“Chronic fatigue syndrome is a name that prevents a proper diagnosis of the real disease" - Comments by Billie Moore

Billie and her son, Eric, 2006

Billie Moore has been on the Board of Trustees of the New Jersey Chronic Fatigue Syndrome Association since 2007. She also served as a Consumer Reviewer for Chronic Fatigue Syndrome for a Peer Review Panel of the Dept. of Defense in 2011. 

She is NJCFSA's representative to CFSAC; NJCFSA is a non-voting patient advocacy member of CFSAC. 

Her son, Eric, had ME for 20 years and died in 2011.

It is not too late to submit comments to the IOM Committee. Send your comments to: mecfs@nas.edu 

Reprinted with permission.
________________________

COMMENTS REGARDING THE ME/CFS DEFINITION STUDY for the public record.

To: Institute of Medicine Panel Members, Diagnostic Criteria for ME/CFS

From: Billie Moore

New Jersey Chronic Fatigue Syndrome Association Advocacy Chair, speaking for myself.

Date: January 22, 2014

It is important to mention once again that the IOM has embarked on a study to determine the definition for Myalgic Encephalomyelitis, often called Chronic Fatigue Syndrome or ME/CFS, when the recommendation of the HHS’s Chronic Fatigue Syndrome Advisory Committee specifically called for the adoption of the Canadian Consensus Criteria definition (Myalgic Encephalomyelitis/Chronic Fatigue Syndrome: Clinical Working Case Definition: Diagnostic and Treatment Protocols) rather than a new study.

Fifty experts in the field of ME/CFS also urged the adoption of this CCC definition and the abandonment of this expensive, time consuming and unnecessary study being undertaken by the IOM. (Fifty experts, it must be noted, is a large proportion of experts in a field with very few knowledgeable practitioners and researchers, unlike most disease fields in medicine where the patient population is over one million in the U.S. alone.) And 170 patient advocates urged the same.

The advice and urging were ignored by the HHS, and here you are.

Since the study is moving forward, I will address my comments to what is a key issue – whether ME is a subset of CFS or whether ME is a separate disease. First, a brief history.

The disease was first called ME in the 1980’s and was subsequently changed to CFS, with a very broad definition including depressive and other psychiatric symptoms included. The Fukuda definition of 1994 did not improve on prior definitions, and significantly did not require that post-exertional neuroimmune exhaustion (PENE) be present for a diagnosis of CFS. (See the International Consensus Criteria definition.) It also required a six-month waiting period before a diagnosis of CFS could be given, a time-requirement no other disease requires. The Canadian Consensus Criteria, which has been recommended to be adopted by CFSAC, was developed in 2003 by ME/CFS experts.

The term ME/CFS was also developed in the mid-2000’s because patients and expert physicians alike recognized that “chronic fatigue syndrome” was a vague, diminishing name with strong overtones of “it’s all in your head.” The joint name, ME/CFS, is now used by many patients and patient groups and much of the Dept. of Health and Human Services. It is still not correct, however.

What has emerged from these decades of misnaming and sloppy defining (prior to the CCC) is confusion and incorrect diagnoses which included those with psychiatric problems. Four possibilities exist regarding the two terms, ME and CFS:

1. Either these two terms define the same illness; or

2. ME is a subset of CFS; or

3. ME and CFS are two entirely different diseases, or

4. the term “chronic fatigue syndrome” is a false construct and invalid.

To address the possibilities listed above I start with #4 and state that “chronic fatigue syndrome” does not exist. It is too vague and imprecise a name for a disease with many discrete and measurable symptoms. (You should have in your packet of reading materials a table called, “Myalgic Encephalomyelitis (ME/CFS) Table of Biological Abnormalities, Clinical/Lab Tests and Drugs with Potential for Repurposing” which gives an outstanding overview of abnormalities of the disease we are trying to properly define.) However, because CFS has been in the literature for 25 or more years as a result of the naming in the 1980’s and 1990’s and definitions mentioned above, there is a logical hesitancy to chuck it into the medical wastebasket. But “CFS” must be chucked, and it is my hope that those on this panel will do away with “CFS” and its poor definitions and properly define the disease which is well defined in the CCC and ICC definitions.

#1 and 3 follow from #4. CFS is a falsely created and defined set of imprecise symptoms; therefore, it is not the same as ME. It is also not a disease that is different from ME. It is not a disease at all.

Number 2 will take some of your time in discussions, I am sure. However, if you can consider that #4 is true, it, too, becomes a non-possibility: ME cannot be a subset of a non-disease.

Nevertheless, let me address this point. For 25 or so years this disease has been called chronic fatigue syndrome. Very likely the majority of patients who have been diagnosed with CFS as far back as the 1990’s meet the ICC definition of ME. (At least three studies have shown that this is the case.*) Why were those patients diagnosed with CFS and not ME? Simply because that while their symptoms met the criteria for ME, ME was not being used as a label in the U.S. until the mid-2000’s. Their symptoms had not yet been listed and explained properly until the CCC came along; everything was called CFS. The CCC definition refers to ME as “sometimes called” chronic fatigue syndrome and adopts the combination name, ME/CFS.

However, what has evolved somehow is the thinking in some quarters that ME is a subset of CFS. No. Some patients have been diagnosed with CFS who do not have ME because of the overly broad CFS definitions still in use (Fukuda, Reeves). But what they really have is unknown because they do not meet the criteria for ME, and “CFS” doesn’t define anything accurately.

“Chronic fatigue syndrome” is not real. It is a false construct. It is a name that prevents a proper diagnosis of the real disease. ME is a neurologic and immune disease with distinct and measurable abnormalities. ME is not a subset of this oddity “chronic fatigue syndrome.” Please dispense with it and adopt the CCC or ICC definitions, the latter being the more up-to-date and comprehensive of the two. At long last, give this devastating, life-robbing disease a proper name and definition that can be used with confidence by experts and non-experts alike to diagnose and treat it.

