Occupy CFS

A study published last month* by a group from Norway reports a systematic review of ME/CFS case definitions. It’s important because the Institute of Medicine panel is likely conducting a similar process as part of its work, but the study is also significant for the startling and overreaching conclusions drawn by the authors.

Quick Summary:

• This study collects and examines case definitions for ME/CFS, and extracts prevalence data. This will be a helpful future reference.
• The authors then step far beyond their own evidence and draw a variety of overreaching conclusions.

• Future analysis must be wary of cognitive bias in order to avoid the same mistakes.

What They Did

Bruberg et al began by searching the literature for case definitions of ME/CFS (a term encompassing CFS, ME, and combinations of the two). The group then looked for validation studies, meaning studies that applied one or more case definitions to patient populations. Of the twenty case definitions identified in the first step, seven have never been validated in a patient population. Thirty-eight validation studies were identified that test one or more case definition, but the authors were very critical of the quality of those studies saying, “We did not identify any study which rigorously assessed the reproducibility or feasibility of the different case definitions.”

The authors categorized the validation studies, and the summaries and Tables 1-4 are actually quite helpful. Five studies independently applied several case definitions to a single patient population, and thirteen studies took patients diagnosed under one definition (usually Fukuda) and sequentially applied case definitions assumed to be more specific. The authors caution that this second group of studies “should be interpreted with great caution” because there were so many differences in diagnostic method and definition application, including different thresholds for exclusion of psychiatric comorbidity. Finally, twenty-one studies presented prevalence estimates and the authors extracted data on prevalence rates for Fukuda, Oxford, and the Australia 1990 case definitions.

Personally, I find the collection of these studies and extraction of data to be both interesting and important for future reference. I think it is very likely that the IOM panel is conducting a similar review of the case definition literature, as well. Rigorous examination of case definitions and their applications is essential for the IOM and for the ME/CFS field in general. Bruberg et al found this body of evidence to be “methodologically weak and heterogeneous, making it questionable to compare the case definitions.” What I found both startling and disturbing is that the authors then go on to draw a number of conclusions that are simply not supported by the evidence they examined.

Insupportable Conclusions

First, the authors state, “We found no empirical evidence supporting the hypothesis that some case definitions more specifically identify patients with a neuroimmunological condition.” But they didn’t look! The validation studies they examined applied case definitions, but frequently by questionnaire or retrospective analysis. The studies examined by the authors did not take a group of patients, separate them by case definition, and then look for signs of a neuroimmunological condition. There are studies that do conduct such experiments – testing for immune markers, gene expression response to exercise, etc. – but the authors did not look at those studies because their systematic review was not designed to capture them. It’s overreaching to conclude that something doesn’t exist when you didn’t actually look for the evidence to begin with.

Second, the authors argue that for clinical purposes, a broad definition can be helpful because it identifies more patients who may benefit from treatment. They point to the PACE trial as showing the effectiveness of CBT and GET “irrespective of the case definition which had been used.” But they fail to mention some of the PACE follow-up papers, including this one in which the PACE authors admit that the application of the CDC definition in assessing recovery “may have been inaccurate because we only examined for accompanying symptoms in the previous week, not the previous 6 months” as required by the CDC criteria. This is just one example of the many documented methodological problems of the PACE trial, all of which are ignored by Bruberg et al. A glaring example of this is the statement that “existing evidence indicates that side effects of cognitive behavioural treatments or graded exercise therapy are negligible.” This conclusion ignores analysis by Tom Kindlon of the problems with reporting harms in studies like PACE, and published data from trials that did not find treatment benefit for CBT/GET.

Third, the authors argue that the organic vs. psychic disorder debate should be abandoned because most medical disorders have a complex aetiology, and psychological treatments can have benefit in “clear-cut somatic disorders.” But they then point the finger at patients: “Unfortunately, patient groups and researchers with vested interests in the belief that ME is a distinct somatic disease seem unwilling to leave the position that ME is an organic disease only. This position has damaged the research and practice for patients suffering from CFS/ME.” This statement is patently outrageous simply on the basis of logic. They present no evidence to support their accusation that the organic disease-only position has damaged research and clinical practice. Furthermore, they completely ignore the very real and logical possibility that the reverse is true! In other words, it is equally possible that the the people with vested interests in the belief that ME/CFS has psychosocial causes are unwilling to leave that position, and have damaged the research and practice for patients suffering from the disease. I can present ample evidence that this is the case, but the authors seem to dismiss the hypothesis outright.

