Open Survey: Assessing Attitudes Toward Case Definitions: Consensus Building Amongst Researchers, Physicians, Patients, and Activists - Lenny Jason

"Many controversies have emerged around the development of case definitions for ME, ME/CFS, and CFS. We have developed this brief questionnaire that assesses key issues involving case definitions. Thank you for the time you have provided us to give us feedback on these important issues for the patient and scientific community."

"What is the purpose of this research?

We are asking you to be in this research study because we are trying to learn more about perspectives on myalgic encephalomyelitis/chronic fatigue syndrome (ME, ME/CFS, and CFS) case definitions, cardinal symptoms, as well as other issues relevant to ME and ME/CFS research and policy. This study is being conducted by the Center for Community Research at DePaul University under the supervision of professor Leonard A. Jason. We are a research team at DePaul University that has been studying ME, ME/CFS, and CFS for decades."

https://redcap.is.depaul.edu/surveys/?s=4CCENA4KHL

 

Questions I had issues with.

"Do you believe that ME is a separate illness from either CFS or ME/CFS?" Answer Yes or No

"Do you believe that ME, ME/CFS, and CFS are three separate illnesses?" Answer Yes or No

Confusing. Asking about three separate things in each question and then only offering two options. What if someone thinks, for example, that ME and CFS are the same? If they do then they can't express that opinion. Should have offered all the options as possible answers, i.e. all three are the same illness, X and Y are same, X and Z are same, Y and Z are same, none are the same.


"Do you feel that patients should be differentiated into those who meet a case definition requiring post-exertional malaise versus case definitions where post-exertional malaise is either not mentioned or included as an optional/minor criterion? This issue was discussed in a recent study by Wormgoor and Rodenburg 2021." Answer Yes or No
Thread discussing the study linked to, The evidence base for physiotherapy in myalgic encephalomyelitis/chronic fatigue syndrome when considering PEM: a systematic review (2020) Wormgoor

"Do you think that to meet a research case definition there should be some exclusionary conditions?" Answer Yes or No

And I think there should have been an "I don't know" option for the two questions above.


And finally, I'm not even sure what is meant here. Is it that they are asking whether a case definition, for example, should ONLY include long duration and exclude short duration, or are they asking whether a case definition should be split into long duration and short duration subtypes? The use of the word include suggests the former to me.

"Do you think the case definition should include certain subtypes? (Select all that apply)

Duration of illness (long duration vs short duration)
Onset type (eg., infectious onset, Epstein Barr Virus onset, stress onset, sudden onset, gradual onset, etc.)
Trigger type (eg., Mold, Lyme disease, etc.)
Comorbidities (eg., comorbid Long COVID, fibromyalgia, multiple chemical sensitivities, etc.)
Severity of illness (eg., mild, moderate, severe (housebound) and very severe (bedbound))
Course of illness (eg., symptoms are persistent, recurrent, fluctuating, constantly getting worse, constantly improving, etc.)"

 

Here are the comments I put at the end, for what they are worth.
If you haven't submitted yours yet, it may be better not to read them if you don't want to be influenced.


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I have said there should be exclusions for a research definition but would like to highlight that I wouldn’t necessarily want the same exclusions for a clinical definition or clinical practice. Similarly for what it is worth, I would be willing to accept a clinical set of criteria that was looser.

It doesn’t fit in with the options but I would like to like to see composite scores used for symptom criteria so it is not all or nothing with regard to symptoms. A lot of symptoms are not ubiquitous in patients but are common and it increases the confidence that a patient has the condition if they have the symptom. Also scoring for each symptom would be based on the frequency and severity of the symptom. So you might enquire about maybe 10-30 symptoms. And the possible scores might be 0 to a number in the hundreds. Then a threshold could be arrived at for a definition. But individual research studies could also look at the data by stratifying or subgrouping by different symptom scores. Also each symptom wouldn’t necessarily need the same weighting i.e. a particular symptom that was considered as better at distinguishing the illness from other conditions might get a weighting of 2/3/4/whatever.

It is possible with such a system that I would be happier with fewer required symptoms. I think for various reasons people may focus on some symptoms but not focus on or report others but still have them. For example, early on in my illness when I was undiagnosed, I really missed exercising and was really focused on trying to exercise more so was very focused on symptoms that stopped me doing that like what seemed like muscle sprains. I ignored some other symptoms.

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I have picked up from Prof Jason that he isn't into symptom counting but that doesn't necessarily mean he is correct.

 

I lose track of all the names. Is the author a BPS adherent or not?

 

Thank you, @NelliePledge

 

Is the case definition an actual problem? As in, does finding consensus, impossible in the current context, get us any further? Does it lead to any improvement for patients? Make better research? Doubtful, since anyone can make the choice to any case definition they want, apparently. And it conflicts with both training and culture that it's all mood disorders, or whatever. 99% of physicians clearly don't care, they need this to be formal, they need a test, a test that is recognized, politically and professionally acceptable. They need validation from their peers that what they are doing is worth doing, and chronic illness is explicitly excluded from that.

What does get medicine to make a move? A biomedical breakthrough. I'm sure the same issues were common with things like ulcers and pre-breakthrough autoimmune diseases, endless debate about how to make a case definition. It shouldn't be this way, but this is the reality we have to deal with. Debating is for entertainment. We could keep debating this for decades and it wouldn't get anywhere.

I really think the idea of finding consensus is impossible. Not without a complete paradigm shift. Where we are is basically like the last episode of Game of Thrones, where (roughly) the idea that patients should have any say about their health is subject to raucous laughter. Small people are meant to be rules. By rulers. There is no consensus to be had here any more than it's possible for NASA to find consensus with astrologers. We are simply not working on the same set of problems, and none of our detractors are interested in what's real, they like the imaginary models too much for this.

 

I forgot to mention the Bansal criteria in my submission.
He uses symptom scoring.
He proposes a threshold of 8+ out of 13 for clinical criteria and 10 out of 13 for research criteria.
I think having stricter criteria for research/looser criteria for the clinical scenario is justified.