*1, Jason, L.A., Brown, A., Evans, M., Sunnquist, M. , & Newton, J.L. (2013). Contrasting chronic fatigue syndrome versus MyalgicEncephalomyelitis/chronic fatigue syndrome. Fatigue: Biomedicine, Health & Behavior, 1, 168-183.

2, Jason, L.A., Sunnquist, M., Brown, A., Evans, M., and Newton, J.L. (in press). Are Myalgic Encephalomyelitis and chronic fatigue syndrome different illnesses? A preliminary analysis. Journal of Health Psychology.

3. Brown, A. A., Jason, L. A., Evans, M. A., & Flores, S. (2013). Contrasting case definitions: The ME International Consensus Criteria vs. the Fukuda et al. CFS criteria. North American Journal of Psychology, 15(1), 10

Battle Lines

After listening to the IOM public meeting, after reading about what is happening to Karina Hansen and Justina Pelletier, and after poring over comments on blogs, forums, and news articles, there is no doubt in my mind that we are at war.

It is a war being waged against ill children, and against their mothers and fathers, who have been slapped with unverifiable, and outmoded, psychiatric diagnoses in order to strip them of their civil rights.

It is a war against people who suffer from complex, debilitating, expensive illnesses.

It is a war in which our enemies are ignorance, laziness, vested interests, venality, apathy, and lockstep thinking that places institutional welfare above the needs of individuals. 

It is a war against the weak, waged by the strong.

This war has been instigated, and is being perpetuated, by our most powerful health institutions:

• The continued refusal by NIH to fund meaningful research into ME, which essentially hamstrings our ability to initiate trials for treatments,

• The CDC’s 30-year perpetuation of a ridiculous name that allows doctors to dismiss us (as well as anyone else who presents with “fatigue”),

• The DSM’s expansion of “somatoform disorders,” which provides the justification for labeling us as neurotics,

• The possible inclusion of “Bodily Distress Syndrome” in the WHO ICD -11, a move which would effectively reclassify CFS, FM, IBS and other complex illnesses as psychiatric disorders,

• The creation of panels – the IOM and P2P – which are given the unlimited authority to create and disseminate information across the entire spectrum of health care, but which have no expertise, no experience, and no knowledge of the illness they are charged with defining.

In the context of war, these can only be considered onslaughts.

Why are they doing this?

The answer to that question is actually fairly straightforward: money. As a result of the recent economic downturn, cash-strapped governments with extensive social welfare programs, such as national health plans, are cutting back on social services. The same holds true of insurance companies, which, in the US, always put profits first. Medical care is very expensive, which means insurers, whether they are state or private, will seek any means of reducing medical reimbursement – let’s say by reclassifying diseases which are complex, but not well-established in the medical mainstream, as psychological conditions. (The remedies for anxiety disorders – GET, CBT, antidepressants – are much cheaper than Ampligen.)

That’s the macro analysis. On a local level, people who are part of institutions with long-standing positions and policies have a stake in making sure the boat is not rocked. Their advancement within an institution is dependent on their ability to do only those things which are consistent with what has been done before. The primary goal of any institution is to perpetuate itself.

On a micro level, there are a number of people in influential positions (e.g. Simon Wessely) who have a personal stake in defending the status quo, some of whom have reaped substantial benefits (e.g. a knighthood) for doing so.

What should we do?

There are two trains of thought as to how we should proceed:

1) “If you can’t beat ‘em, join ‘em." This is the approach taken by the Let’s Get It Right Group. The idea is to work within the framework of existing power structures. This is called “reform.”

2) “If you can’t join ‘em, beat ‘em.” This is the approach taken by Jeanette Burmeister, and others. The idea is that HHS has repeatedly let us down, and if we work within their rules, they will do so again. This approach is often called “revolution.”

Arguments about the merits, and pitfalls, of each of these approaches – reform vs revolution – have raged since human institutions were formed.

And they rage within our community today, with acrimony on both sides. But must we fight amongst ourselves about the single most effective strategy?

If the institutions – and individuals – waging this war are hammering us with everything they’ve got, why can’t we do the same?

Weaponry

We have a large community, one that provides us with a broad range of talents, expertise, and experience.

Why not make good use of the entire range of what our community has to offer?

• Those who have rapport with government institutions can exert pressure from within. The IOM presentations were a good example of this. Each group’s statement is now part of public record.

• Those who have experience with litigation can place legal pressure from without. Legal pressure is essential for any change in the US. It is the way our entire system works.

• Those who are adept at public pressure can organize demonstrations (May 12 is coming up!) Public demonstrations and events are a great way to gain the attention of the media. And, because this is the 30-year anniversary of the Incline Village outbreak, now is the time.

• Those who can write should take advantage of the huge opportunities available on the Internet. Public comment on news articles is particularly effective, but there are also review sites, emails that can be sent to public officials and representatives, and old-fashioned letters to the editor. (Even if they don’t get published, editors always read these to gauge the interest in certain topics among their readership.)

• The power of social media is something that cannot be denied. When hundreds of people tweeted about the inclusion of “Bodily Distress Syndrome" in the WHO ICD-11, WHO responded with a statement. Even if you can’t get out of bed, you can tweet, you can “like” posts, you can sign petitions.

There is no institution, here or abroad, that is unaware of the power of public outcry.

So, let’s fight back on all fronts. Together.

HealClick.com: New Patient Community Seeks to Turn Patient Sharing into a Tool for Research

Joey Tuan, Beth Mazur, and Cari Allshouse have launched a project to bring ME/CFS sufferers together, and to gather data for future medical research. This is exactly what the community needs - a way to find a "buddy," and a secure means of gathering medical data.

There are only 9 days left to fund this ambitious project. Click HERE to donate.

___________________

Reprinted with the kind permission of HealClick.