Fourth, the authors conclude that, “Development of further case definitions of CFS/ME should be given low priority.” Instead, “Priority should be given to further development and testing of promising treatment options for patients with CFS/ME.” How is it possible for effective treatments to be developed if there is not an accurate case definition to identify patients for the studies? The authors admit that classifying severity and symptom patterns to identify potential predictors of treatment benefit “might be useful.” I would say that such work is essential. Furthermore, given the strong methodological criticisms of validation studies raised by these authors, their analysis seems to support a strong argument for further work on validating and refining case definitions, rather than throwing up one’s hands and working with what we’ve got.

Conclusion

Overall, the authors’ collection and review of the ME/CFS case definitions and validation studies will be very useful as a future reference. But they leap far ahead of what their review data suggests, and draw multiple conclusions with little or no supporting evidence. By failing to correct for their own cognitive biases, the authors allow assumptions to contaminate their conclusions.

The IOM panel, AHRQ systematic review, P2P Panel, and other researchers must be mindful of this in conducting their own analyses and drawing conclusions. Otherwise, we are at great risk for the cognitive bias and overreaching of Bruberg et al to become embedded in the literature and accepted as true.

*Bruberg K, Fonhus MS, Larun L, Flottorp S, Malterud K. Case definitions for chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME): a systematic review. BMJ Open2014;4:e003973 doi:10.1136/bmjopen-2013-003973

Yay! The CFS Advisory Committee will meet on Tuesday, March 11th from 12-5pm. This is the makeup day for the meeting cancelled on December 10, 2013. Boo! This is another webinar, and one can only hope the technical aspects will be smoother than the mess in December.

Yay! Registration is not required for this webinar. Boo! No instructions have been posted for joining the meeting.

Yay! The agenda is available. Boo! The minutes from the December 11, 2013 meeting are not.

Yay! The public comment originally scheduled for December 10 has been placed on this meeting’s agenda. Boo! No other public comment – including written comment – is being accepted for this meeting.

Yay! We will hear updates from the agencies who did not present at the December meeting. Boo! We are not scheduled to hear anything from NIH and CDC – which means no update on the multisite study or the P2P meeting. Given the importance of both, this is a gaping hole in the agenda.

Yay! We will hear more from the two working groups. Boo! It remains to be seen if they have made significant progress in the last three months. I hope so.

Yay! I’ve been told there will still be a “spring meeting.” Boo! No idea when that will be, whether it will be in person, etc etc etc. Edited to add: An email on the CFSAC listserv dated November 12, 2013 stated that spring 2014 CFSAC meeting will be an in-person meeting.

Yay! CFSAC meetings are, at the very least, a chance to learn more about what is or is not being done at the agencies. Boo! There is no time on the agenda for public Q&A, so we have to rely on the CFSAC members to ask the right questions. This has been a problem in the past. In fact, the lack of questions from members means that many issues are discussed at one meeting and then never followed up on at subsequent meetings. Case in point: CFSAC asked to see copies of the CDC’s revised Toolkit prior to its release, and further suggested that a black box warning regarding exercise be placed on the CDC’s website. There has never been follow up on the record for either issue.

As an aside, I refreshed my memory of the December meeting by rereading my commentary and I had forgotten how disastrous that meeting was. It’s hard to imagine next week’s meeting being worse, but I’m also not getting my hopes up. CFSAC has a long way to go to make up for the deficiencies of the December 2013 meeting.

I had it all under control. After coming down with a virus (mild flu?) in January, I got the message and seriously scaled back my activity. Joe Landson, Denise Lopez-Majano, and Claudia Goodell all came through for me in a big way, and shared tremendous guest posts to give me time to recover.

It took close to three weeks, but I clawed my way back to what my friend Trina Berne calls “Normal Shitty Baseline.” I felt my brain waking back up, and was able to start cooking dinner again. Then earlier this week, my husband came down with flu-like symptoms plus sinus infection symptoms. He went on Tamiflu and antibiotics, and while the drugs have helped, he has been pretty sick and miserable all week.

I bet you see where this is going. My nasal congestion and sore throat returned yesterday, and I woke up at 5am with a fever. Now I’ve got a call in to my doctor to see if I need Tamiflu. Because really, this is exactly what I needed to happen now. NOT.

