On May 12 – International Awareness Day for ME/CFS/FM/MCS/etc – Phoenix Rising joined with 8 other US ME/CFS patient organizations and 26 independent patient advocates to call on the Department of Health and Human Services (DHHS) to finally fix the problem of the many and diverse case definitions associated with our disease. In a letter to Secretary Sebelius, Dr Howard Koh, Dr Thomas Frieden and Dr Francis Collins, we explained our concerns about the current definition activities of the DHHS in relation to “Chronic Fatigue Syndrome”, and listed the steps we believe must be taken to rectify the situation.
You can read our letter to the DHHS here.
The signatories on the letter are:
Chronic Fatigue Syndrome, Fibromyalgia and Chemical Sensitivity Coalition of Chicago, CFS/Fibromyalgia Organization of Georgia, Inc., MAME (Mothers Against Myalgic Encephalomyelitis), PANDORA (a.k.a. CFS Solutions of West Michigan), Phoenix Rising, The Fibromyalgia-ME/CFS Support Center, Inc., Rocky Mountain CFS/ME and FM Association, Speak Up About ME, Wisconsin ME/CFS Association, Inc., Bobbi Ausubel, Rich Carson, Lori Chapo-Kroger, R.N., Kati Debelic, R.N., Mary Dimmock, Pat Fero, MEPD, Joan Grobstein, M.D., Jean Harrison, Eileen Holderman, Suzan Jackson, Jill Justiss, Mindy Kitei, Michele Krisko, Denise Lopez-Majano, Mike Munoz, Matina Nicolson, Donna Pearson, Leela Play, Justin Reilly, J.D., Mary Schweitzer, Ph.D., Meghan Shannon MS MFT, Marly Silverman, Rivka Solomon, Tamara Staples, Charlotte von Salis, J.D., Michael Walzer.
For those of you who wish to sign this letter and become a part of this important initiative, we will provide a mechanism to do that within a few weeks and will send out additional information at that time.
Why have we written this letter?
Of all the issues that we face today, the one issue that has created the most problems is the diverse case definitions associated with our disease. This single issue has severely impacted research, drug development and clinical care and misled the medical community on the very nature of this devastating disease, causing many doctors to not believe that their patients are really sick. Until this issue is addressed, patients will continue to pay the price. This must stop now.
Today, the CDC states that there are at least 5 different definitions for “CFS”. Three of these definitions – the Canadian Consensus Criteria, the ME International Consensus Criteria and the Pediatric Criteria – require hallmark criteria like PEM/PENE and neurological, immunological and energy production impairments. Unfortunately, two of the most commonly used definitions, Fukuda and Oxford, do not require these hallmark criteria. In fact, Oxford only requires 6 months of disabling fatigue – no other symptom – and allows primary psychiatric disorder.
The result? Myalgic encephalomyelitis, the disease seen in outbreaks throughout the twentieth century and recognized by the World Health Organization in 1969, has disappeared, and in its place we are left with “CFS”, an amorphous umbrella of unrelated fatiguing conditions including, according to the literature, depression, deconditioning, medically unexplained chronic fatigue, and for some researchers and clinicians, fatigue due to “excessive rest” or “false illness beliefs”. In clinical practice, the diagnosis of CFS is given to a heterogeneous mix of patients – those with ME, those with the varied fatiguing conditions listed above, and those who were misdiagnosed or whose doctors use the diagnosis of CFS as a catch-all for unexplained fatigue. And in 2012, an American Family Physician article proclaimed that Oxford and Fukuda are the appropriate definitions for “CFS” and further stated: “[CFS] patients with poor social adjustment, a strong belief in an organic cause for fatigue, or some sort of sickness benefit (i.e., financial incentive) tend to have worse responses to [cognitive behavioral] therapy.”
Exactly what disease are we talking about here?
Patients have paid dearly for the proliferation of these overly broad and non-specific definitions – bedbound or homebound, unable to work or take care of their families, suffering for 10, 20, 30 or more years from the myriad symptoms that plague their bodies, unable to get adequate medical care and ultimately more likely to die prematurely from cancer, cardiovascular disease and suicide.