By Emily Craven

By the time I was 25, I’d already been sick with Myalgic Encephalomyelitis (M.E.) for four years, had seen nearly two dozen doctors, had to quit my dream job, and moved back in with my parents. One day, on a rare coffee shop excursion, an old friend asked me, “What do you do about an illness with no recognized treatment?”

It was a good question. I explained that I’d been combing patient forums and online message boards. I was slogging through endless posts, weighing anecdotes about treatments relieving or inflaming symptoms, despite having no way to know if the person posting and I reacted similarly to such treatments. “I spend a lot of time banging my head against the Internet,” I told my friend; I truly felt isolated and helpless.

An Ambitious Endeavor

Enter Joey Tuan, Bath Mazur, Cari Allshouse, and their new startup, HealClick.com. The founders set out with two objectives: 1) To make it easier to find relevant patient-shared information, and 2) To use this information to further medical research.

Launched last month, HealClick is a revolutionary free platform that connects patients with chronic illness from all over the world. This hybrid of a social network and an enhanced medical forum uses volunteered symptom and treatment information to match members with similar users, while at the same time building a confidential database to be used for research. It serves over 20 autoimmune, neuroimmune and other poorly understood illnesses. The ultimate goal is to turn amassed patient experiences into research-friendly data that will yield treatment solutions.

“If we can arm ourselves with a database that truly captures our health over time, we can then present M.E. and other poorly understood conditions as problems worth solving to the researchers, backers, and philanthropists that can help us solve them,” says co-founder Joey Tuan.

Since the January 14th launch, 1294 patients have joined HealClick. Combined with the 900-plus beta users who shared their experiences prior to the launch, the site now has over 2200 members. Together they have already generated 2247 treatment reviews.

Finding Our Patient “Doppelgangers” and Getting Answers

HealClick makes it possible to not only get information about your illness directly from other people who have your diagnoses, but also to learn who these people are and what their health and treatment experiences have been like. Using matching technology analogous to dating sites HealClick creates a “similarity score” between patients. It uses each member’s symptoms, diagnoses and treatments to suggest other members that are health-compatible based on their input. In addition, the site boasts a number of traditional social networking features, such as chat, personal messaging and detailed profiles, making it easier for members to get to know each other. There are also innovative variations of social networking features such as the ability to mark a post as “helpful” and to send other members “LUV” (support) for posts and comments. The result is a light-hearted, supportive community that lessens the isolation of being chronically ill.

HealClick also sets itself apart from other patient forums with its ease of use and by serving numerous diagnoses. First, highly-sought treatment reviews are in a dedicated section instead of buried among endless posts. Second, users with over 20 autoimmune and poorly understood illnesses can easily discuss their treatments and coping strategies for common symptoms, such as nausea. This information would typically be scattered across more than 20 separate diagnosis-specific forums. Discussions can also be sorted into categories for one diagnosis only, making it adaptable to the user’s needs.

Building Technology to Collect and Prepare Data for Research

The HealClick team recently launched its Indiegogo crowdfunding campaign in order to develop the components necessary to collect data and make it research-ready. “If we raise just $50,000, we can build a state-of-the-art website to help millions of patients,” Tuan said.

Among the features slated for development are an integrated mobile health tracking app and upgraded servers to sustain HIPAA-level encryption of the patients’ data. The team is also working on refining the matching process through the addition of standardized lab results and natural language processing.

As a patient, I’m very enthusiastic about an easy-to-use tracking app, one which will allow me to daily track my symptoms and treatment by answering a few simple questions. In addition to tracking my own health, the data collected by this app can be anonymized and made available to researchers to identify correlations and patterns over time. The idea that I will be able to contribute data for desperately-needed research from my bed is particularly exciting. Many chronically ill patients together could change the course of research for their conditions simply by clicking on a few answers each day.

Ultimately, HealClick strives to empower patients by designing the tools for them to educate each other and to directly participate in research solutions.

To join: www.HealClick.com

To support the fundraising effort click HERE.

____________________

Emily Craven is a former domestic violence advocate and Reed College graduate. Prior to becoming ill she was an avid traveler and passionate about education. She has severe Myalgic Encephalomyelitis and has been battling the invisible illness since 2002.

IOM Public Meeting, January 27, 2014: Part 2: The CDC Multi-Site Study and P2P

Below is a detailed summary of the second hour of presentations given at the IOM public meeting on January 27, 2014. The two presenters were Elizabeth Unger, who talked about the CDC Multi-site Study, and Susan Maier, who explained the P2P process.

Dr. Unger's presentation of the CDC study demonstrated the flaws that have been apparent in every federally-sponsored study of ME/CFS to date. The instruments used in the study are old stand-bys, and of dubious relevance. The reason they were chosen for this study is that, with one exception (the DePaul questionnaire), they are standard tools of the trade. The problem is that ME/CFS is not a standard illness. (To quote an old adage: You can't fit a round peg into a square hole.)

Several of the questionnaires covered anxiety and depression. The results proved - again - that ME/CFS is neither. Yet, Dr. Unger reported that mental health was fairly normal in ME/CFS patients with a distinct tone of surprise. Conspicuously absent from the CDC's roster was a single objective measurement (e.g. 2-day CPET). 

The IOM panel, particularly those with ME/CFS expertise, asked pertinent questions, most of which Dr. Unger was unable to answer. Dr. Rowe posed the question of whether a sample of patients who had been ill, and treated, for a long time was appropriate for establishing diagnostic criteria. He pointed out that the illness changes over time. (Actually, it is the patients who change. As Dr. Cheney pointed out 20 years ago, our bodies continually adjust to an ongoing disease process.)

Dr. Unger was followed by Susan Maier (Deputy Director of the Office of Research on Women's Health (ORWH)), who gave a presentation that was so laden with jargon and bad syntax that it was nearly impossible to follow. (She compensated for her inability to form a coherent sentence by a great deal of hand waving.) Maier's subject was the Pathways to Prevention (P2P) panel that is being assembled to review research definitions for ME/CFS.

