Signs of Intracranial Hypertension, Hypermobility and Craniocervical Obstructions in patients with ME/CFS (Pre-print 2019/published 2020) Bragée et al

As I have probably said earlier I cannot see what cervical disc bulges have to do with brainstem or CSF pressure or hypermobility ... or whatever. It all seems a rag bag of unconnected spinal issues that Peter Rowe has been musing about for twenty years without any evidence.

 

Thanks.

I found an analysis of the Raine Study, around 60% of the girls aged 14 scored positive on the Beighton Score with a cutoff of 4, and 36% with a cutoff of 6. So yes, those scoring positive on a Beighton and thus meeting the threshold for hypermobility (and/or EDS - don’t know enough about difference between hypermobility and hEDS), according to this study, seems to be quite a high number in the normal population.

https://www.sciencedirect.com/science/article/pii/S0022347616310447

Edit: this may be the Seckin study. This also talks about why prevalence rates are different in different studies. They estimate: “ a prevalence of GJH in a university-aged US population as 12.5% using a Beighton score of ≥5 and as 18.2% using a Beighton score of ≥4“. They did not find an association between hypermobility & musculosketal pain, although they did find one for back & neck pain, but pointed out other studies did not find this particular association.

https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6744937/

 

I think the point about all this, is that if there actually is any substance to it, then it needs rigorous science to get to the bottom of it. I include Ron Davis in the rigorous science description, because he is not going to be encouraging / enticing pwME to go off half-cocked towards risky surgery when there is inadequate scientific evidence either way.

There may well be something worthy of further scientific investigation, but those investigations have to be scientifically very sound, especially as any apparently supportive material, be it good science or otherwise, is being propagandised almost to steer pwME towards such surgeries.

It's about lack of evidence at this time, and even worse, misrepresenting information as if it is good scientific evidence when it is not, and using that to mislead desperate pwME.

 

It will be interesting to se the publication. We will sharpen the criterias in next study for certain, and if you have a more reliable score, it would be wonderful to use as well. We do really not wonder but the hypermobility per se, but the possible collagenopathy and its consequences. Nonsens is a strong word for me in this area, but noted as a problem.

 

I just don't... get Jen & Julie here.

JE's position, as far as I have understood, is basically that the bias in selecting patients for the studies in question will be insurmountable the way everything is set up. Which makes sense to me (but that doesn't necessarily mean a lot, I have no idea how this is supposed to go). Therefore, any paper that is published based on those selection criterias will be inherently misleading, much in the same manner the psych papers we tend to get angry about a lot are. Asking to do this properly isn't 'suppressing science', it looks more like 'asking for solid work' to me. Whether or not papers need to be withdrawn or just put into the right context I do not know - most of the psych studies, in my opinion, need to be read correctly more so than be retracted but this here may be a more time-sensitive issue so I really don't know.

On pretty much everything else the argument always seems to be 'we want solid studies done before we start operating on people'. Which, again, doesn't seem to be unreasonable.

I guess there is this meta-discussion on whether someone who had an ME-diagnosis and later resolved the associated problems by taking care of any structural issues was incorrectly diagnosed because ME 'is something different' or whatever, but I don't see this mattering all that much as long as we do not know what 'ME actually is', so to speak. If there are structural problems causing symptoms we should be able to ask for solid science on that and it shouldn't really matter in and of itself how that is called and if other comorbidities are present. I don't understand how any of this could even get to the point where we seem to be right now. Have I missed something elementary?

 

Healthcare systems built on profit-before-patients, and self-publicists?

 

Obviously there’s a context where views vary in the ME community around the area of study in this paper

I’d prefer if posts in this thread were about the science

 

I think the counter argument to this is, which I’ve seen pointed out on various social media when talking about CCI, is people who have severe symptoms - they say they don’t have time to wait, and want to go ahead with surgery, either on their surgeons recommendations or just through their own severity of symptoms. Or they want to know how to help themselves so it doesn’t get more serious.

Just thinking about this, I also think this is maybe a US vs Non-US issue? Because those in the US see the surgeons for free very often, as they have an insurance based system. So he is not an outsider or someone who seems to be providing quack treatments, to them. Whereas to others outside the US, they pay hundreds of thousands of pounds to get surgeries done in the US or in Spain, or have to track down specific surgeons, and it’s seen as a possible quack treatment because the research into traction, what the surgeons are doing, Their measurements, (in controlled studies ) isn’t there. But in America it’s not because it’s enough for them that the surgeons are practising.

 

You'd think they might find it worrying that the surgeons are practising on them.

 

You would think so.
Personally I would like to see these surgeons given 500 MRIs of healthy people and 500 MRIs of those with CCI or even ME type symptoms. And see them calculate measurements (CXA, the other measures they use) for both groups. In one study. Without bias. Without knowing who is in which group. Or even, get outside people to calculate these.

Then I’d like to see these people - both healthy and ME undergo traction, dose dependent like J Edwards mentioned, so they don’t even know what amount of traction is applied, and Id like to see what both healthy people and those with ME say about it.

Just don’t understand why the surgeons don’t have this data. And why people are ok with that. It’s also in my mind probably what the Bragee study should’ve done first - especially the measurements.

