clearly defined ‘ME’ (a disease of unknown causation) has markedly improved via surgery. Many of us feel better with our heads pulled on. (And by the way, whether or not it is something that can be fully fixed surgically is actually also a secondary question to the larger question, understanding our pathophysiology).

The end point of a scientific investigation not having arrived yet can’t be used as a justification for not investigating or as a rebuttal for a hypothesis.

Many patients report feeling better with the lightening process but should that be promoted in the same way?

Observations are interesting but then a push to unproven diagnosis and potentially dangerous treatment shouldn't follow next. It should be investigation, gaining understanding and testing theories in controlled environments.

Of course there is an issue around opportunity costs. That is limited money and people to explore different angles so the question becomes one of where should research dollars be put is it for example in this (which seems implausible to many scientists or is it in the Lightening process where the 'observation rate' is higher but also implausible or perhaps they should be spend on omics studies that show some promise and which scientists think offer a the best route.

 

[Saz94]

Many patients report feeling better with the lightening process but should that be promoted in the same way?

Observations are interesting but then a push to unproven diagnosis and potentially dangerous treatment shouldn't follow next. It should be investigation, gaining understanding and testing theories in controlled environments.

Of course there is an issue around opportunity costs. That is limited money and people to explore different angles so the question becomes one of where should research dollars be put is it for example in this (which seems implausible to many scientists or is it in the Lightening process where the 'observation rate' is higher but also implausible or perhaps they should be spend on omics studies that show some promise and which scientists think offer a the best route.

Part of the Lightning Process is literally training people to say and believe that they're better, no matter how they actually feel.

So I'm not sure that that works as an analogy.

 

[Diluted-biscuit]

Part of the Lightning Process is literally training people to say and believe that they're better, no matter how they actually feel.

So I'm not sure that that works as an analogy.

Some people claim homeopathy and dozens of other similar “therapies” have helped them. There’s plenty of choice for analogies.