In all respects, the P2P process is even more far-fetched than the IOM's. The members of the panel not only can't be experts in the field of ME/CFS, they can't have anything whatsoever to do with the illness. What's more, they don't even have to be physicians. They can be "highly regarded lawyers, ethicists, methodologists" - and, one presumes, expert butchers, bakers and candlestick makers.

This hodge-podge panel will review material that has been culled (by yet more non-experts using "standard methodology") and listen to presenters, each of whom has exactly 20 minutes to state their case "pro or con." Maier explains that this process is similar to the jury system, with a prosecution and a defense. The jury knows nothing initially, but is educated - over the course of two days - in everything they need to know to make a judgment.

Not only is this the height of absurdity, it is very definitely not the jury model. In our justice system, the defense and prosecution are able to question the jury to discover if they have any prejudices that might influence their decision. With 85% of physicians in the U.S. believing that "CFS" is a mental illness, and the majority of the public probably believing the same, can we really expect this panel to be unbiased? In any event, there is no mechanism for finding out if they are.

And if this were a trial, as Maier proposed, wouldn't the defendants (ME/CFS patients) have the right to face their accuser? Doesn't a jury have to deliberate until they reach a decision - instead of being given a deadline of 24 hours? Wouldn't they have more than two days to hear arguments? Wouldn't the defendants have a say in what constituted evidence?

To employ a more apt judicial metaphor, the P2P panel isn't a jury - it's a kangaroo court.
______________________________ 
This article originally appeared on ProHealth.

Note: You can read summaries HERE of the introductory segment, including Dr. Rick Erdtmann, Director of the Board on the Health of Select Populations, Dr. Ellen Wright Clayton, IOM Committee Chair, and Dr. Nancy Lee, Designated Federal Officer to CFSAC, study sponsor [HHS] representative.

By Erica Verrillo

The second hour of the IOM public meeting held on January 27, 2014 consisted of reports from related federal projects: The CDC Multi-site Clinical Study and the Pathways to Prevention Program (P2P), formerly known as the NIH Evidence-Based Methodology Workshop.

Each of these studies has profound implications for the ME/CFS community. The CDC study provides data that will be used by both committees, as well as researchers and clinicians, and the P2P panel reviews definitions that will be used by researchers.

You can read the meeting agenda HERE.

You can watch the presentations HERE.

You can read information about the committee members HERE.

It is not too late to submit comments to the IOM Committee. Send your comments to: mecfs@nas.edu 

---

Elizabeth Unger - Methodology for the CDC Multi-site Clinical Study (18:07) (Video)

Dr. Elizabeth Unger is Chief of the Chronic Viral Diseases Branch (CVDB), Centers for Disease Control (CDC). She began her talk by stating that physicians worldwide recognize an illness that is called CFS or ME, and that there are more similarities than differences in the case definitions for how this illness is described.

Dr. Unger pointed out that the illness is very complex and patients are heterogeneous. The heterogeneity of patients in clinical trials and research studies and in epidemiologic studies can confound results. This may be contributing to the lack of uniform advancement in studies of this illness.

Some of the confounding factors for research have been: severity of illness, duration of illness, co-morbid conditions, medications, and demographics (age, sex, etc.) In Dr. Unger’s opinion, medications have been the least investigated, and could be most problematic for understanding the illness.

Clinicians need a case definition that is as clear and simple as possible so that this illness is recognized and patients get the case they deserve.
When designing the multi-site study, the CDC asked themselves two questions:

1. Is refining or adding criteria going to produce a homogeneous population?
2. What do you do with patients who don’t fit any one definition?

As a starting point for these questions, Dr. Unger quoted Stephen Holgate as saying “To call CFS/ME a single disease greatly underestimates the complexity of the problem” and Dr. Anthony Komaroff as saying, “I suspect that none of these case definitions is likely to describe a very homogenous group of patients."

Because the CDC realized case definition alone was not going to be sufficient, they decided to capitalize on the expertise of clinicians who had the greatest experience treating the illness.

The multi-site study examines standardized data from patients aged 18-70 from seven specialist practices in the U.S. The enrollment criteria relied on clinical expertise rather than on any one case definition. I am using the term “CFS,” but we made it clear to physicians that CFS, ME, post-infectious fatigue, or any of these synonyms or possibly related illnesses, were eligible to participate. The only exclusions were HIV-positive patients, and patients older than age 62 at diagnosis.

Participating Physicians:

• Benjamin Natelson
• Nancy Klimas
• Lucinda Bateman
• Charles Lapp
• Andreas Kogelnik
• Daniel Peterson
• Richard Podell

Stage 1: First Year of Study

Dr. Unger devoted the bulk of her talk to describing Stage I of the study, which was the first year of gathering data obtained by physical examinations, patient questionnaires, and data extracted from medical records (e.g. medical history, medications list, lab tests, family history, infection and immunization history).

Data was gathered from 471 participants who filled out questionnaires either at the time of the clinic visit or in the week before. These included: Patient Health Questionnaire Depression Scale, Generalized Anxiety Disorder 7-Item Scale, Self-Rating Depression Scale, CDC Symptom Inventory (which includes 19 symptoms experienced during the previous month), the Medical Outcomes Study 36-Item short form (which measures quality of life), the Multi-Dimensional Fatigue Inventory-20 (which measures general fatigue, physical fatigue, mental fatigue, reduced motivation and reduced activity), questions from the DePaul symptom inventory, NIH PROMIS forms (pain, fatigue and sleep), sleep questionnaires, and Brief Pain Inventory.

Demographics

The patient population consisted of slightly overweight, middle-aged (48.2) white females with college education. Half were married, and 75% were unemployed, though most did not receive unemployment benefits.

The mean age at diagnosis was 38.4%, with sudden onset reported in 65.4%. Mean duration of “fatigue” was 14.3 years, ranging from 2.5 to 52 years.