 

Looking for some clarification—are you saying you believe that believe hEDS doesn't exist based on your own clinical experience?

 

My belief is primary based on reading the literature on classification and knowing the people who have been involved in creating this diagnosis over the last few decades. I co-authored one of the first papers putatively linking hypermobility to other problems and as part of that I learned that physicians working in this area tend not to understand how reliable evidence works.

My own clinical experience over 30 years is also relevant in that I never met a patient who I thought this diagnosis would be useful for. I worked alongside a clinic where the diagnosis (or an older equivalent) was repeatedly made for reasons that were never clear to me.

To be of any real value EDS has to refer to a demonstrable dominant Mendelian trait (or sex linked recessive). That is not the case for 'hEDS' in general. There are a very small number of people with dominant traits whose EDS probably best falls under 'hypermobility-type' but the great majority of people given the hEDS diagnosis are just rather mobile, and most of them not abnormally so.

 

There are lots of people who don't have insurance in the USA or have poor insurance or their surgeon of choice is not part of their insurance network (this is a very common problem). Our health care system is fragmented and is based on primarily insurance provided by one's employer or the government, if you are disabled.

If your surgeon is out of network, you can petition for coverage. Not always successfully.

 

Your last sentence here sounds like you believed it's hugely overdiagnosed, not that it doesn't exist at all/is never a valuable diagnosis. Is that correct?

 

What probably exists is a very small group of people with a Mendelian trait that predominantly produces hypermobility. 'hEDS' is a concept defined by a committee of people who are not by and large geneticists and is defined by criteria that have no genetic basis. I suspect that geneticists would want to have nothing to do with the term hEDS. They would probably classify a patient as having EDS on the basis of mutation XYZ in gene ABC manifesting, in their case, with certain clinical signs. Since the mutations are likely to be extremely rare and also heterogeneous muddling up with the catch-all term of hEDS would be unhelpful because each individual would have no predictable similarity to any other unless they had exactly the same mutation.

 

From the experience of my own family, I also wonder if the coincidence of two heritable tendencies can present as 'EDS'.

In a generally hypermobile female group, where only a minority have problems with repeated subluxations from normal movements, it's noticeable that the joints involved are only the ball-and-socket and condyloid types. I suspect some of our joint sockets might just be a bit shallow, which in someone who didn't also have laxity would never cause any issues at all.

 

That makes sense to me, Kitty. Potentially that could describe a family member who has an hEDS diagnosis. She experiences subluxations in multiple joints including hips with pain and disability. Bilateral hip dysplasia was identified in her mid forties and surgery followed - periacetabular osteotomy - to one hip which has improved the subluxing on that side. She has only had imaging done on her hips though.

 

Hi there friends, no foes for me. I think a forum like this should not be a harsh semiacademic scene, but a place for information on a mutual respectful way. Our article is not aimed as an advocacy for surgery but trying to sort things right and present important clinical findings. So i get a bit personal here.
I have worked for decades trying to help patients with severe pain, like cancer pain, fibromyalgia, back pain. In 2018 the region (county council) asked us to establish a clinic for ME as the existing one hade waiting list exceeding 3 years. So we did, and within some months all waiting lists in Sweden was gone. That stretched our resources and I met a lot of patients in a short time. I was astonished with the different arena I now entered. Sure I had met patients with ME, patients with CCJ injuries and hypermobiility syndroms. Now and then. But suddenly every second patient had rather severe neurological findings, like segmental (often cervical) hyperestesia, weakness, allodynia (pain at harmless stimuli) and so forth, and as many were hypermobile, easaly putting there flat hands on the floor - something not very usual in a pain clinic. Most surprising was that these findings had not been described earlier, which we soon understood, as we could found out that very few had undergone any physical exam at all, at least not described in medical records.
So we decided after 300 patients that this must be published, maybe other clinicians can confirm, or reject or findings. Most of the patients were willing to participate in this retrospective study.
Under the time we - as always- examined the MRI:s from both brain and cervical spine. And was happy to find both students and radiologist wanting to make more specific descriptions on sign of our focus.

So , sure I cohort is biased, as it consists of patients with severe ME, often with very long history of symptoms. But not biased towards hypermobility or cervical problems. They were refferred by over 150 different physicians, mainly GP:s from all over the county, and 30 percent from other regions. Hypermobility or cervical junction problems were never a reason, but PEM and fatigue.

We are overwhelmed by the response to our article, sometimes overinterpreted, though. 9000 viewers and 3000 downloads show that there is a well motivated and large interest in this field, and we will do our best to take our responsibility, and not claim or advocate for treatments based on the findings, but for hypothesis testing and further research. We soon initiate some studies related to IH (not IIH), and somewhat later more prospective study with control groups. And the we come back and present results. My time and resources do not allow me to give personal answers to all or engage in discussions in different forums. Pardon for that, we will meet again, dont know where, dont know when. All support to your important work, and respect to all sufferers.
Dr Bjorn (known as angry doctor in Sweden after a televison serie where i tried to help ignored patients)

 

Karolinska Institutet: Neck injuries and hypermobility are common among patients with ME/CFS

Continue reading:
https://news.ki.se/neck-injuries-and-hypermobility-are-common-among-patients-with-mecfs