As Dr. Carruthers stated in the ME International Consensus Criteria, “Research on other fatiguing illnesses, such as cancer and multiple sclerosis, is done on patients who have those diseases. There is a current, urgent need for ME research using patients who actually have ME.” We must have a disease appropriate definition for ME that is separate and distinct from all the other unrelated conditions encompassed by the overly broad, fatigue-focused “CFS” definitions.
What are we asking for?
Our letter to the DHHS asks them to:
- Adopt a disease-appropriate case definition for ME now, utilizing the Canadian Consensus Criteria as recommended by DHHS’ own advisory committee CFSAC, and train doctors with appropriate medical guidance.
- Stop using the terms “CFS” and “Chronic Fatigue Syndrome” along with the non-specific definitions like Fukuda and Oxford and the medical education material based on these definitions.
- Manage the adoption of the Canadian Consensus Criteria to ensure that insurance and disability do not lapse and that no patients fall through the cracks.
- Fully engage ME stakeholders in the planning and execution of the adoption of the Canadian Consensus Criteria.
Is this the right thing to do?
You may ask whether we really know enough about the disease or whether we need more study before we change definitions. Certainly, with more study, we can better operationalize the definition and validate biomarkers to make patient diagnostics easier. But in the meantime, we know that PEM/PENE is a hallmark symptom that reflects a distinctive biological pathology and we must utilize a disease definition that requires that symptom.
Some of you may prefer the ME International Consensus Criteria over the Canadian Consensus Criteria. The ME-ICC certainly has some excellent features. But practically, the Canadian Consensus Criteria has been used clinically and in research for a decade. Studies have been done with it. The U.S. government has posted the IACFS/ME Primer, based on the Canadian Consensus Criteria, on DHHS’ Guidelines.Gov. This is more likely to be acceptable to DHHS and is a reasonable first step, especially when considered against the alternative of continuing to use Fukuda while more study is done.
What about dropping the name “CFS”? You may be concerned that this means we will lose the literature base that has provided insights into the pathology of ME. Admittedly, some of the best articles used the term “CFS”. And so do some of the worst. The point is that the literature base is a mess because multiple diverse and unrelated definitions have inexplicably been allowed to use the very same name for years. We all should stop using the term “CFS” because it no longer has any real meaning.
Finally, what about the name ME? Does it really describe the disease? Is there a better name? That is a question that science will need to decide over time, something that has happened in many other diseases. But what is clear is that “chronic fatigue syndrome” will never be an appropriate name and should never have been established as the alternative or synonym for ME.
Patients have borne the brunt of the failure to address the definitional issues for the last thirty years. We cannot wait for more study to finally stop the harm being done to patients, especially given that more study with non-specific definitions will only perpetuate the problem. The time to address this problem is now.
Questions and Answers
We realize that patients, carers and advocates may have a number of questions about this initiative, and we hope that the following questions and answers will address any concerns you may have.
1. We can not abandon the patients that have been incorrectly given a “CFS” diagnosis.
This is very true. It is critical that implementation of this change is carefully managed so that these patients are re-evaluated and given a correct diagnosis. If unexplained conditions remain, it will be necessary to perform the studies needed to understand these conditions and establish more appropriate names and definitions.
2. We can not afford to have our disability or insurance impacted.
Yes, this is very important. It will be important to have a carefully thought out implementation plan that manages this to ensure that patients do not lose disability or insurance benefits.
3. The vast majority of the 6000 articles in the literature use the name “CFS”, not “ME. If we stop using the name “CFS”, we will lose all that literature.
Currently, when the search term myalgic encephalomyelitis is used, the CFS literature is returned. This will not change. But that literature base contains both articles relevant to ME and also a significant number of articles about “CFS” and child abuse, false illness beliefs, deconditioning, etc. This creates significant confusion for anyone trying to use that literature. For that reason, the non-specific term “CFS” should be abandoned by the U.S. and more specific terms like ME used going forward.
