• Welcome to Phoenix Rising!

    Created in 2008, Phoenix Rising is the largest and oldest forum dedicated to furthering the understanding of and finding treatments for complex chronic illnesses such as chronic fatigue syndrome (ME/CFS), fibromyalgia (FM), long COVID, postural orthostatic tachycardia syndrome (POTS), mast cell activation syndrome (MCAS), and allied diseases.

    To become a member, simply click the Register button at the top right.

How Jason 'operationalised' the CCC and compared it to other criteria - 2012

Firestormm

Senior Member
Messages
5,055
Location
Cornwall England
Contrasting Case Definitions for Chronic Fatigue Syndrome, Myalgic Encephalomyelitis/Chronic Fatigue Syndrome and Myalgic Encephalomyelitis

Note: Link gives access to full paper

One way in which the CCC was operationalised - something that the IOM has said a criteria is in need of I recall - was by Jason. He applied a measure of severity and frequency. Currently the 2003 CCC and other criteria, merely ask if you have a symptom, not how often you have it occur...

To operationalize the Canadian clinical ME/CFS case definition, we used data from the CFS Questionnaire to assess how often the person had experienced the symptom (fatigue, postexertional malaise, etc.) over the past 6 months using the following scale: 0 = never, 1 = seldom, 2 = often or usually, 3 = always.

To be counted as “persisted or recurred,” the individual had to have a frequency score of 2 or higher (See Jason, Evans et al., 2010). In addition, it is important that fatigue and the other core symptoms were either moderate or severe on a 100-point scale.

This “severity index” has not been well defined in previous ME/CFS criteria. We now specified that existing symptoms need to be rated at a 50 (as moderate) or higher to meet criteria. Both ratings of frequency and severity, therefore, had to be satisfied in order to qualify for each individual symptom.

I don't have the time or really the inclination to review this paper properly. Indeed I probably read it when it was published - but have forgotten the salient points now.

Here's a few highlights from a quick read through. The issue of selecting an existing criteria is clearly not a straightforward one, and even Fukuda appears to have some benefits.

I'm not sure if Jason has published the study he refers to in my 'conclusion' at the bottom, but if he has then it might also be something the IOM should consider.

Incidentally, the Statement of Work from HHS, refers to a 'Revised Canadian ME/CFS Definition' published in 2010 - and not something referred to by the experts - as another criteria they want or feel is in need of consideration as part of the IOM remit.

This is again another paper from Jason (thanks @Ember for finding where it had been referenced :)) but I haven't yet read it or considered what impact it might have on the 2003 Canadian Criteria - even if I was so inclined.

And then of course you have the NICE criteria encapsulated in it's 2007 Clinical Guideline and the ME ICC published in 2011. Both of which are also up for consideration by the IOM committee.

Anyways...

Discussion

...Table 2 indicates that both the ME/CFS and the ME groups had more impairment than the comparison Fukuda CFS groups. In addition, the most impairment on physical issues involved the ME group.

We did not directly contrast the ME/CFS and ME groups, but in general, impairment was significant for both groups. In addition, the ME group appears to have less mental health issues than the ME/CFS group.

It does appear that requiring specific criteria, such as what occurs in the ME and ME/CFS case definitions, does select individuals with more functional disabilities.

In the original Canadian ME/CFS criteria (Carruthers et al., 2003), symptoms could be rated as mild, moderate or severe, but it was unclear whether symptoms needed to meet specific frequency and severity levels to be counted.

In other words, it was possible to count just whether symptoms occurred in order to meet the ME/CFS criteria. Had just occurrence of symptoms been employed (rather than needing to meet cutoff scores for frequency and severity of symptoms) for the core ME/CFS Canadian criteria symptoms in Table 3, 105 of the individuals in the current sample (over 90%) would have met ME/CFS criteria.

However, when using the symptom frequency and severity cutoff points as specified by Jason et al. (2010), only 50% met the ME/CFS criteria. It is clear that these individuals with ME/CFS had more severe functional disability and symptoms than the Fukuda CFS {a} group....