Dr. Unger commented that the patient population studied through these specialty clinics was not necessarily representative of the patient population as a whole. Most patients were highly educated and insured, and all had been seen and evaluated by other physicians.

Results

The highest PROMIS scores were in generalized fatigue, physical fatigue, and reduced activity. On the SF-36 mental health was relatively preserved, while vitality was reduced. Functional measures included the mean number of hours for vertical activity (7.5 hours), the mean number of hours for horizontal activity (12.8 hours). The mean days per week in which patients exercised was 3.3.

Overall, 83% of patients reported pain in the previous week. There was a good correlation between PROMIS scores and the Brief Pain Inventory (.75).
The CDC symptom inventory showed the most prominent symptoms to be Post-Exertion Malaise (PEM) and unrefreshing sleep, followed by muscle pain and memory and concentration problems.

In the context of other illnesses, PROMIS scores showed that CFS patients experienced greater fatigue, sleep disturbance, and pain impairment than patients with chronic pelvic pain, spinal cord injury, Muscular Dystrophy, Post-Polio Syndrome, and MS.

---

Q&A with Dr. Unger (11:01) (Video)

Question – Nancy Klimas: Have you been able to do an analysis of different case definitions on this data set?

Answer: We have not done that but it is certainly possible. The data is there. That requires a lot of interpretation that we feel will require a lot of dialogue with people, in other words, how to set the criteria for each of the points in the disease criteria.

Question – Nancy Klimas: You have the DePaul symptom inventory on the list. Lenny Jason has an algorithm on an excel spreadsheet that we can use to compare Fukuda to the 2003 Canadian.

Answer: It still requires some interpretation and discussion. The data is there.

Question – Lily Chu: What is the timing and how will the committee receive data? My second question is, I can understand why you are asking clinicians to come up with who fits the CFS diagnosis. Is there any thought given to asking two or three physicians looking at the data if they would agree that this person has CFS? My third question is, you have measures of activity, but other than upright activity, are there any subjective questionnaires of what people do day to day?

Answer: None of these patients were diagnosed by a single physician. They are referrals. I think they [the physicians] are competent in their diagnostic skills.

As for measures of activity, we do not have narrative of what patients do in a given day. But all the questionnaires have time frames. They vary from being vague, others are for two weeks, and the DePaul is for six months. Each has a narrow-minded way in which they are asking questions. This illness defies neat categories. We have to be careful interpreting the data we have.

About logistics – we have the baseline data ready. We can provide the committee with tables and answers.

Question – Dr. Clayton: Did the patients consent to data sharing?

Answer: Patients consented to data sharing within the study. We feel we can release the analyses, but not the raw data, to the IOM.  We feel that’s within informed consent.

Question – Peter Rowe: I have a question about the relevance of prevalence data. You have got patients who have been treated and followed for some length of time. It seems to me it would be problematic to apply those to case definitions. My hope in clinic is that I will have some impact on some patients’ initial CFS symptoms  within a short period of time. So the problem might be that if people have been treated by such an experienced group, some of their symptoms that might be defining symptoms for CFS/ME will be gone by the time you’ve captured the data. How are you approaching that dilemma?

Answer: It is a dilemma. We sort of try to do one step at a time. We are aware of that. I was glad we had 80 patients that were only 2 ½ years from the onset of their illness. In the next phases of the study we have asked physicians to enroll patients earlier on in their disease course. As a last point, we have the intake form which gives a picture of the patients when they first come in, but even there, because this is a referral clinic, this may not be what the illness looked like at first. It’s not only the patients’ response to therapy, but the illness itself changes with time. That is a limitation that we have.

Question – Cynthia Mulrow: I want to understand the representativeness of this sample and what likely spectrum of CFS they might represent. If a large spectrum has not been captured, then it may not be right for us to go overboard using these data to help inform diagnostic criteria.

Answer: We think that it is data that could be used to inform you but we don’t think that absolute decisions can be made solely on the basis of this data. It’s a beginning point. I think that the patients that are in this study should meet the case criteria, whatever the case definition is. That’s not to say others wouldn’t.

The other piece of data in the comparison we have is the data on our population-based surveillance. We use many of the same instruments, so we have a data from those two and we can see how they compare.

This is to me one of the most important steps the field can take is getting some consensus on what are some shared measurements that studies and researchers and clinicians can count on and use, so that we can start making comparisons. One thing we’ve been lacking is measurements of this illness we can use, that’s why I like the PROMIS instrument so much. It compares patients with other patient groups.

Question – Lily Chu: Regarding timing there are studies, and patient anecdotes, indicating that sore throat and flu-like symptoms go down with time and neurologic symptoms go up over time. To Cynthia’s point, there are some epidemiologic studies that suggest that minority and lower socio-economic groups are high risk for CFS and may have more severe cases. This group [CDC study population] is highly educated, has resources to see referrals, etc.

---

Susan Maier - NIH Evidence-Based Methodology Workshop (18:27) (Video) 


Dr. Susan Maier is the Deputy Director of the Office of Research on Women's Health (ORWH). She began her presentation by clarifying that the Pathways to Prevention Program (P2P) is a collaboration between NIH Office of Disease Prevention and the Trans-NIH ME/CFS Research Working Group.

The goals of the program are to identify research gaps and methodological scientific weaknesses in a scientific area, to suggest research needs, and to move the field forward through an unbiased and evidence-based assessment of a public health problem.

The P2P Program is for any disease, illness or syndrome that needs a more in-depth understanding, keeping in mind that the goal is to serve the community of researchers.

Timeline

The P2P process was begun in June 2012 when the Trans-NIH ME/CFS Research Working Group and ORWH began formulating a proposal. The Trans-NIH ME/CFS Research Working Group proposal was accepted by the Office of Disease Prevention in December of 2012. The first working group meeting to plan the workshop was held in January 2014. The tentative date for the workshop is December 2014.