What’s weird about it is how different this is from my usual ME/CFS pattern. For most of the last 19 years, I haven’t caught upper respiratory infections very easily. My husband gets several a year, but I almost never catch them. I know many patients are susceptible to any infection that comes along, but I seemed to be in the category of patients who were not. I always attributed it to a heightened immune response connected to ME/CFS.

Catching two bugs in a month is extremely unusual for me. It could be random chance, of course. But as with most symptom changes, I always wonder: is my ME/CFS changing? Is my immune system starting to break down after almost two decades of illness? Will this be the new normal?

Hopefully, I won’t get as sick as I did last month. Hopefully, I’ll be able to follow through on some of the posts I have planned for March. Hopefully, this is just a glitch and I won’t be dealing with fevers and congestion all the time.

Because life with ME/CFS can be miserable on a good day, and dreadful on bad days. Because I hate being held hostage by my body. Because I am already so limited by this disease, I resent the hell out of anything that limits me more. Because I would rather be writing.

The final post in this stretch of guest authors comes from Claudia Goodell. Claudia is among the most proactive ME/CFS patients I know, trying to make a new life for herself with this disease while also participating in advocacy.

I am one out of 1 million Americans waiting for decades in a medical “no man’s land” for solutions to a debilitating disease with no known cause, NO approved treatment and none in the pipeline. We have no designated specialists, and no cure in sight. We feel abandoned by our government who funds research on our disease at a rate less than that of male pattern baldness, we feel failed by the researchers and drug companies who can’t seem to make progress fast enough, and we feel ostracized by the medical profession who throws us around like hot potatoes hoping someone else will handle us. If we are fortunate we have a support system and receive disability, but many struggle alone with no finances and no one to help them, some of whom are completely bedbound. We are so determined to return to the healthy active lives we once knew that some of us will try whatever we can to get well.

When I was in graduate school my professor of Auditory Neuroscience and Psychoacoustics lectured us about sound pressure. In teaching us the mathematical equation for sound traveling through the acoustic system, he made sure we understood that if one looked at only the first part of the equation it would appear that an acoustic signal actually gained energy as it passed through the middle ear. However, this increase only compensates for the loss of energy that eventually occurs when the sound enters the fluid filled inner ear. The net amount is actually a slight loss in energy, and if you see the entire equation this is clear. In order to make this point he taught us, “There ain’t no such thing as a free lunch” (TANSTAAFL), meaning that even if something seems like it is free, there is always a cost, no matter how indirect or hidden.

While I didn’t retain much of my hearing science knowledge, I remembered TANSTAAFL, and ME/CFS reminds me of this every single day. It’s as though I am an old fashioned wind up clock ticking along and then running down. As I run out of energy my tick tock sound gets slower and slower.  I sit on the table for various intervals, until someone randomly walks by, sees me and decides to rewind my mechanism. I may be mid-way between fully wound and fully spent; sometimes they rewind me all the way, and other times just a few rotations. I never know how much energy I really have. I just keep tick-tock-ing at whatever level I am capable given the amount of energy at any one time.

I worry. I worry that if I stop ticking I’ll suffer a slow, progression of this awful disease that forces me to stop moving.  It’s not because I want to stop moving, or because I’d rather sit around than be active. Nothing could be farther from true. But every time I feel well enough to move, and I get out there and do the things I love, at a much reduced level than before the disease I am left feeling a relapse of symptoms for days, weeks or months. This is not motivational, but fortunately I was an athlete before becoming sick, and I am a determined person.

I do all the good things I can to stay in control of my symptoms as best I can. I avoid foods and drinks that my body doesn’t tolerate, and I take only the few medicines and supplements I really need. I insure ample good quality sleep, drink plenty of water, get regular massage, meditate, walk, do yoga, advocate, and I paint. Although this practice gets me close to maintaining some sort of balance between staying somewhat active and being too sick to move, unfortunately none of this is enough to create what could even loosely resemble a full life. I am unable to work, unable to travel without relapsing, unable to participate in sports at a level I would like, and socializing is minimized. So, to quote a famous movie, “I’m not dead yet”, but I’m not really living either. I’m occupying no man’s land with the rest of my fellow patients, and none of us wants to be here.

I continue to struggle with the crash from hell, but Denise Lopez-Majano has graciously provided a guest post. Her thoughts on ME-frustration are right on target for me this week! As a caregiver for her two adult sons, Denise is intimately acquainted with the frustration we feel.

Anything to do with this illness is fraught with frustration. A few examples:

Getting a diagnosis often takes years during which time patients and their caregivers are frustrated at every turn.

