First of all, thank you so much to everyone who responded to the #MEAction survey to make our response to the NIH as comprehensive and representative as possible!
On May 24th, the National Institutes of Health (NIH) released the document Request for Information: Soliciting Input for New Research Strategies for Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS). A wide variety of patients, scientists, advocates and organizations formulated and sent their responses. #MEAction releasing a survey in which over 1800 stakeholders participated. It can be found here. #MEAction also took the opportunity to gauge patient responses to a variety of important issues, and to gather demographic and other important data.
These are the results.
A survey was created through Google surveys by #MEAction containing 109 questions allowing respondents to rate the importance of each item 1-5, in which items marked ‘1’ are least important to research and items marked ‘5’ are most important to research. The survey provided a neutral summary of the potential significance of each item, and respondents were encouraged to skip items they did not feel they understood rather than choose an ‘average’ value. An item that rated 4/5 or better was considered to be of significant concern. The survey also contained 13 questions about demographics and open-ended responses for each section, allowing respondents to add additional comments and concerns.
Responses were solicited through meaction.net via its news platform, the online community Phoenix Rising, and the social media outlets Facebook and Twitter.
Items were added to the survey at the request of community members, and respondents had the option to skip items they did not understand, which creates some heterogeneity in the number of responses per item.
Survey questions were divided up into the following categories:
- Specific challenges to research
- Specific solutions to these challenges
- Pathogenic Triggers
- Non-pathogenic Triggers
- Additional avenues for research
Data was gathered anonymously, marked only with time completed. Data was scrubbed to eliminate accidental double-submissions and to more accurately calculate numerical values, e.g. if a respondent wrote that they contracted ME in “1998 or 1999” the figure 1998.5 was substituted in order to estimate average time of onset for all respondents.
Respondents self-identified as 92% white, 82% female, and were generally of European descent.
Limitations of the survey include that it was only provided in English, the limited period of time allotted to respond, which may have preferentially selected for retired or disabled respondents. The act of completing a 100+-question survey may have been untenable for severe and very severe patients. Here ‘severe’ is defined as househound, and ‘very severe’ is defined as bedbound. This creates the possibility that respondents are generally healthier than the referent population.
In order to gather information from individuals with a variety of perspectives, clinicians and caregivers were also encouraged to respond.
Specific Challenges / Barriers and Potential Solutions in ME Research
The greatest barriers to research in ME and potential solutions are rated in the figures below.
“Lack of money and stigma/poor understanding are intimately related. They create a feedback loop with lack of researchers and lack of good information. Until we push money at this in a big way, along with good information, these barriers will remain.”
“NIH needs to recognize that fishing expeditions are needed, and that well-posed single hypothesis/single outcome studies are not the only ones needed to advance ME/CFS research.”
“Because this disease has been sorely neglected for 30 years, the ME community knows more about it than the scientific community as a whole, and NIH in particular. You need to work with specialists OUTSIDE NIH, researchers such as Ron Davis at Stanford, clinician/researchers such as Dan Peterson of Incline Village, Jose Montoya of Stanford, and John Chia in L.A. You need to work with patients who have a scholarly background. Finally, there has been a CFS Advisory Committee to HHS since 2003 (before that there was a CFS Coordinating Committee). The public members on CFSAC have worked hard to produce a set of recommendations every year, and every year it has been ignored. It would be a good first start to take those recommendations seriously.”
Research Definitions and Name of the Illness
The #MEAction survey revealed that patients overwhelmingly prefer the Canadian Consensus Criteria (CCC) definition of myalgic encephalomyelitis (62%) or the International Consensus Criteria (ICC) (45%) over other proposed criteria, including the IOM’s SEID, Ramsay’s criteria, Fukuda and Oxford. The Fukuda and Oxford criteria were considered the least favorable criteria by respondents, at 12% and 11% approval rates, respectively.
Approximately 30% of respondents agreed that patients should meet multiple different criteria; however, respondents considered it extremely important that NIH studies use one, consistent definition in clinical trials (4.45 / 5). This reflects the idea that, while multiple definitions may be useful clinically, research definitions should be more stringent and consistent, so that it is clear which illness is being studied, and to what patient population data may apply.
Although the study of subtypes, clusters, and the differentiation between ME and CFS definitions was not listed as a potential research priority on the survey, enough individual comments reflected this concern that a discussion of ME subgroups emerged as a theme:
- The presence of multiple subgroups with symptom-clustering in ME
- That ME and CFS are not the same illness, and should be studied separately
- Early-onset and chronic patients should not be placed in the same cohorts (vis a vis Lipkin and Hornig’s cytokine studies)
“The fact that M.E. appears to me to be an umbrella title for half a dozen sub groups means that history of the symptoms is critical. What caused one type of M.E. would not be responsible for all sub groups so research results should be split to show which sub group responded best to which treatment. My M.E. differs from others that I know so it is important to know what results are relevant to which sub group.”
“…we should NOT hold this disease to a higher standard than all others by hunting for one, elusive biomarker or by insisting that results that are frequently abnormal and may be sensitive but not specific are useless. We could probably get very close to diagnosis with panel of 3-5 commercially available tests: e.g., natural killer cell function (properly prepared and executed), sed rate, cognitive testing and where available, an exercise test. It’s also very important that patients be tested for individual pathogens to help determine what antivirals/antibiotics, etc. may be of some benefit to them.”
Click here to read part 2 of 3, and thank you again for making it possible for your community to send such a data-driven message to the NIH!