The Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) Common Data Elements (CDE) Working Group and the National Institute of Neurological Disorders and Stroke [NINDS]/Centers for Disease Control and Prevention (CDC) CDE Team released their draft version of the ME/CFS CDEs for public review on 12/15/2017.
Public comments are to be submitted by 1/31/2018.
The general NINDS CDE section explains the purpose of the CDEs as follows:
“Many of the CDEs will overlap across study types, which allows for comparisons and meta-analysis across studies. Consistency of the data elements and the CDE formats is kept in order to ensure the ability to transfer critical medical information electronically from one center to another. This consistency also allows for continuity across different disease areas. The goals of the NINDS CDE initiative are to increase the efficiency and effectiveness of clinical research studies and clinical treatment, increase data quality, facilitate data sharing, and help educate new clinical investigators.”
MEadvocacy.org has vigorously advocated for the adoption and use of criteria created by ME experts, like the International Consensus Criteria (ICC), for selecting individuals for research. The aspired goal is to make sure the cohorts being studied include patients who suffer from the same disease (ME) - in exclusion of those suffering from other conditions or idiopathic fatigue.
Broad criteria created by the CDC like the 1994 Fukuda or 2015 IOM do not ensure patient selection will exclude those who suffer from other fatigue-inducing illnesses. (as shown by Leonard Jason and Frank Twisk published works)
An expectation that the common data elements (CDEs) would be based on clearly defined patient populations does not appear to be met by the current CDE draft because NIH/CDC does not require a specific research criterion to be used for all the federally funded studies. Additionally, the CDEs are too vague in their description of post-exertional malaise (PEM) thus risking inclusion of patients with other diseases in studies for ME.
MEadvocacy.org represents patients who fit the experts’ criteria such as the International Consensus Criteria (ICC) for myalgic encephalomyelitis - including the severely ill. In reviewing the proposed common data elements, they fail to accurately select a true ME population. The conflation of persons with ME (pwME-ICC) with persons suffering from conditions with similar symptoms such as CFS, fibromyalgia, postural orthostatic tachycardia syndrome (POTS), Ehlers-Danlos syndromes (EDS), depression or idiopathic fatigue will confuse study results which will only cloud and continue to stunt scientific advancements of the disease ME.
Historically, ME as defined by the ICC has been buried under CDC overly broad definitions such as Holmes, Reeves, Fukuda and most recently IOM. Additionally, the US government health agencies have refused to adopt definitions created by actual ME experts with extensive hands-on experience in treating and researching the disease. This government refusal to recognize ME has caused a lack of research in the exclusive #pwME-ICC (#MEICC) population. The proposed CDEs further aggravate the problem, because they leave it up to the researcher to pick any research criteria of their choosing. Thus, the ME population will again not be clearly identifiable within the broader CFS patient population.
In their draft, the CDE working group for PEM acknowledges they neglected the severe population. They state: “While there is little formal research on subtypes of ME/CFS as it pertains to PEM, severely impaired patients may experience PEM with significantly smaller levels of exertion. Recommendations made by this working group may need to be modified or adapted for this group.”
CDEs Do Not Replace Case Definition
NINDS and CDC do not recommend which research definitions federally funded ME researchers should use. In their CDE description, they state:
“Researchers conducting the studies using CDEs will determine the case definition and enrollment criteria that best fit their research objectives. The CDEs are methods of collecting data in a standardized manner… The intention is that CDEs will be applicable independent of the research case definition.”
This is an extremely troubling statement from NIH/CDC!
They are advising federally granted researchers to choose whichever enrollment criteria they wish to use. They state: “whichever criteria best fit their research objectives” - so if, for example, they want to show GET is good for pwME, that’s fine - just use the inadequate Reeve’s criteria!
The CDEs are meant to serve as a method of collecting unified data in a standardized manner, independent of criteria. The real issue here is if the data is taken from cohorts that do not suffer from ME, what disease is NIH/CDC looking at with their CDEs?
The CDE PEM Draft
Since PEM (or more accurately, PENE) is the hallmark and distinctive symptom of an ME diagnosis, great care should be taken in its description and definition. Yet, the current CDEs define PEM as a positive finding of only 1 of the following 5 questions taken from the DSQ questionnaire. (with a moderate severity and frequency)
- Dead, heavy feeling after starting to exercise
- Next day soreness or fatigue after non-strenuous, everyday activities
- Mentally tired after the slightest effort
- Minimum exercise makes you physically tired
- Physically drained or sick after mild activity
Any one of these questions can be applied to many other illnesses or basically be a result of deconditioning. They do not define PEM per the Canadian Consensus Criteria (CCC) nor Post- Exertional Neuroimmune Exhaustion (PENE), in the International Consensus Criteria (ICC) which require a much more significant impact on daily living and is a unique experience seen in ME.
The ICC was created by ME experts for diagnostic and research purposes. The IC Primer lists many recommendations for biological tests to be taken to confirm a proper ME diagnosis - not simply relying on subjective questionnaires.
The goals of the CDEs are admirable but, it can only be effective if the data being looked at and shared are actually based on the distinct disease being discussed. For example, if the CDEs were used to compare ME with major depressive disorders (MDD) and the ME cohort mistakenly included those suffering from MDD and not ME - it will skew the picture.
MEadvocacy’s concern is that the current NINDS/CDC CDEs draft will not accomplish its intent because:
they do not specify the research criteria to be used across all federally funded studies (i.e the ICC)
they rely on subjective questionnaires most of which were not created for the distinct disease ME
- PEM/PENE, the hallmark feature of ME, is very poorly defined.
Therefore, MEadvocacy recommends that:
- All federally funded researchers use the ICC which was created by ME experts for diagnostic and research purposes.
- New questionnaires be designed which are strictly created with ME patients in mind.
- PENE be strictly defined as per ICC in order to weed out those who suffer from fatiguing conditions - not ME
What You Can Do
If you agree with MEadvocacy’s recommendations, you can send an email with MEadvocacy’s blog (copy and paste or a link), stating that you endorse MEadvocacy’s recommendations to NINDSCDE@emmes.com (due date is 1/31/2018)
* Please note - CDE instructions state: After January 31, 2018, feedback can still be sent at any time through the website or by emailing NINDSCDE@emmes.com. Proposed changes will be brought to the oversight committee (OC) for review.