4. We have more important issues to deal with such as funding, and attracting new doctors and researchers.
It is critical that we have more funding but if we don’t fix the definition issue first, we will continue to study the wrong disease and have progress impeded by poor definitions. The resultant confusion will make it difficult to attract young researchers and doctors who will not see career opportunity in “CFS”.
5. Research centers have recently been established and if we stop using the name “CFS” we will confuse our donors.
It is true that a number of research institutes have recently been opened and some of them use the term “”CFS” or even “CF”. But the donors to these institutes today have a personal connection to the disease. They will continue to fund. Attracting additional funders, however, will be negatively impacted by the confusion around the disease. The sooner we can resolve this issue, the better in the long run.
6. CFS biobanks have been established using Fukuda and we don’t want to lose those samples.
The biobanks that have only been characterized by the Fukuda definition could contain a mix of patients with the hallmark criteria of ME and those who do not have these hallmark criteria. Using these mixed samples will continue to confound research. It is important that we have a well-characterized set of samples in the biobank and know which samples are from ME patients.
7. ME may not be the right name. Shouldn’t we wait for the science to figure out what the right name is?
It is possible that with further study, we will determine a better name than ME and it will naturally evolve. But ME, adopted by the World Health Organization in 1969, is the best placeholder until that time and avoids the serious issues caused by the use of the term “CFS”.
8. The best course is to tighten up the “CFS” definition, not get rid of it. Then we can keep the literature base, the biobanks, etc.
There are two problems with this approach. First is the long history of the term “CFS”, which is non-specific and now widely associated with diverse conditions, especially including psychiatric issues. This has severely tainted the term and made it clinically meaningless. Second, the term “CFS” is used for those studying patients that meet Oxford criteria (essentially chronic fatigue) and we have little control over that continued usage.
9. Lenny Jason recently published a paper that reports that the ME-ICC and the Canadian Consensus Criteria include more psychiatric co-morbidities than the Fukuda and recommends that more study be done. Does that mean we should wait to recommend any criteria until then?
- Dr. Jason’s paper did find that the ME-ICC found more psychiatric co-morbidity than Fukuda. But Dr. Jason acknowledged the need for more study because this one used a questionnaire designed for Fukuda CFS, and that they were unable to assess one of the key ME-ICC criteria because data on this criteria was not available. Further, the study did not look at homebound or bedbound patients.
- But what is also significant in Dr. Jason’s study is that ME-ICC identified a much tighter group of patients (39 compared to 113 for Fukuda) with more of the functional impairments and physical, mental and cognitive problems seen in ME-ICC patients than in those meeting the Fukuda criteria.
- Clearly additional study is needed to operationalize the definition and to improve how it characterizes the disease, especially around subtypes. But continuing to use the 19-year-old consensus-driven Fukuda definition – which is also not operationalized and does not describe subtypes – in the meantime is not going to advance that knowledge and will only continue to hurt patients.
- The Canadian Consensus Criteria has been used clinically and in research for over 10 years and better represents the disease. Using the CCC now will allow us to begin to make forward progress in research and identifying treatments, and begin to address the disbelief in the medical community.
10. Is this the same thing as the Name Change initiative?
No. This is first and foremost about the definition being used – adopting a definition that effectively describes the disease and stopping the use of the definition – and name – that have created so much confusion and so many problems.
11. Why CCC and not ME-ICC?
The CCC has been used clinically and in a number of studies, providing the experiential foundation for its use. It is expected that as additional data is obtained, this definition will evolve. This must be done in partnership with the experts who developed the ME-ICC and the CCC.
We hope and believe that this initiative will be welcomed by the majority of the patient community, and we hope that the questions above have addressed any concerns. Of course there is always room for debate over details, but very few if any of us are happy with the existing definitional mess, and this letter represents a consensus amongst 9 patient organizations and 26 independent advocates on the best path towards change. As such, we encourage the community to get behind this initiative and seize this opportunity to resolve the problem of the broken case definitions used for our disease.