Getting to see a specialist is also a lengthy and frustrating process for most patients, often involving lots of travel and other expenses in addition to medical expenses.

Processing insurance (if one is lucky enough to have it) is frustrating, time consuming, and hugely cognitively taxing (even for healthy people).

Then there is the frustration of trying one treatment modality after another to try to regain some function.

The unpredictability of the illness is a constant frustration as one tries to learn what is a symptom of the illness and what needs to be addressed by another medical professional (if we can find one who believes in us).

Trying to avoid the exertion that brings on a crash is an exercise (pun intended) in frustration as crashes can be brought on by exertion beyond our limits, by additional illness (example, a cold on top of everything else), and sometimes for no apparent reason because we can be as diligent as possible about staying within the boundaries dictated by ME, and yet sometimes a crash comes on us nonetheless.

The frustration and desolation of losing connections/contact with others as our lives become more and more circumscribed by limitations imposed by illness.

There is frustration as we try to adjust to the decreases in activity levels ME imposes on us.

The frustrations of dealing with the daily stigma/disbelief we encounter from those who don’t understand -  whether it be implying we don’t try hard enough, or “I must have that, I’m tired also”, and on and on.

As limiting as baseline is for most of us, when patients are below baseline, the level of frustration increases with heaping doses of fear piled on top as well and it is a very unwelcome combination.

There is the (often unspoken) fear that this crashed level may become the new baseline and the fear of all the the ensuing adjustments that would need to be made and the fear of the additional losses, increased isolation, additional disbelief, etc…..

Additional frustration gets added to the mix because there is so little we can do to alleviate a crash. It makes us feel even more powerless in our dealings with this beastly illness. For me it is particularly frustrating not to be able to help loved ones/friends get back to baseline.

Frustration and feeling powerless often stops us in our tracks. How do we get going again? How do we deal with the frustration and powerlessness?

I have noticed that when I am feeling particularly frustrated/powerless I often listen to intense music. The music seems to encourage me to validate the pent-up feelings of loss, grief, frustration and powerlessness and I can then turn back to advocacy/caregiving with a bit more determination. While for many, listening to intense music – especially during a crash – would not work, it seems to help me.

What have you tried?

What has worked for you?

Credit: Jacquelyn Martin

I continue to struggle with the crash from hell, but Joe Landson has graciously stepped up to provide a guest post. His chance encounter on the streets of Washington, DC gave him a powerful insight into our own advocacy situation.

One Sunday in early January, I mustered the energy to take my mother to the National Gallery of Art in Washington, DC, just a few blocks from the Capitol Dome. We walked past a steam grate with the usual collection of homeless men gathering warmth from it. However, one man was different, and stood out. First, he was white. Second, he was very young. Third and most oddly, he made direct eye contact with me, following my movement as I walked past holding an umbrella over my mother. The moment stuck with me, but what could I do? My first duty was to my mom.

The next morning I saw this article about him. For those of you who don’t have time to read the article, an Associated Press journalist photographed the very same man for a story about the cold snap here in the mid-Atlantic. While scanning the news, a friend of friend recognized the young man’s published photo, and alerted his parents. They in turn brought him home to upstate New York. He had disappeared from their house on New Year’s Day.

Yes, it is the same man as the one I saw. I recognized him instantly in the article photo, and the Federal Trade Commission building is directly across from the National Gallery of Art, where my mother and I went.

At first, I couldn’t help but wonder at how unlikely and lucky this rescue had been. Then I wondered if his rescue would ‘take’, if whatever compelled him to leave would be resolved, or if he would be back out on the street again in a month or three. And then after that, I wondered if he was us.

We with ME/CFS are homeless patients with an orphan illness. No medical specialty has claimed us. We have no medical authority to trust. To casual observers, we are obviously lazy whiners; to those paying attention, we are an unsolved mystery. In any case, we are waiting on a steam grate for some random investigator to publicize an image of us that will produce a shock of recognition for the awful disease we know we have.

We are waiting for our image to be recognized, because it’s hard to believe that anything we say makes any difference. I certainly get that feeling from the many meetings I have attended. Certainly nothing the homeless man said in a news interview could have mattered to the journalist, even if she wanted to help him. He was, quite literally, background for a story. But publishing his photograph possibly helped more than anything else she could have done.