Making a Case for ME/CFS

• Necessity – lack of physician recognition, lack of biomarkers, lack of understanding about the scope and range of the illness
• Urgency – there are no FDA-approved treatments
• Public Health Problem – people are sick and there is an unmet need

Working Group Meeting (January 2014)

The working group is composed of experts in the field, patients, advocates, caregivers and Federal partners (e.g. CFSAC, HHS )

They had three tasks.

1. Refine the questions for the evidence review. The evidence review is performed by an evidence-based practice center – this is a contract done through AHRQ (Agency for Healthcare Research and Quality).  
2. Develop the workshop agenda: Topics, speakers and format.
3. Nominate panel members.

Questions considered by the panel

Questions had to be developed and refined, because the questions form the basis for the agenda of the workshop. (Who will speak and on which topics.)

• How do ME and CFS differ?
• What tools will allow us to define subsets across the entire subset of CFS?
• What are the characteristics of patients who respond to specific treatments across the spectrum of CFS?
• What does research on ME/CFS tell us about clinical diagnosis of ME/CFS?
• Have previous research findings shaped current clinical practice or are research and clinical practice completely separate?

Two days will be spent listening to speakers on topics that have been defined by the committee. Speakers are experts in the field - clinicians and researchers – who represent a pro or con position concerning existing case definitions in order to provide a balance. Each speaker will be allotted 20 minutes, followed by a question and answer period.

Nomination of Panel Members

Panel members are highly recognized experts in their areas. These are not experts in ME/CFS, and not necessarily medical doctors or researchers. They may be attorneys, methodologists, ethicists, public representatives – as long as they have no direct connection with CFS. None may be employees of the Federal government.

What happens next?

ODP will set a date for the speakers. Evidence-based practice center will cull the evidence using standard methodology. Every meeting must be approved, so this workshop must be approved by the Federal government. Once the approval has been granted all information will be made public, including speakers and evidence.

The end result is a set of recommendations from the panel based on what they have read and heard. A follow-up (1-9 months later) will then work on how to process the recommendations and put them into practice.

P2P Contacts:

Paris Watson, Senior Advisor, P2P, Office of Disease Prevention (ODP)
watsonpa@mail.nih.gov  prevention@nih.gov

Susan E. Maier, Deputy Director of the Office of Research on Women's Health (ORWH) Susan.Maier@nih.gov

Mariela Shirley, Chair, Trans-NIH ME/CFS Working Group shirelym@od.nih.gov

---

Susan E. Maier - Q&A (14:59) (Video) 

Question - Theodore G. Ganiats: At the end there are some recommendations. Are those recommendations for research agenda for the NIH, or are they for practice?

Answer: It has the potential to be both, but our focus is on research that is used for translation into clinical care means we have to focus on “besting the science.”

Question - Theodore G. Ganiats: In the group of panelists, there was nobody from primary care. Given that most patients with this disease, whatever we call it, will be seen I primary case, I think that adding primary care would be important.

Answer: Absolutely.

Question - Lily Chu: On the list of refining questions, it says, “What does research on ME/CFS tell us about clinical diagnosis of ME/CFS?” That seems very similar to the charge of the IOM. How does that work between the NIH and IOM?

My other question is that I’ve been reviewing the P2P website to understand how the process works – you can’t be on the panel if you are a clinician with any experience, or you cannot be a researcher that has published on it? That was concerning to me because this is a complex illness.

One of the requirements for the topics is that it be non-controversial. This is a field that is undergoing a lot of changes. My concern is that there is not going to be anyone with any clinical or research experience on the panel.

My third question is, according to my understanding, the panel writes the document in 24 hours after the workshop?

Answer: The panel receives the evidence report, and any other documentation that is submitted, six weeks before the workshop. At the workshop, the panel will listen to speakers, and speakers will provide additional information that may not be in the evidence report. For example the CDC study may not be published yet. But that information is very important for the panelists.

Yes, the report is written in 24 hours. They do have a day and a half total. That report will be posted on the website for public comment. After 14 days, it will be taken down and the comments will be reviewed. The report will incorporate that content.

There is a link between what the IOM is doing and what we are doing. So, we hope to be able to work with the IOM committee to share the synergy.

About the panel requirements –  the panel members may not be clinicians who see patients with ME/CFS or researchers who study ME/CFS. They could be a clinician that treats pain disorders, or an oncologist that treats patients with cancer-related fatigue, they could be in a research area that has recently gone a case definition review, such as PCOS. They are highly regarded experts in their field, just not in ME/CFS. That content comes from the speakers.

Think about this: It’s a jury model. You have the defense, you have the prosecution. They know the case very well. The jury is sequestered; they don’t know, they don’t know anything. The jury hears the evidence. They make their decisions based on the evidence.

Question - Nancy Klimas: I’m challenged, because I am on both groups. My understanding is that IOM is not able to put anything out until its final report. It would be very difficult to “synergize.” But it can certainly hear, so it’s a one-way direction from P2P. There is so much that would be duplicated, literature reviews and so on, you have this group doing a comprehensive review of the entire literature, obviously that would be something this committee could really value.

The case definition charge itself is difficult. If this group is really focused on a clinical case definition, then the P2P is really focused on a research case definition.

Answer: The goal of the P2P is not to develop a research case definition, but to review it,  review the evidence supporting all case definitions that have been used.

Question - Nancy Klimas: How do you see the one-way direction of this information flow? I am worried about the time lines. When will the Portland group have the evidence from ARHQ?

Answer: The evidence review will be completed roughly six weeks before the workshop. We are “planning” for December 2014.

Question - Nancy Klimas: Might it be possible in order to facilitate this group’s work, to ask that the case definition part be available to this committee sooner than that, because it is too late to be useful on our timeline.

Answer: The two processes are different. Our purpose is different. Our questions will be heavily focused on the research part. That doesn’t mean it won’t be useful for clinical use. Our evidence may not exactly dovetail what you are looking for.