In the present study, we found significantly higher current psychiatric comorbidity rates for those with ME/CFS versus Fukuda CFS {a} (58% vs. 20%). In the prior study, using just symptom occurrence to classify patients, Jason et al. (2004) found that the ME/CFS group had significantly lower current psychiatric rates (48%) than the Fukuda et al. (1994) CFS group (75%).

It is probable that the current study selected a more seriously impaired group of patients with ME/CFS and that their impairments were across a broad array of both physical and mental health areas. Katon and Russo (1992) have argued that a requirement of more symptoms to meet criteria could inadvertently select for individuals with psychiatric problems.

Similarly, Kroenke (2003) found similar results examining 15 variables within a fatigued sample. Upon examination of 13 of these 15 variables in our sample, we found that a greater number of symptoms was associated with increased psychiatric comorbidity as well as an ME/CFS diagnosis. It is certainly possible that the larger number of symptoms of higher frequency and severity among patients meeting the ME/CFS criteria versus Fukuda CFS {a}, accounts for the higher levels of psychiatric comorbidity and more functional
impairments on the Role Emotional and Mental Health SF-36 subscales among the ME/CFS criteria group.

In contrast, the ME criteria required four versus the seven ME/CFS symptoms to meet its case definition. Of interest, there were no significant psychiatric rate differences for the ME versus Fukuda CFS {b} groups (44% vs. 37%).

There were also no significant differences between the ME and Fukuda CFS {b} groups on the SF-36 scales measuring Role Emotional and Mental Health areas....

...But the lack of differences on the psychiatric items suggests that the ME criteria selects individuals with less psychiatric comorbidity and mental health issues than the ME/CFS group.

It is possible that sudden onset, postexertional malaise, a neurocognitive and autonomic symptom identify individuals with fewer emotional and mental health problems, but when additional symptoms are required, this selects both more physical and mental problems.


It should also be noted that in previous studies that have found more disability/symptoms causing psychiatric problems, there were confounds, with symptoms counting for psychiatric diagnoses.

In addition, there might be a number of reasons for a higher rate of psychiatric disorders associated with more symptoms and/or disability. For example, it could be more “depressing” to have more symptoms and/or disability interfering with what one can do.

Perhaps similarly, one might be more anxious about how one might be able to cope in the future.

Across medical conditions, illnesses/diseases that are more disabling and/or have more symptoms associated with them/interfere with life more are associated with higher rates of psychiatric problems.

Conclusion

The current CFS case definition of Fukuda et al. (1994) has been used internationally by researchers for over 15 years. It is possible that some patients meeting these criteria do not have core symptoms such as postexertional malaise or memory/concentration problems.

By specifying seven symptoms as with the ME/CFS criteria or by specifying four symptoms with the ME criteria, it may be possible to identify a more homogenous and impaired group of patients. The current study suggests that the ME and ME/CFS criteria might be used to identify patients with possibly more homogenous and severe symptomatology and functional impairment.

Some might conclude that the ME/CFS definition requires too many symptoms that may at the upper ends select for psychiatric disorder. Others might feel that we should just expect higher levels of a variety of both medical and psychiatric disorders if this definition is utilized. Still others might feel that the severity dimension be relaxed as in the previous ME/CFS study (Jason, Torres-Harding, Jurgens, & Helgerson, 2004) that did not require such a high level of symptom severity.

The present study suggests that the initial definitions of ME (Dowsett et al., 1994; Goudsmit et al., 2009; Ramsay, 1988) and later on the Canadian criteria of ME/CFS (Carruthers et al., 2003) appear to select a group of patients that have more severe functional impairments, and physical and cognitive symptoms.

The ME/CFS criteria appear to identify more impairments in symptoms, whereas the ME criteria appear to identify more impairment in functional status, except for emotional and mental health domains.

It would be premature to make any definitive conclusion at this time concerning which explanation is more valid. Certainly, there is a need for more research on this topic, with larger data sets. We are currently collecting more data on this issue, and we hope in the future to be able to offer more definitive interpretations of the findings reported in the current study.
 
Last edited:

peggy-sue

Senior Member
Messages
2,623
Location
Scotland
Who actually wrote this? I trust it wasn't Jason!
There is a (repeated) glaring example of really bad English in it, something which would not help anybody to take what is writtten seriously!

