From Jennie Spotila's Occupy CFS blog, 29 March 2015.
You may remember This Week in Virology (TWiV) from their XMRV coverage several years ago. I’ve remained an avid listener of the show, simply because it is such a great ongoing conversation about science. And TWiV has continued its coverage of ME/CFS, most recently in discussing Dr. Ian Lipkin’s latest paper and the IOM report.
I was fortunate enough to meet the show’s host, Dr. Vincent Racaniello, in 2011, and have corresponded with him since. So after TWiV discussed Lipkin’s paper, I wrote in, and then they read and discussed my letter. For ease of listening and sharing, I’m gathering the whole sequence here. UPDATED March 30, 2015 with transcript and follow up letter.
TWiV Episode 329: discussion of the Lipkin paper and the IOM report occurs at approximately between 11:15 and 25:10 in the episode.
I wrote the following lengthy email to the show afterwards:
Dear Vincent and friends,
I’m sure you have received other emails regarding your discussion on Episode 329 about ME/CFS and Dr. Lipkin’s latest paper, but as a long time fan of the show who has lived with ME/CFS for more than twenty years, I could not resist chiming in with my two cents.
Dickson commented that patients with CFS are self-diagnosed, going to doctors who are prone to believing their symptoms. For the vast majority of patients, this is not true. The IOM report said that 800,000 to 2.5 million Americans may have this disease, but that 80-90% of them are undiagnosed. I have never met an ME/CFS patient who easily found doctors to believe them. Every single one of us has been dismissed or even ridiculed by one or more doctors. We’ve been told that we are lazy malingerers, that we have psychological problems, or that we just need to reduce stress. It is simply not the case that people “feel lousy,” as Dickson put it, go to the doctor, and come home with the CFS label.
As Alan noted, there is no doubt that the controversy over case definition has hindered progress in this field. Dickson was right to ask how many diseases are really included in an ME/CFS population, and he pointed out that absent an objective test, conclusions are preliminary. The IOM made recommendations to address that. First, the SEID criteria are no longer a diagnosis of exclusion. If a patient fits the criteria, then he or she should be diagnosed with the disease. The onus is put on physicians to make differential diagnoses, and physician education will be of critical importance if these recommendations are to succeed. Second, the IOM explicitly stated that their recommendations were based on current evidence and should be revisited in no more than five years. They anticipated that new evidence, such as Dr. Lipkin’s paper or the studies demonstrating brain inflammation, would change our understanding of the disease’s pathogenesis and perhaps validate biomarkers for diagnosis.
The Lancet editorial stated that the IOM was heavily and negatively lobbied for undertaking this study, but failed to acknowledge that there were legitimate scientific and political reasons for this opposition. First, at the time this study commenced, the IOM had never produced a disease case definition. There was no guarantee how many ME/CFS experts would be appointed to the panel, and the community feared this “definition by committee” would be created by people who knew nothing about the disease. Second, the ME/CFS field has upwards of twenty proposed definitions, so spending one million dollars to create the twenty-first seemed like a questionable strategy. Third, given the IOM’s prestige, we knew that we would likely be stuck with the results for a long time, and if the case definition was flawed then this would have long-term negative consequences. Fourth, the Department of Health and Human Services pursued this IOM study in secret. No ME/CFS experts were consulted about it, and the Department’s own CFS Advisory Committee was not informed of the plan. ME/CFS advocates discovered the contract by accident, and HHS completed the deal over the vociferous objections of advocates and researchers alike.
All that being said, it is not true that the entire ME/CFS community continued to negatively lobby the IOM after the study began. Certainly, some advocates did so and have continued to object (and not without reason). But many advocates, myself included, gave presentations to the IOM panel and provided written input. In fact, the public access file for the study, which contains all of the written materials submitted to the panel, totals over 5,500 pages. In my opinion, there is ample evidence that the panel listened to this public input.
The Lancet editorial also mentioned the PACE trial and the Cochrane review on exercise therapy. This brings me back to Dickson’s question about how many diseases are lumped together in the CFS category. Depending on the case definition being used, it could be more than one. The Oxford CFS definition, in particular, is overly broad, as it requires only six months of subjective fatigue. A patient population defined by only that subjective symptom is larger and less precise than a more narrow definition like the IOM SEID or the Canadian Consensus Criteria. In fact, a recent report from an NIH panel and a systematic evidence review by the Agency for Healthcare Research and Quality both recommend that the Oxford definition be retired because results from studies of this broad group cannot be accurately applied to the more specific ME/CFS group. This is important, because most of the studies in the Cochrane review on exercise in CFS were Oxford studies. The PACE trial also used the Oxford definition to enroll subjects. In fact, most of the studies purported to show that exercise is beneficial for patients with CFS have used the Oxford definition. In contrast, there is a growing body of work, such as this paper by Keller et al, showing that ME/CFS patients (defined more narrowly than Oxford) have measurable abnormal responses to exertion. The case definitions are not interchangeable, and research results should not be extrapolated from one group to the other.
Patients with ME/CFS do not just “feel lousy.” This is a serious disease with serious consequences. I have been housebound, and sometimes bedbound, for more than twenty years. The most severely ill patients suffer from numerous neurological symptoms, including ataxia and allodynia. This disease costs our economy billions every year in lost productivity, and yet NIH spends only $5 million a year on research. Even Dr. Lipkin has stated publicly that his grant application was denied after a reviewer claimed biomedical research was unnecessary because ME/CFS is a psychological disorder. The IOM report is an exceptionally well-referenced document, and states unequivocally that this is not a psychological disorder. It is my fervent hope that we can leave that controversy behind, and move forward with rigorous, well-funded research into carefully characterized patients. This is the only way we will find treatments and get our lives back.
