Do people with ME/CFS and joint hypermobility represent a disease subgroup? An analysis using registry data, 2024, Kathleen Mudie et al

It looks as if this has come from staff at Solve ME. If so it is very disappointing to see such a muddle from a support/funding group.

It isn't very clear from the slides but it looks as if the hypermobile people are only different in that they self-report diagnoses associated with hypermobility! And another study showed nothing.
 
I'm assuming that by "significant differences" they mean those that were statistically significant. These were:

Self-reported EDS
Family history of EDS
Self-reported POTS
Loss of balance, unsteadiness on feet when standing or inability to focus vision

There are some other differences listed, presumably the ones not reaching statistically significant differences.
 
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Looking at the slides, there are a lot of assumptions being made about people with joint hypermobility also having problems throughout their bodies with connective tissue, and hypotheses about this linking with POTS and cranial hypertension. I thought that only applied to people with the rare genetic severe form of EDS. ???

There is a slide about this study we have a thread on here:
The Presentation of ME/CFS Is Not Influenced by the Presence or Absence of Joint Hypermobility, 2021, Vogel, Rowe et al
The slide says:
Do JH+ ME/CFS represent a subgroup?
Hypothesis: JH+ ME/CFS = earlier onset of symptoms, increased severity, more co-existing conditions, lower HRQoL !
Found: joint hypermobility was not associated with clinical characteristics of the illness “This paradox requires further exploration”.
Click to expand...

Then the slide about the self reported data from the Solve study:
• 687 ME/CFS (CDC-94/CCC)
• aged >18 years
Do JH+ ME/CFS represent a subgroup?
Hypothesis: JH+ ME/CFS and JH- ME/CFS = sub-groups
-> could lead to targeted treatment for a beneficial impact on HRQoL
JH+ = Beighton score >= 5 if under 50 & >= 4 if over 50 (12%)
Click to expand...
It's not clear whether the 12% refers to the population prevalence comparitor they are using, or to the result from their data.
The next slides indicate self reports of Joint hypermobility and EDS are raised. And they go on to link these with POTS and OI symptoms.

Unfortunately I think none of our team managed to watch the presentation as it was at the end of a long day. Once we get the videos we'll be able to report more fully on what was actually said about the data.

I've done some searching on Twitter to see whether anyone had reported on this talk. This was the only tweet I found:

The tweet says:
Results are still in early stages. Have not yet seen statistically significant subgroup based on JH+.

I looked at the abstract provided to participants for this talk. It says nothing about hypermobility, it's just a general abstract about the future research use of the patient data from the registry.
 
 
Trish said:
It's not clear whether the 12% refers to the population prevalence comparitor they are using, or to the result from their data.
Click to expand...
I've now watched the presentation. The 12% is the proportion of the ME/CFS patients in the Registry who may have hypermobility based on Beignton scores. She did point out this is only an indicator of hypermobility, not a clinical test.
Given that is towards the lower end of estimates she quoted from population studies for the general populatlon, I don't see how joint hypermobility can be a predisposing factor for ME/CFS, therefore a subgroup, though I can see that pwME who happen to have hypermobile joints may have some joint pain and attribute it to both conditions.
My notes on the talk are here:
https://www.s4me.info/threads/iacfs...uly-9-am-to-5-pm-edt.28754/page-2#post-432961
 
glennchan said:
Genome-Wide Association Study data on ME/CFS suggests that hypermobility isn't caused by genetics. https://www.medrxiv.org/content/10.1101/2022.09.09.22279773v2 That points towards some environmental factor that led to hypermobility before the onset of ME/CFS.
Click to expand...

Das et al is discussed here: https://www.s4me.info/threads/genet...al-analysis-2022-das-et-al.29445/#post-436520 and here https://www.s4me.info/threads/blog-new-study-links-14-genes-to-me-cfs.29511/#post-437195
 
glennchan said:
Genome-Wide Association Study data on ME/CFS suggests that hypermobility isn't caused by genetics. https://www.medrxiv.org/content/10.1101/2022.09.09.22279773v2

That points towards some environmental factor that led to hypermobility before the onset of ME/CFS.
Click to expand...
This looks like nonsense. The SolveME registry data shows no increase in hypermobility over population rates, as did the UK ME/CFS biobank data. So your 'points to' conclusion doesn't follow. In fact the data says the opposite of your hypothesis.
As for your 'data' on vaccine injury and hypermobility - you have said yourself it's not scientifically valid, so why publicise it?
 