It has been said before: 80 percent of success is showing up. So I’m beginning to wonder if all we can do is show up and wait – in other words, the two things we are least able to do. We desperately need help now if we are to recover anything resembling lives for ourselves. We only survive the endless waiting by NOT showing up – by skipping out on work, family duties and events, and virtually every aspect of public life.

It is very, very hard for most of us to show up and be seen: By doctors who don’t get it. By disability adjudicators who don’t believe us. By government officials with no sympathy. We meet cynical contempt everywhere, over and over again. Yet I’m becoming convinced that being seen, like formerly homeless Mr. Simmons was seen online, is the only hope for us, the only effective thing we can do. We can speak all we wish, to demand attention and more research, and we have at least partly succeeded here. However our words are powerless to influence the medical ‘facts’, as the licensed fact-makers see them. To the doctors and researchers, we can only mutely present ourselves for observation.

Last week, my body gave me the finger. I can’t even say I didn’t deserve it. After more than 19 years, I still don’t always listen to my body and this makes her very, very angry with me.

I’ve previously talked about how rotten 2013 was for me personal life-wise. And about how advocacy work around the IOM contract has been overwhelming. But I am a stubborn woman, especially when it comes to learning from my mistakes. Already depleted, I threw myself into the research and analysis of the IOM Panel in December. I spent the month of January preparing my presentation to the IOM (written and video available). And there was that little thing called “the holidays” smack dab in the middle of it.

I thought I had everything under control. I pared January down to the essentials – only what absolutely positively HAD to be done in order to prevent physical, financial or emotional harm to me and my family. And coming to the end of the month, I thought I nailed it. My presentation was ready and basically memorized. I submitted my written version with references on time. And the absolutely-positively-must-be-done-in-January list was mostly accomplished. I was determined to attend the meeting and give my presentation in person.

Then I got sick(er). Not just a crash. I mean SICK. There’s some difference of opinion in the house about whether it was a bad cold on top of a crash (husband’s theory) or influenza partially mitigated by my flu shot (my theory), but it doesn’t really matter. I was bedridden with a fever, etc etc for five days, and the IOM meeting was on day three.

So despite all my preparation and organization and determination, I could not give the presentation in person. My husband later said that he thought the trip to DC would have ended in an ambulance ride. He says I haven’t been this sick since my two day CPET in April 2012.

I gave my IOM presentation from bed, feverish and not really sure if I was saying it right. It seems like I got my point across. And I’ve certainly dealt with the disappointment of missing out on something I really wanted to do before. Endlessly. For more than 19 years. The reality of this disease kept many people from attending the meeting (or even watching it). The reality of this disease exacted a high price from the patients who were able to attend.

But this felt a bit different to me. I felt like I was being punished by my body. That she reached the breaking point with the I’ll-just-push-a-little-harder routine, the endless repetition of I’ll-just-keep-going-until-the-end-of-the-month /project/year/controversy/crisis. And of course, my body had reached the breaking point and I was the one responsible.

Every single patient-advocate I know does this. I started a list, and realized I couldn’t even name you all here because every one of us does this. Sometimes, a patient has to drop out of advocacy for awhile (or forever) to try and recover. Most of us keep going until we just can’t. As soon as we can get back up, we are at it again. ME/CFS voices must be heard, and that means that those who are able and willing must speak. And pay the price for as long as we can.

I know there’s a balance point, a magical formula for parsing out work and rest. It’s just that I don’t like where that balance point is. There is too much advocacy work to be done, and nowhere near enough of us to do it. So many of you are putting yourselves at risk to make things better for all ME/CFS patients. I have to help.

My body gave me the finger. I deserved it. It remains to be seen if I actually learn from it this time around.

This is the text version of my presentation to the Institute of Medicine Panel today. I delivered my comments remotely, because a fever has kept me bedridden for three days. I tried to speak as naturally and extemporaneously as possible, so there are some differences between the spoken and written versions. I also submitted a much longer version with more details and references to the Public Access Folder. Update January 31, 2014: Video of my talk has been posted.

My name is Jennifer Spotila and I have been disabled by ME/CFS for more than nineteen years. I’ve been involved in advocacy for most of that time, including past service on the CFIDS Association Board, and appointment to the FDA’s Patient Representative Program. I write about politics and other ME/CFS issues at occupycfs.com. My perspective on this IOM study is informed by all of that experience, and I believe the one thing you must keep in mind throughout this study is to be as accurate and precise as you can in order to create sensitive and specific diagnostic criteria.