Quote; "In addition, the ME group appears to have less mental health issues than the ME/CFS group."

The correct wording is; "the ME group appear to have fewer mental health issues..."

And on that topic, I wish they would distinguish clearly between "mental health issues" and the clinical depression that having this damned disease can often cause.:mad:

(grumpy and nitpicking today... :p )
 
Messages
5,238
Location
Sofa, UK
And on that topic, I wish they would distinguish clearly between "mental health issues" and the clinical depression that having this damned disease can often cause.:mad:
That's my main problem with that paper, and I think it's been misunderstood by some because of that vagueness. Just describing simply "mental health issues" is too vague; if that means depression and anxiety measured in a certain way (eg HADS) then it doesn't necessarily imply anything 'psychiatric'. It's also been suggested that there may be pathological processes in severe ME that are also associated with provoking depression and anxiety, so even if there is more depression and anxiety in a cohort that doesn't necessarily mean it's a bad definition that is selecting more patients with 'only' psychiatric illness. So it's really quite complicated to interpret this paper, and although some have interpreted it to suggest that CCC and ICC have problems and may select too many psychiatric patients, I think that's much too simplistic.

Perhaps also worth noting that Jason is one of the 50 experts who signed the letter against the IOM contract and supporting immediate adoption of the CCC by HHS, so whatever the detail his conclusion seems to be pro-CCC, for now, at least.
 

Firestormm

Senior Member
Messages
5,055
Location
Cornwall England
The main reason for posting this again, but on this Forum and at this time, was the reference to 'operationalising' the criteria, which is something HHS/IOM also believe is necessary to consider in addition to agreeing a clinical criteria.

I thought reference to the actual term 'operationalising' and separately 'validation' of criteria came from HHS/IOM, but the only reference to the words I can now find are from the CFIDS Assoc. America.

However, others including myself have wondered what this operationalisation might actually mean as it is a term and a process with which we were unfamiliar. And how a criteria once agreed upon by experts and independently, is then measured as being effective.

It clearly isn't enough now to take a criteria and then simply run with it: what about those with a diagnosis now who might not meet the new criteria? And if it is a new criteria, would everyone need reassessing?

I don't think Fukuda was ever operationalised, or the NICE criteria either for that matter: at least I don't recall reading any papers about the NICE criteria which is an amalgam of others.

Jason above provides one example of operationalising the criteria - instead of simply asking people 'do you have this symptom' out of a range, one would be asked 'how often do you have this symptom' and 'how severe is the symptom' and then the results used to determine diagnosis.

Other examples must exist including perhaps, studying the effects of each criteria on patient cohorts, perhaps even replicating the above paper in full using all the criteria mentioned by HHS/IOM, trying to determine which method of diagnosis results in the more accurate cohort/the more accurate definition of this disease...

Jason is also asking above, what the effect is of having more symptoms to choose from, compared to a more focused list, and if the results indicate more issues that might also cross with a psychiatric diagnosis.

However, in the absence of a clear biomarker(s) I do wonder if any of this effort is actually worth it. At the end of the day, are we not still reliant upon various consensus opinions as to what is/what isn't ME?

IOM have 18 months to do their work and $1 million. It would be useful to know how they might seek validation and roll-out of any new criteria that is fundamentally different to Fukuda.
 
Last edited:

Ember

Senior Member
Messages
2,115
This study is another attempt by Dr. Jason to promote his own ME definition: “To meet full criteria of ME, patients must have had an acute onset and qualify for the three major ME symptom categories (postexertional malaise, neurological manifestations, and autonomic manifestations):”
ME Case Definition

We created a revised case definition for ME based on past case definitions, which include the Ramsay (1988) definition, the Dowsett, Goudsmit, Macintyre, and Shepherd (1994) “London” criteria, the Hyde (2007) Nightingale definition, and the Goudsmit, Shepherd, Dancey, and Howes (2009) criteria. Specifically, we used past case definitions to create a revised criteria based on the cardinal features of ME. The revised definition stipulates that ME had an acute onset that could be categorized into three categories: ME—viral (ME was precipitated by a virus), ME—infectious nonviral (ME was precipitated by a nonviral infection such as a tick bite), and ME—other (ME was precipitated by trauma or chemical exposure). These categorizations were based upon patients self-report. Additionally, patients had been asked to categorize their onset as sudden or gradual. In addition, they were asked: “Over what period of time did your fatigue related illness develop?” The responses included the following answers: within 24 hr, over 1 week, over 1 month, over 2–6 months, and so on. To meet ME sudden onset criteria, patients needed to indicate a sudden onset and the illness developed either over 1 week or within 24 hr.