I'm pretty sure there is something abnormal going on with the joints of a subgroup of patients but it's not showing up in controlled studies designed to detect an assocation between hypermobility and ME/CFS. That could mean it's a very small subgroup or that there is no association with ME/CFS or that joint hypermobility is too unspecific a criterium to detect it.

I say pretty sure because the descriptions sound abnormal. I never quite understood what precisely the patients are describing.

It's also possible that hypermobility as defined in these studies is a largely normal trait in the population that has had a false theory built around it. Attributing the problems with regulating blood pressure to connective tissue laxity seems like an intuitive explanation. Perhaps the people who really have more a POTS phenotype and also have some benign hypermobility just got it wrong.
 
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Jonathan Edwards said:
On what basis do you say that? Was there any analysis of hypermobility in the study?

Edit: the word hypermobility does not appear in the paper.
Click to expand...

If there is a higher incidence of hypermobility and there is a genetic basis for that hypermobility, then it would show up in the GWAS study.
 
Merged thread

Do people with ME/CFS and joint hypermobility represent a disease subgroup? An analysis using registry data, 2024, Kathleen Mudie et al

Background: Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) is a chronic, multifaceted disease that affects millions globally. Despite its significant impact, the disease's etiology remains poorly understood, and symptom heterogeneity poses challenges for diagnosis and treatment. Joint hypermobility, commonly seen in hypermobile Ehlers-Danlos Syndrome (hEDS), has been observed in ME/CFS patients but its prevalence and clinical significance within this population are not well-characterized.

Objective: To compare the characteristics of ME/CFS patients with and without joint hypermobility (JH+ and JH-) as assessed using the Beighton scoring system, and to explore whether JH+ ME/CFS patients exhibit distinct disease characteristics, comorbidities, and health-related quality of life (HRQOL).

Methods: The study used cross-sectional, self-reported data from 815 participants of the You + ME Registry. Participants were categorized as JH+ or JH- based on self–assessed Beighton scores and compared across demographics, comorbidities, family history, and symptoms. HRQOL was assessed using the Short Form-36 RAND survey and Karnofsky Performance Status.

Results: 15.5% (N = 126) of participants were classified as JH+. JH+ participants were more likely to be female, report Ehlers-Danlos Syndrome (EDS), Postural Orthostatic Tachycardia Syndrome (POTS), and a family history of EDS. They experienced worse HRQOL, particularly in physical functioning and pain, and a higher number of autonomic, neurocognitive, headache, gut, and musculoskeletal symptoms. Sensitivity analysis suggested that ME/CFS with concurrent JH+ and EDS was associated with more severe symptoms and greater functional impairment.

Conclusion: ME/CFS patients with joint hypermobility, particularly those with EDS, demonstrate distinct clinical characteristics, including more severe symptomatology and reduced HRQOL. These findings highlight the need for comprehensive clinical assessments of ME/CFS patients with joint hypermobility. Understanding these relationships could aid in subgroup identification, improving diagnosis, and informing targeted therapeutic approaches. Further research is warranted to explore these associations and their implications for clinical practice.

https://www.frontiersin.org/journals/neurology/articles/10.3389/fneur.2024.1324879/full
 
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ME Research UK

·
The heterogeneity of ME/CFS, as evidenced by diverse symptom profiles
potentially signifying variations in disease mechanisms, presents a
challenge in the development of a diagnostic test and treatments. A
recent cross-sectional study (https://bit.ly/3xA7S4B), involving over
800 ME/CFS participants, explored this heterogeneity in relation to
the presence or absence of joint hypermobility. The findings revealed
ME/CFS participants with concurrent joint hypermobility, particularly
those with Ehler’s Danlos syndrome (EDS), “demonstrate distinct
clinical characteristics, including more severe symptomatology and
reduced [health-related quality of life] HRQOL.”

Read more: https://tinyurl.com/msr5m8fb

https://twitter.com/user/status/1777622485678145899
Click to expand...
 
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