The Challenge

You are facing an enormous challenge, the result of decades of inaccuracy and imprecision in the definition and diagnosis of our disease.

Multiple names and definitions have been used in the last 30 years, and you should go back as far as the 1950’s to examine the descriptions and definitions of ME. Each definition carries with it a rationale, an associated description of the disease, and a set of limitations. Prevalence rates vary because each definition draws a different circle around – or within – a patient population.

Another challenge is that there are no gold standard biomarkers for the heterogeneous Fukuda population. Despite that, we are very close to establishing one or more diagnostic marker, and a number of you have been responsible for that important research. But the breadth and weaknesses of the case definitions have been a huge obstacle to achieving this.

Finally, as you no doubt realize already, there are competing schools of thought on case definition. Does the mixed bag of definitions describe one disease or more than one disease? How do we identify a more homogenous cohort? What should we call it? Who is competent to diagnose it? We do not agree on the answers to those questions because there are no easy answers.

Potential Pitfalls

As in any controversial subject, there are potential pitfalls that could complicate your work.

First and foremost is the fatigue paradigm. Severe fatigue lasting longer than six months is an extremely common symptom experienced by 4-5% of the general population. One study of newly diagnosed MS patients found that nearly 30% had been diagnosed with severe fatigue or CFS in the three years prior to the MS diagnosis. Because it is based on fatigue, the Fukuda definition has been used as a wastebasket for people with unexplained fatigue, consigning them to medical purgatory when they may have more treatable conditions.

It might help to draw a comparison to chronic pain. While pain conditions are researched across diseases to identify common mechanisms and treatments, we do not define and diagnose them that way. We do not diagnose fibromyalgia, vulvodynia, and migraines as one disease that is subtyped based on where the pain is located in the body because chronic pain is simply too common a symptom. I believe the same is true for the fatigue paradigm. Severe fatigue is simply too common a problem to form the basis of an accurate and precise case definition, regardless of how you try to slice it into subtypes.

Second, as Charmian pointed out, it is imperative to recognize which definitions and exclusionary conditions were used in each research study. If you do not take this into account, you will not be able to sort through the evidence with any resulting clarity.

Third, I believe it would be a mistake to lose sight of the impacts your report will have in the real world. As Lori and Pat have said, you will have an impact on research and clinical trials. You will have an impact on policy. You will have an impact on clinical care. The stakes are very high, but there is a solution.

Accurate and Precise

I believe it is possible to create diagnostic criteria that is both sensitive and specific for ME/CFS. To do so, you will need to be as accurate and precise as possible.

Start by setting aside the fatigue umbrella, because severe chronic fatigue is common to many diseases, and should not be used as the foundation of a precise case definition. Instead, focus on the core symptoms of disease. Across multiple studies and surveys, post-exertional malaise – not fatigue – has emerged as the key and most disabling feature of this disease. PEM is an exacerbation of multiple symptoms after mental or physical activity, and can be measured through both self-report instruments and objective biological tests. It is also notable for its use in distinguishing between people with ME/CFS and those with major depressive disorder and other illnesses with a fatigue component. Another core symptom identified in the research is cognitive impairment, particularly memory and concentration problems. Unrefreshing sleep and autonomic symptoms also rise to the top. There are many other symptoms as well, but your diagnostic criteria will be more meaningful if you focus on the core features.

Another approach that will help you succeed is to use frequency and severity thresholds, in addition to a list of symptoms, to identify a more precisely defined patient population. In one study, 34% of healthy controls reported at least 4 out of 8 Fukuda secondary symptoms. When higher cutoff thresholds for frequency and severity measures were used, that number dropped to 5%. If you combine a core symptom set with thresholds for frequency and severity of those symptoms, I believe you will be able to establish accurate diagnostic criteria that have high sensitivity and specificity.

Precision is the most important tool at your disposal, and yet it will not remove all ambiguity and uncertainty because there is so much we do not know about this disease. Three hundred years ago, cancer could only be diagnosed when it advanced to externally palpable tumors. The definition of cancer has evolved from the most severe and easily detected form of the disease to the sophisticated detection and subtyping methods of today.

Your case definition is part of an iterative process, like all case definitions. Most criteria broaden over time, such as the 2011 revision of the diagnostic criteria for Alzheimer’s Disease.  But that only happens after diagnostic criteria have been tested and validated on the more severe forms of disease. Precision based on what we know, is the first step.

