Unintended Consequences of not Specifying Exclusionary Illnesses for SEID (Jason 2015)

[Bob]

I thought it was his son? Or maybe that was someone else.

I can't rely on my memory, so please don't rely on me, but I think Dr Jason had ME/CFS himself.
Ronald Davis of the End ME/CFS Project has a son with severe ME, and Dr Chia's son has/had ME.

 

[Sasha]

I can't rely on my memory, so please don't rely on me, but I think Dr Jason had ME/CFS himself.
Ronald Davis of the End ME/CFS Project has a son with severe ME, and Dr Chia's son has/had ME.

Yes, all that's correct.

 

[Valentijn]

We need to rename the term PEM, it's not any better than saying we have CFS. Yes, the delayed response is the big one.

Something with "Delayed" instead of "Post" would be quite an improvement.

Delayed Exertional Exacerbation (of Symptoms)?
Delayed Exertional Symptom Exacerbation?

 

[lycaena]

Something with "Delayed" instead of "Post" would be quite an improvement.

Delayed Exertional Exacerbation (of Symptoms)?
Delayed Exertional Symptom Exacerbation?

I like it, seems more accurate then "post" something ... but then
I just saw this poster on Facebook, it's about Lupus. "over doing it yesterday means ... today is canceled" sounds like a delayed response, hm
https://www.facebook.com/tom.kindlonpwme/posts/860349300726264

 

[jimells]

The big problem with describing symptoms for doctors is that it has to be concise. The ICC excerpt above is huge. That's nice for someone seeking deeper expertise, but is the average GP going to read all of that carefully enough to understand and accurately apply it?

Yet another reason why actual knowledgeable specialists should be diagnosing this illness instead of harried GPs who don't have the time to learn the details.

In my opinion, the aspects which need to be emphasized are:

1. a trivial exertional trigger
2. a delayed response
3. appearance or exacerbation of neurological, immune, and/or autonomic symptoms

Yes, these do seem to be the defining characteristics.

I have a problem with the entire approach of diagnosis by checklist. Not being a healthcare professional I'm only guessing, but I should think that the patient's overall story would be important to determining an accurate diagnosis.

It looks to me like we are fast approaching a time when more and more diagnoses will be made by software instead of expensive healthcare workers. Anybody with a masters degree and a checklist is now qualified to diagnose depression, for example. No need to rule out well-known illnesses with similar symptoms. Just mark a few boxes on a form and schedule the patient for some magic CBT...

 

[Valentijn]

Yet another reason why actual knowledgeable specialists should be diagnosing this illness instead of harried GPs who don't have the time to learn the details.

Yes, specialists would definitely be better. But we just don't have enough of them yet, especially as part of the mainstream medical service.

I suppose it would be nice to get a simple definition for GPs which is good enough to identify likely ME/SEID patients and refer them to someone with more knowledge about the disease for confirmation of diagnosis. Though that still requires a lot of specialists which don't exist currently.

 

[Mij]

I like it, seems more accurate then "post" something ... but then
I just saw this poster on Facebook, it's about Lupus. "over doing it yesterday means ... today is canceled" sounds like a delayed response, hm
https://www.facebook.com/tom.kindlonpwme/posts/860349300726264

I would be interested to know what others symptoms besides fatigue they are experiencing the next day.

This is from lupus.org
As with most chronic illnesses, leading a healthy lifestyle will help lessen the impact of your symptoms. We know that even small lifestyle changes can help with many symptoms of lupus including fatigue. Lifestyle factors that influence fatigue and energy levels include exercising regularly, avoiding tobacco and drug use, limiting or avoiding alcohol, eating healthy (limiting fast food and high sugar foods, eating more lean meats, fruits and vegetables for example). Several studies in which patients with lupus with low levels of physical activity participated in supervised exercise programs have shown that exercise tolerance, fatigue, and quality of life improve slowly over time. Injury from the exercises is rare despite it being a common fear of patients prior to starting a program.

 

[SOC]

In my opinion, the aspects which need to be emphasized are:

1. a trivial exertional trigger
2. a delayed response
3. appearance or exacerbation of neurological, immune, and/or autonomic symptoms

That looks good to me, although we may need to underline and put in bold and red type the word 'trivial' to get GPs to pay attention to the importance of this. 'Delayed' would probably need to be defined as well, or we'll get GP's thinking an hour later is delayed.