Question - Nancy Klimas: Can those of us who are on both committees have permission to share?

Answer: The content of the meeting is not finalized. All the things we discussed are on sheets of paper and maybe a couple of computer discs. When it’s pulled together, at that point you can share it.

Question - Nancy Klimas: I’m totally confused.

Answer: Paris Watson can give you that specific guidance.

Question - Cynthia Mulrow: My understanding is that protocols for evidence reports are publicly available. Are there ways that we can get access to search strategies that are used in the evidence report tied to specific questions, so that we can peruse those before the final report comes out? My understanding is that these evidence centers develop evidence tables before they finalize the report. Is there a way that our committee can have access to those interim products?

Answer: I don’t know the answer to that. But I can certainly ask.

[Dr. Clayton announces a BREAK.]

IOM Public Meeting: Part 1 - The Process

Below is a detailed summary of the first hour of the IOM public meeting held on January 27, 2014. Even though the agenda was largely focused on bureaucratic process, it is well worth listening to these presentations. 

The underlying tone of the introductory statements was defensive, no doubt due to input from ME/CFS advocates and experts who believe the IOM process to be a waste of time and money, if not outright harmful. There were several references made to the "rigor" of the process, which, in Dr. Clayton's words, made "everything else pale in comparison," and an emphasis on procedure.

There was, however, no mention of specifics. While it was made clear to those listening that many steps would be followed, that these steps would be reviewed and overseen, and that the final product would be examined and re-examined, one crucial question remains unanswered:

If the IOM review is supposed to provide the basis for a new case definition, exactly who determines which material will be read by the committee? Do they plan on reading everything that has ever been published on ME/CFS? If not, how do they intend to cull their material? Dr. Erdtmann mentioned that all "outside" material would be available to the public. Is there "inside" material? If so, what is it?

Unfortunately, we will never know the answer to these questions. This is a "confidential" process, in which members of the public will not have access to the materials, the interim reports, or anything else of substance.
  
While the non-expert members of the committee demonstrated their professionalism by asking appropriate questions, it is clear that they are in over their heads. Given the limitations imposed on most of the committee members by sheer lack of knowledge, it is hard to imagine how they will be able to sort through the vast, contradictory corpus of literature on ME/CFS. They have neither the perspective that long association with patients brings, nor the critical capacity that a thorough understanding of our political history provides.

_______________________

By Erica Verrillo

On January 27, 2014, the Institute of Medicine held a public meeting concerning the IOM’s review of diagnostic criteria for ME/CFS.

The first half of the meeting consisted of presentations by government and IOM representatives. The second half included presentations from patients and patient advocates, as well as public comments.

This article covers the initial presentations, in which the IOM process was described.

• Rick Erdtmann outlined the four steps of the IOM study.
• Ellen Wright Clayton, Committee Chair, welcomed the participants and established the limits of the public session.
• Dr. Nancy C. Lee, representing the sponsor of the study, HHS, talked about some of the background that led up to the IOM review.

You can read the meeting agenda HERE.

You can watch the presentations HERE.

You can read information about the committee members HERE.

It is not too late to submit comments to the IOM Committee. Send your comments to: mecfs@nas.edu 

---

Rick Erdtmann – The IOM Process (7:24) (Video)

Dr. Erdtmann is the Director of the Board on the Health of Select Populations, which is in charge of the committee. He outlined the four stages of the IOM study.

1. Define the problem. When this is accomplished, the committee prepares a Contract of Work.
2. Select experts to author the report. The selection of a provisional committee is accomplished through an “extensive search.” The provisional committee is then reviewed to ensure that there is no conflict of interest, and that there is a “proper balance of views.”
3. Gather information based on presentations and research by the committee and staff. Any information from outside sources is listed in files available to the public, but the actual deliberations of the committee are not revealed. A confidential report is then drafted.
4. Peer review process. Volunteer reviewers who roughly match the expertise of the panel are nominated to review the draft. That slate is subject to internal academy review “above and beyond” the purview of the study committee. The reviewers are asked to make critical comments, to clarify errors and offer new insights and suggestions. Reviewer comments are submitted anonymously, and the committee then addresses each comment and modifies the draft. The Report Committee oversees the review process, and ensures that the comments are addressed. Then the report is released to the sponsor and to the public.

________________________

Ellen Wright Clayton, Committee Chair - Introduction to Public Session (4:17) (Video)

Dr. Clayton welcomed the committee and expressed her enthusiasm for the project. She then stated the committee’s task, which is “to evaluate comprehensively the current criteria for the diagnosis for ME/CFS.” Dr. Clayton stressed that the public session was for information gathering only, and that any views expressed by committee members were not to be taken as indicators of the committee’s position. While questions were not allowed from the floor, Dr. Clayton pointed out that “probing questions” would be posed by committee members. After repeating several times that the process of review would be rigorous, Dr. Clayton then introduced the sponsor’s representative. [HHS is the sponsor of the IOM study.]
_________________________

Nancy Lee - Background and Charge to the Committee (15:51) (Video)

Dr. Nancy C. Lee is the Designated Federal Officer for the Chronic Fatigue Syndrome Advisory Committee (CFSAC). Dr. Lee reported that in 2012 CFSAC recommended that HHS convene a workshop to reach a consensus on clinical and research case definitions for ME/CFS. The Secretary of HHS asked IOM to use its consensus process to provide guidance to the broader medical community on how to identify and diagnose ME/CFS in the clinical setting. This includes primary care providers as well as some specialty groups. HHS asked that four specific tasks be addressed. The charge of the IOM committee is to address four specific tasks:

1. Conduct a study to identify the evidence for various diagnostic clinical criteria for ME/CFS using a process with stakeholder input, including practicing physicians and patients.
2. Develop evidence-based clinical diagnostic criteria for ME/CFS for use by clinicians, using a consensus-building methodology.
3. Recommend whether new terminology should be adopted.
4. Develop an outreach strategy to disseminate the definition nationwide to health professionals.