Thank you for continuing the conversation about this disease. Vincent, I also send you best wishes for your son’s recovery.
On TWiV Episode 330, Dr. Dickson Despommier read my email and it was discussed at some length by the group. The discussion runs from 12:57 to 29:10, and the wonderful Russell Fleming has kindly provided me with a transcript of the discussion. As you can see, a few additional questions were raised, so I sent this follow-up email to TWiV:
Don’t feel obligated to read this email on air because I’m not trying to monopolize discussion, but I want to answer a couple questions raised during the Episode 330 discussion of my letter re: ME/CFS.
Rich asked if I had a role in a CFS organization. When Vincent and I met, I was finishing my term as a member of the Board of Directors of the CFIDS Association (now called the Solve ME/CFS Initiative). Since the end of my term in 2011, I have not had a role in that or any other ME/CFS organization. Instead, I’ve focused my efforts on my blog, Occupy CFS, and on individual advocacy efforts.
Alan commented that giving more money to ME/CFS research means taking money away from another disease. It’s not that straightforward. I have documented that in FY2014, 62% of disease categories listed by NIH received an INCREASE in funding over FY2013. ME/CFS was one of only 17 disease categories out of 244 that received funding essentially flat with 2013. The remaining 31% of categories saw funding decrease. HHS employees frequently tell us there is no money, but that can’t be true if the majority of disease categories are seeing an increase in their funding. Since Alan mentioned HIV/AIDS, I checked their funding and found that HIV/AIDS research increased by $80 million from 2013 to 2014, and is projected to receive a $22 million increase in 2015. Couldn’t NIH reduce that to an increase of $15 million, and spend the other $7 million on ME/CFS? Both categories still go up, and ME/CFS’s budget would more than double. Obviously, there is money – it’s just not coming ME/CFS’s way.
There are several other things that could be done to increase ME/CFS research funding at NIH. ME/CFS does not have an Institute home. Instead, research is coordinated – with no budget and no dedicated program officer – through the Office of Research on Women’s Health. Setting aside the fact that men get ME/CFS too, this arrangement essentially means that ME/CFS research has to shake a tin can and try to get an Institute’s attention and interest. Moving the portfolio to one of the Institutes might help this situation. Second, NIH could issue an RFA with set aside funds for research into a high priority area such as large-scale validation of biomarkers, rigorous prevalence and epidemiology studies (to get at the “true case” question), and/or large -omics studies. NIH last issued an RFA for this disease in 2006, but has refused every subsequent recommendation and request to repeat the process.
Kathy asked how many non-ME/CFS experts served on the IOM panel. While the community feared there would be few or no experts, in the end the IOM appointed 8 members who had clinical, research, or personal experience with ME/CFS and 7 members who had none. The vast majority of the report’s peer reviewers had ME/CFS experience, as did every guest invited to present to the IOM panel at its public meetings. There were a diversity of views represented among those groups, but that is as it should be.
Alan rightly questioned my statement that HHS pursued this study in secret. I have accumulated documentation through personal communications with HHS employees and members of the CFS Advisory Committee, and through FOIA requests. HHS began working on the contract in early 2013, and employees were explicitly instructed not to share any information with the Advisory Committee or the public. HHS did publish notice of its intent to sole source the contract on a federal contract site on August 27, 2013, but made no announcement and never published the information in the Federal Register. Advocates discovered the posting and began an opposition effort, and the sole source contract was cancelled on September 4, 2013. No additional details were made public, despite repeated requests to HHS, until the September 23rd announcement of the contract signing as a Task Order under NIH’s standing contract with the National Academy, thus avoiding the requirement to give public notice of the specifics in advance. I made an attempt at summarizing the timeline, and why this approach was so disastrous for the HHS-community relationship, in this post just after the contract was signed.
On a more personal level, the most telling part of your discussion was Vincent’s statement that he was advised to avoid the CFS label for his son because then no doctor would do anything to help him. This happened to me. One infectious disease expert I consulted early in my illness said, “You have CFS but there’s nothing we can do for you. We can arrange for counseling to help you live with it. Maybe you’ll get better in five years.” In fact, there are things patients can do to help alleviate or cope with symptoms, but there is no systematic way to access those treatments, and almost no ME/CFS specialists to consult. The conservative estimate is 800,000 people have ME/CFS (using the 1994 Fukuda case definition), and 80% have not been diagnosed. That means 640,000 have absolutely no help and nowhere to go because they don’t even know what they have. The other 160,000 who do have the label may have to wait months or years to access a specialist, or they give up and turn to alternative medicine, or their non-specialist doctors prescribe unproven therapies like antidepressants and exercise which could be dangerous to them. We cannot allow this to continue.
My thanks to all of you for covering this issue. Public dialogue like yours can only help.
My sincere thanks to Dr. Vincent Racaniello and the TWiV hosts for continuing the conversation about ME/CFS. They do great work. Listen to the podcast!
JENNIE SPOTILA, who has had CFS for the past 20 years, is a former chairman of The CFIDS Association of America and has testified on many occasions before the US Federal Government's CFS Advisory Committee in Washington. She tweets as @knittahknits