Yes, specialists would definitely be better. But we just don't have enough of them yet, especially as part of the mainstream medical service.

I suppose it would be nice to get a simple definition for GPs which is good enough to identify likely ME/SEID patients and refer them to someone with more knowledge about the disease for confirmation of diagnosis. Though that still requires a lot of specialists which don't exist currently.

I agree this would be best. It's also what happens with some other serious illnesses. The local GP uses a simple diagnostic to determine suspected MS, ME, whatever. S/he refers the patient to a specialist for a full work-up. The specialist is the one who actually diagnoses the serious illness. A treatment plan is put in place. The local GP does routine follow-up, but the specialist supervises, does periodic checks, and deals with complications that are beyond the GP.

We are probably relatively close (5-10 years) to having a minimally adequate number of specialists in the US. Not anywhere near enough to have one in every city, but possibly enough to manage the majority of PWME with the help of cooperative GPs. I'm thinking about once a year visits with more routine stuff being handled by the local GP. It's very from ideal, but better than what we have now. And better than leaving PWME completely in the hands of local GPs.

 

[SOC]

I like it, seems more accurate then "post" something ... but then
I just saw this poster on Facebook, it's about Lupus. "over doing it yesterday means ... today is canceled" sounds like a delayed response, hm
https://www.facebook.com/tom.kindlonpwme/posts/860349300726264

That could easily be exercise intolerance, possibly related to OI. The consequences of overdoing could last for 24+ hours in all kinds of conditions . The thing with PEM is the the response is delayed, not just that the response lasts more than 24 hours. You feel okay when doing the thing -- it's one or two or three days later that the PEM hits.

That isn't to say the every PWME feels fine immediately after exertion, many don't. But that is most likely exercise intolerance, which many of us have in addition to PEM. If you have both, it's likely you first get exercise intolerance symptoms, followed by exacerbation of neurological and immune symptoms (PEM) a day or more later.

 

[Sasha]

I agree we need a better acronym. Nobody knows what 'malaise' even means. My GP thought it meant 'nauseous'.

Delayed Post-Exertional Symptom Exacerbation?

 

[ljason]

Dear blog responders to the latest article from our research group.

First let me thank you all for your excellent suggestions, even if some might have been painful to read. Given the abuses that have occurred in this field for so long, having an active and caring community of patient activists is critical, and I and my research team welcome your comments and critical evaluations of our work.

With that having been said, I wanted to respond to a number of issues that have been posted over the past few days. I usually do not respond to individual posts, but given the number and nature of the posts, I would like to respond to a few issues.

First, the PEM study that we had recently solicited patients to respond to was an effort to use all the phrases that describe PEM that we could find, including from Ramsay to the present. We did not like many of them, so we agree that many items did not well tap this set of symptoms, which I continue to believe is one of the fundamental symptoms of ME. What we wanted was to get patient reactions, and we now have about 500 people who have let us know, in their own words, what PEM is, as well as what phrases best capture it. We are now analyzing these data, and clearly, many of the phrases that are in the literature are not very good, but now we also have patient reactions to support our feelings. So, thank those of you who were willing and able to fill out this questionnaire.

Second, regarding SEID, our latest article used 4 data sets from the past, one going back to 1994. Certainly, the ways we tapped the symptoms were varied, and some were better efforts than others. What we wanted to demonstrate is that there is a possibility, that with few exclusions for SEID, many people with other illnesses might be brought into the SEID category and this might have unwitting consequences. As some of you know, I do believe that many people with a primary affective disorder can be misclassified as having CFS or ME. This is of importance because one of these disorder, Major Depressive Disorder, is one of the most prevalent psychiatric conditions. If we bring such individuals into our SEID categories, I believe it will be more difficult to identify biological markers and understand who is included in treatment trials and who is helped. In an article I published with a graduate student many years ago, we found that with the right questions, it is possible to differentiate those with a primary affective condition from CFS with 100% accuracy. So, it can be done, and needs to be done in our studies that are ongoing. However, if new case definitions (like SEID) do allow these individuals with primary affective disorders to be included, then I do think that there are unintended consequences that might be detrimental for our field.