Dr. Lee asked that the committee consider the variety, range and severity of symptoms. “It is also important to distinguish between generalized chronic fatigue with known etiology and unexplained fatigue without associated multi-system illness and the more specific syndrome of myalgic encephalomyeltitis.”

"As you probably know there has been support from advocates, as well as some of the members of this committee, for the Canadian Consensus Criteria of 2003. Criteria should account for different sub-populations, such as children," she said. “The committee may well decide that different criteria are needed for the different entities that fall under the broad umbrella of CFS.”

The third task is to evaluate the terminologies for ME/CFS. These terminologies include but are not limited to: neuro-endocrine immune disorder, myalgic encephalomyelitis, chronic fatigue syndrome, and chronic fatigue immune deficiency syndrome. Different names may be warranted.

“Please know that there are many people – scientists, clinicians, advocates - who believe the name ‘chronic fatigue syndrome’ does harm to patients. I agree.”

Dr. Lee asked that the committee recommend strategies to disseminate these criteria nationwide to patients, and health care professionals. “Pushing out the criteria to a wide audience of health professionals is such a critical need. Whatever the committee develops needs to get into textbooks, widely read journals, and be at the top of a google search.”

Dr. Lee asked the committee to coordinate their efforts with two ongoing efforts: The CDC multi-site study and the NIH Pathways to Prevention (P2P) Program for ME/CFS.

Dr. Lee closed her presentation with the statement that “Patients and providers need help now.”

Dr. Lee Q&A (7:08) (Video)

Question - Dr. Clayton: There are two other efforts that we will hear more about. But I would like to hear from you how these efforts are going to interweave to create a coherent whole.

Answer: A better answer to your question is going to come from my two colleagues. For the CDC, that was designed to gather data to address the issue of what are their symptoms, what are their lab values. So, they are gathering data. The hope is that that information will provide a large body of information.

The other part of the recommendation that we received from CFSAC in 2012 asked for research definitions to be developed. So, the NIH has sort of taken that, and thought about it in different ways. So they are going to develop a research [definition?] going forward, and they are going to provide a thorough review of existing case definitions. So there is going to be some overlap.

Question – Dr. Lily Chu: Patients have had some questions about what type of patients this case definition will apply to. So, I am thinking about a patient coming in to see a typical, average doctor; how do they go about diagnosing CFS? That is my framework. So, I want to make sure that that is the same framework HHS is thinking about.

My second question concerns the different case definitions that are listed - that’s a starting point. But if we have points that we want to add to that case definition to make a new case definition that the panel believes to better represent patients, then that’s what we should be aiming for, not just the existing definitions. Is that right?

Answer: It’s whatever you decide. It could be the existing one, it could be the 2003 [CCC] if you all decide that’s a very good definition. If you decide you like the 2003, or something else but you want to tweak it a little or come up with whole new measures, because the 2003 is eleven years old. So, we defer to the expertise that is assembled around the table; we defer to you.

But, again, to go back to the first point, I am hoping that this will be something that can be taught to many, many providers, so that when they come in they can put it on their differential diagnosis, so they can say this is what people have to they can get on the phone and ask an expert about it. It’s what you do when you don’t see something commonly, but you know you have to make a good diagnosis.

Question – Nancy Klimas: I’m trying to be clear because I actually serve on two panels, the NIH working group and this one as well. This group is looking for a clinical definition – as in the practice of medicine.

Answer: This could be for researchers, but this is also for the doctor or the nurse practitioner or the PA, or other health care providers that somebody walks in and it’s for them to help this person who has this diagnosis that they are not quite sure what it is. Research may come out of this. But this is really for helping patients get the kind of care they need.

Question – Nancy Klimas: In a clinical sense sub-grouping would also be addressed.

Answer: Sub-grouping is good.

Question – Betsy Keller: I have a question about the charge of ultimately creating the set of criteria that will help with the diagnosis. That’s part of the statement of task that you just described to us. Can you explain why HHS stopped there, when you have just acknowledged that it’s very difficult for patients to find providers for the illness? Are there any plans for HHS to provide a website so patients can find qualified providers to treat the disease?

Answer: I think that the government does not endorse specific providers. When we have the report we will hopefully work with the big organizations, but we don’t have the product that we can push out right now. CFSAC has a committee that is looking at educating primary care providers. A lot of that is outside the government.

Question – Betsy Keller: I am concerned about getting this information in some form that is accessible to the patients, because we’re way behind the eight ball.

Answer: Right, and so that’s task four – for you all to help us. We have the advisory committee [CFSAC] that can help us with that as well.

Question – Nancy Klimas: Task three is to address the name. There has been a tremendous amount of public discussion on this that the CFSAC minutes would reflect. In your mind’s eye is there a role for additional public input?

Answer: Absolutely.

Question – Nancy Klimas: In my mind I split it into two things. We’ll hear again and again about the name being harmful. What we need from the committee is constructive advice on how to go forward. Not to spend many hours discussing what is out there, but where do we go from here? And as a committee member, I would welcome public input.

Answer: I think that we really look to the stakeholder advocacy community for suggestions on that task.

Question – Nancy Klimas: I don’t understand the mechanism of how that would happen. Today we have this public testimony, and I am sure it will be very useful. Is there some way to have to input of the public as the process goes on about various elements?

Answer – Carmen Mundaca: We have established an email, and we have delivered comments to you. This email will be open throughout the process, and there is no deadline for submitting. We will have that as a regular means of communication with the public. The email is: mecfs@nas.edu 

Answer – Lily Chu: We are going to talk further about how public input will be gathered and put into the final documents.

Dr. Clayton: I confess that if we get through federal presentations sooner than anticipated that gives us time for public input and I’m not too sad about that.

[End of Part 1]

This article first appeared on ProHealth.