Conclusion

In summary, you face the enormous challenge of creating diagnostic criteria for a disease with conflicting case definitions, no gold standard biomarkers, and competing views about how to define the problem. You must avoid multiple pitfalls, including the high prevalence of the symptom of chronic fatigue in the general population and the effects of those competing case definitions on research results, and never lose sight of the very high stakes we face. But there is a way for you to overcome all of these challenges, and that is by being accurate and precise. If you set aside the fatigue umbrella and focus on the core symptoms of this disease, if you establish frequency and severity threshold requirements, and if you recognize that your diagnostic criteria will be part of an iterative process of refinement, then I believe you can create diagnostic criteria that will advance clinical care and research, instead of putting us further behind: a definition that is sensitive and specific for ME/CFS.

I have submitted more details in writing, including references, for your review. Many of my fellow advocates have done the same, and I hope you will review that material with the same attention you have given us this afternoon. If you have any questions about what I’ve presented or if I can assist you further, here is my email address: jspotila AT yahoo DOT com. Patients are an essential resource for your task, and I hope you will involve us to the fullest extent. Thank you for the opportunity to speak to you today.

Next Monday, the IOM Panel creating new diagnostic criteria for ME/CFS will hold a public meeting. This may or may not be the only public meeting for the study, and it will be webcast. As you can see, I am one of seven advocates on the agenda at the invitation of the IOM. Contrary to what some people may think, I believe it would have been wrong to decline the invitation.

Image credits: suzannegaudetbenefit

I don’t know how the IOM selected the people and organizations to speak at the meeting. IOM staff would not release a list of the people invited, just the final list of seven who accepted. They may have invited others, although I haven’t heard anything about it through the grapevine.

There’s been some grumbling about it, including whether we were selected because of the positions we’ve taken on the IOM contract or because of what IOM thought we would say. Honestly, I have no idea. I received the invitation and immediately accepted. Some might think this makes me a sell-out, or that some or all of us should have declined.

In my personal opinion, that’s bogus.

I see this through the lens of my legal training. When the Supreme Court invites you to present oral argument, you don’t refuse to show up because you don’t like Justice So-and-So’s politics. Your role is to show up and present the best argument you can, whether or not you believe that you will win.

Like it or not, the IOM study is happening. None of us can predict the future: maybe the contract will be rescinded, or maybe not. But as of right now, the IOM is in the process of creating new diagnostic criteria for ME/CFS. Why on earth should I refuse an invitation to tell them what I think about that?

A fellow advocate commented to me last week that refusing this opportunity to provide input is a bit like refusing to vote and then complaining about the government. I don’t know if IOM will get it right; I can’t even assess the odds of that happening. But I do know that if we refuse to speak or if we withhold our input, then we place a very heavy burden on the ME/CFS experts on the panel to make all these arguments for us. If we refuse to speak out, then we make it easier for the panel to come up with the wrong answer. I have no delusions about whether anything we say will alter the entire course of the study. But I do know that if we say nothing at all, we are playing right into the hands of those who think we’re not “qualified” enough to be worth listening to.

I know there are people who would prefer not to deal with ME/CFS patients. There are people who think patients don’t know enough to speak on scientific or medical questions. I think it likely that there are people who would prefer for us not to show up at all. Fortunately, I have not encountered that attitude among the IOM staff members. Whether any of the panelists feel that way is anybody’s guess. But I’ve always been the kind of person who speaks her mind, even when some people wish I wouldn’t.

There are advocates missing from the list of speakers who I wish had received invitations. Fortunately, at least a few of them have three-minute slots in the last session of the day. The IOM panel needs to hear directly from these advocates, and I’m grateful that they are willing to speak – especially because of the physical and financial costs of attending the meeting in person. For me personally, I am putting my health at risk to attend in person, but I believe there is enough on the line to make it worth the sacrifice.

Anyone can submit written comments to the IOM panel, and I hope that many people will. Instructions are below. Before you dismiss this opportunity, consider this: do you believe anyone will speak for us if we refuse to speak for ourselves?

If you would like to submit written comments for the first meeting, please use the email (mecfsopensession@nas.edu). Please focus your comments on the following question: “What is the most important aspect or information that this committee should consider throughout the course of the study?” Written comments received by January 22, 2014 will be distributed to the committee before the meeting on January 27. After January 27, written comments should be sent to the project email address (mecfs@nas.edu). All comments will be considered by the committee, but they may be distributed after the meeting is adjourned.