For those that want the source of this article mentioned above, it is:

Hawk, C., Jason, L.A., & Torres-Harding, S. (2006). Differential diagnosis of chronic fatigue syndrome and major depressive disorder. International Journal of Behavioral Medicine, 13, 244-251.PMID: 17078775

Finally, we do not have good answers to many of the questions that have been raised, but we will continue to work for better answers to the important issues that the patients bring up to us, and they continue to guide our work. We have listened to patients who have asked us to collect data and answer basic questions regarding this illness regarding issues of epidemiology, the name of the illness, non-pharmacologic interventions, and case definitions. For over 25 year we have tried to correct some of the false beliefs about this illness, first with prevalence data where we challenged the “Yuppie Flu” depiction of patients, then by working to show detrimental effects of the term CFS, and later by providing data to support pacing as the most effective way to help patients cope with this devastating illness. We have for over two decades worked on case definition issues, and we will continue to criticize efforts of others when we feel that mistakes have been made, such as when we challenged the empiric criteria of Reeves a number of years ago, which as his criteria would have led to a vast expansion of those who were considered to have this disease.

Again, thank you for taking the time to discuss the work of our team, and we continue to learn from your insights, your guidance, and your honest reactions to our work.


Lenny Jason

 

[Esther12]

Welcome to the forum @ljason - good to have another researcher commenting here and seeing what criticisms being made of their work might be valid.

 

[Gijs]

It is very sad to see so much discussion about subjective criteria after almost 30 years of debate..... It is time for more objective criteria. Use the 2 day CPET exercise test and make 2 groups.

 

[jimells]

Again, thank you for taking the time to discuss the work of our team, and we continue to learn from your insights, your guidance, and your honest reactions to our work.

You are very welcome. Thank you for engaging with us in spite of those comments that are hard to read. It definitely helps my morale when I see researchers attempting to find answers as opposed to trying to bury the illness.

 

[Bob]

Dear Lenny Jason,
A huge welcome to the forum. Thank you for joining us, and for reading our comments, and sharing your thoughts.
As you probably know, the IOM report and the SEID criteria are a highly-charged and sensitive issue, for reasons I'm sure you understand.

I think I can safely speak for many of us when I say that we read your research with great interest.
It would be great if you could pop by the forum on occasion to answer some questions about your research.
I have questions that I'd love to ask you about the DePaul symptom questionnaire, and other aspects of your work.

Thanks again for joining the forum.

 

[Sasha]

First let me thank you all for your excellent suggestions, even if some might have been painful to read. Given the abuses that have occurred in this field for so long, having an active and caring community of patient activists is critical, and I and my research team welcome your comments and critical evaluations of our work.

Hi Dr Jason, and welcome to the forums. It's great that you've come here and that you're willing in such a gracious way to consider and address the issues that have been raised. As patients, we're used to researchers often talking about us, not to us, and it's very refreshing to see a researcher being prepared to engage with us.

I'm very grateful that although you yourself have been very ill with this disease, you haven't abandoned your fellow patients now that you're well enough to work and have stayed with us, trying to help us all.

I haven't read or commented on your paper (can't concentrate well enough at the moment) but I think we're all familiar with the issue that PEM has been poorly defined and that people wave the term around without really knowing what it means.

It's clearly crucially important that if PEM as experienced by PWME is unique to ME, that we need a clear definition of it. This is true whatever set of clinical criteria - CCC, ICC, IOM or some yet-to-be-determined empirical criteria - are going to be used to diagnose us.

It seems (please correct me if I'm wrong) that the delayed aspect of PEM wasn't used in your questionnaires for this study, nor was 'malaise' defined as being broader than just fatigue. I agree with others who have posted that 'a worsening of symptoms including neurological, immune and autonomic symptoms following minimal exertion and delayed by up to 48 hours' is something like the kind of definition (or operationalization) of PEM that would be likely to distinguish ME from other conditions.

Perhaps we, as a group effort, could attempt to design a questionnaire that defines PEM, and/or a questionnaire that attempts to distinguish ME/CFS symptoms from the symptoms of affective disorders? But perhaps it wouldn't be possible, as it could be quite complex and we'd probably end up disagreeing endlessly, and waring. And it's beyond my capacity to lead any project at the moment. But it could be an increasing idea to explore if we were to do it in a small-ish group, and we agreed some ground rules. Then we could ask Lenny Jason if he would be willing to test it and validate it for us. Seeing as we have the best knowledge, we'd be the best people to do this, if we could agree a way to work together. Well, it's just a thought, and perhaps not such a practical idea.

Dr Jason, as Bob suggests, we have expertise in our own disease. I wonder if we can be of use to you? Many here are scientists like yourself and I'm sure would be happy to be a sounding board for you or contribute to your research; and I'm sure many more would be willing to act as pilot participants (who I know are often very hard to find) to pre-test questionnaires. It's possible for certain projects to set up a private subforum on Phoenix Rising visible only to invited participants, and some of us have already used such subforums to work together very productively.

It's frustrating, as patients, to be the subjects of studies rather than active participants in their design and interpretation, especially because the results will directly affect our lives. I hope you might consider novel ways of working with us: I think that would be a rather trailblazing thing to do in this particular area of research.

Thank you again for coming to the forum.

 

[Sushi]

@ljason Welcome to the forum--we are delighted to see you here!

Perhaps we, as a group effort, could attempt to design a questionnaire that defines PEM, and/or a questionnaire that attempts to distinguish ME/CFS symptoms from the symptoms of affective disorders?

I'd certainly like to hear your thought on this too.

Hi Dr Jason, and welcome to the forums. It's great that you've come here and that you're willing in such a gracious way to consider and address the issues that have been raised. As patients, we're used to researchers often talking about us, not to us, and it's very refreshing to see a researcher being prepared to engage with us.

Yes, your presence here is much appreciated, even though you may have joined because "we were talking about you!" I hope you understand that some of the more difficult comments came from our frustrations and do not reflect any lack of respect for the wonderful work that you do.

Dr Jason, as Bob suggests, we have expertise in our own disease. I wonder if we can be of use to you? Many here are scientists like yourself and I'm sure would be happy to be a sounding board for you or contribute to your research; and I'm sure many more would be willing to act as pilot participants (who I know are often very hard to find) to pre-test questionnaires.

Pre-testing questionnaires sounds like a very good idea. I know I struggled with (and got frustrated) trying to fill out the recent PEM questionnaire as the choices just did not fit my experience. I used the blank spaces for additional comments whenever they were available. I would have welcomed the opportunity to be in a pre-test group.

It's frustrating, as patients, to be the subjects of studies rather than active participants in their design and interpretation, especially because the results will directly affect our lives. I hope you might consider novel ways of working with us: I think that would be a rather trailblazing thing to do in this particular area of research.

The idea of patient-researcher collaboration is extremely encouraging for patients. We need "novel" now as so many years have passed, many of us have been sick for a long time and "traditional" has not worked so well for us.

Thanks again for taking up the challenge and engaging with us here.

 

[Sean]

Thank you, Prof Jason. Plus what the others said.

-------------------------------------------

We are not at a point in all this where we have clear definitions or answers, nor are likely to be any time soon.

But we now have a lot more good researchers, particularly biomedical ones, starting to give it much more serious attention and look at it from different angles, and that is what will lead us to better definitions and answers.

Prof Jason's work on SEID is exactly the sort of thing that needs doing. His latest work is not a final answer, just part of the process for helping to clarify where the problems are and which direction(s) to head in.

The important thing here, I think, is that PEM has been pushed to the centre of the game, where it should be. It's exact nature and role still need to be understood, and that will take time. But at least it is now in the spotlight, and that is a huge win.

(That is one big reason I am okay with the IOM report, it puts PEM at the centre.)

 

[Kati]

Biomarkers. We need biomarkers. And we need them recognized by FDA and governments.

In the meantime, thank you very much, Prof Jason and team for your hard work. Your presence and wisdom at CFSAC is sorely missed. i also noticed how your were rudely cut off at the P2P meeting.

 

[ljason]

Dear Fellow bloggers:
Thank you for all your reactions to my post. I am actually out of town this past week, and am returning to Chicago today. I continue to be most appreciative of all this feedback. My team now has responses from about 500 patients, and we are trying to begin to analyze this data set where we are getting the opinions on PEM. I hope to summarize this information and get it back to the larger patient community. I appreciate all of your willingness to continue to provide us your thoughts.
For those of you who might like to see one of our articles on PEM where we provide some of our thoughts, it is attached.
Once I get back to Chicago, will have more time to go over all your responses in more detail.
Lenny Jason