Robert Miller posted a statement on Facebook last night revealing that he was one of the members of the P2P Working Group that met at NIH last week. I’ve posted his full comment below, with his permission.

Bob is very positive about the meeting and P2P process. I’ve spoken with several people who attended this meeting, and I have heard mixed reactions. Some are positive, some quite negative. The members of the Working Group are all, as Bob says, some of our best experts. Bob says that NIH listened to their input, and that the Working Group “drove the agenda” for the P2P Workshop. Again, I have not heard the same optimistic assessment from everyone who attended the meeting.

As I said before, there are two fundamental problems with the P2P process:

1. The P2P panel cannot – by design – include anyone who has ever published on ME/CFS or taken any position on it. The Workshop could be exactly as Bob and the other Working Group members designed, but non-experts will do the evidence review and non-experts will comprise the entire Panel. That Panel will write the final recommendations, not the Working Group or meeting presenters.
2. There is no transparency. I am very glad Bob came forward to acknowledge his participation in the Working Group. But the questions they finalized at the meeting have not been made public. The roster of the Working Group has not been made public (although word is leaking out). My understanding from two people who attended the meeting was that the discussions at the meeting were confidential. While Bob and the Working Group nominated potential panelists, the actual selection process will be done in secrecy. We will have no input, and no idea who those panelists are until the meeting is about to happen.

This is not acceptable to me.

We are constantly admonished not to question the motives of the people involved in these efforts. I do not question anyone’s motives, nor have I seen evidence of a conspiracy. I agree with Bob’s view that we should engage in these issues in a positive and professional way. My advocacy “career” is based on those values.

But a P2P Panel that will not include any ME/CFS experts? A Panel that will be selected behind closed doors? An evidence review conducted by non-experts? And the outcome of the process is a series of recommendations on diagnosis, treatment and research? No, no, no, and no.

The ME/CFS advocacy community would never have accepted an IOM committee that had no ME/CFS experts on it. We already know that the P2P panel will involve no ME/CFS experts. I do not accept this, and neither should you.

People have asked me what we can do about it. I am actively pursuing several options, and I will keep you posted.

Here is Bob’s statement from Facebook last night:

A brief update for everyone: last week I was invited as a patient representative to the NIH Working Group meeting for the NIH Pathways to Prevention Workshop on #MECFS (happening in the future). This is what was described as an Evidence-based Methodology Workshop at last Spring’s CFSAC meeting. I was asked at the last minute because the original patient advocate could not attend. The Working Group was charged with preparing questions for a thorough evidence-based literature review to identify gaps in ME/CFS scientific research, and we recommended expert speakers and independent panel members for the workshop itself. You can find details of the P2P program below. It is an independent scientific review, and the same process has been used before with another illness.

I want everyone to know my perspective. The Working Group was composed of some of our best experts, and I developed real respect for the person who will Chair the independent panel. Our experts and I had real input into the agenda and questions. The Working Group drove the agenda, and we will participate in the Workshop. I believe the prep work for the Workshop is being done with strong representation from our illness, laying the foundation for a good outcome.

I have been pretty ill in recent months, so I have been stingy with my energy. It has been difficult to post a lot about what is happening in our illness. With this NIH Workshop in mind, and the other governmental initiatives occurring in ME/CFS, I want to encourage patients to engage positively in federal work on our illness. We have had 25 years of inaction by the federal health agencies, and that hasn’t been good for us. All of us have asked for a serious commitment to ME/CFS by the federal government. That is what President Obama promised my wife. These initiatives – the FDA Drug Development Workshop, NIH Pathways to Prevention Workshop, Institute of Medicine Diagnostic Criteria Panel, the CDC’s 5 year Clinical Assessment Study – are all steps toward a stronger federal response. All of these initiatives are not an accident – they are the result of years of work by many of our patients and advocates, to change the federal approach for the better. Patients educated the FDA last spring in a way that has never happened before, and we have the same opportunity at the IOM meeting coming up. The IOM has strong representation on the ME/CFS Committee because patients engaged in it. We need to mix our expert clinicians & scientists with new experts in relevant fields of biomedical research to change our health. I welcome all of these initiatives and know that we will have to do hard work on the details. We won’t agree with everything in these processes or outcomes, but we need government support and action to improve diagnosis, treatment and understanding of ME/CFS. I continue to believe we need to Unite to make the most of new government attention and that 2014 will be a turning point in so many ways. Happy New Year to every one who suffers from ME/CFS. I promise we will move forward together in 2014!