The major symptom categories of ME in the revised case definition included: postexertional malaise, neurological manifestation, and autonomic dysfunction. Postexertional malaise was described as prolonged restoration of muscle power following either mental or physical exertion with recovery often taking 2–24 hr or longer. Neurological manifestations, which included at least one of the following symptoms: short-term memory loss, loss of powers of concentration, cognitive dysfunction, increased irritability, confusion, and perceptual difficulties. Autonomic dysfunction included at least one of the following: neutrally mediated hypotension, postural orthostatic tachycardia, delayed postural hypotension, palpitations with or without cardiac arrhythmias, dizziness, feeling unsteady on ones feet, disturbed balance, cold extremities, hypersensitivity to climate change, cardiac irregularity, Raynaud's phenomenon, circulating blood volume decrease, and shortness of breath. Secondary features of ME included: pain, endocrine manifestations, immune manifestations, and sleep dysfunction. To meet full criteria of ME, patients must have had an acute onset and qualify for the three major ME symptom categories (postexertional malaise, neurological manifestations, and autonomic manifestations). To be counted as “persisted or recurred,” the individual had to indicate a frequency score of 2 or higher. In addition, symptoms had to be rated at a 50 (as moderate) or higher to meet severity criteria. Both ratings of frequency and severity, therefore, had to be satisfied in order to qualify for each individual symptom. The ME group was not made up of all individuals from the ME/CFS group, as the ME group required Autonomic symptoms, whereas the ME/ CFS group only required 2 of the 3 of the Other criteria to be met.
Notice that PEM is defined here as “prolonged restoration of muscle power following either mental or physical exertion.”

Dr. Jason's recurrent theme is that “Katon and Russo (1992) have argued that a requirement of more symptoms to meet criteria could inadvertently select for individuals with psychiatric problems.” But his group ignores the fact that the CCC excludes primary psychiatric disorders when they argue, “Some might conclude that the ME/CFS definition requires too many symptoms that may at the upper ends select for psychiatric disorder. Others might feel that we should just expect higher levels of a variety of both medical and psychiatric disorders if this definition is utilized.” The DePaul group intends to continue in this vein: “We are currently collecting more data on this issue, and we hope in the future to be able to offer more definitive interpretations of the findings reported in the current study.”

Fukuda (1994) was operationalized, but not improved, by Reeves (2005).
 

Firestormm

Senior Member
Messages
5,055
Location
Cornwall England
Couple of things:

I was reading Jason's paper (top of the thread here) and noted they considered 'tick bites' as a trigger for ME/CFS.

I'd not seen that one before in print in a published paper. Not sure if I agree it either. Surely, Chronic Lyme as a diagnosis is diluted by such an inclusion, it also extends the catchment of ME as a distinct disease even further.

Is there anything that can't be seen now as a trigger for ME? Does it matter? What does it mean?

And yesterday, the following paper was published:

The role of clinical guidelines for CFS/ME in research settings

Purpose:

The purpose of this paper is to provide an overview of three sets of guidelines currently available: the Centers for Disease Control and Prevention (CDC) Toolkit; the International Association for Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (IACFS/ME) Primer; and the International Consensus Primer.

Results:


Guidelines vary significantly in the symptoms and comorbidities considered in light of the differing symptom requirements of three case definitions. There is also no specification on how symptoms should be measured, contributing to the significant heterogeneity found in CFS/ME.

Conclusions:

Further revision of clinical guidelines, preferably based on a definition that is well-informed by current empirical studies, is recommended to ensure that guidelines are applied with consistency and understanding in both research and clinical settings.

Might have some impact on the IOM exercise but it's a review of clinical guidelines, so perhaps not. Still I post it here, because it mentions (as does Jason) the lack of an ability to measure symptom severity (Jason also refers to frequency).

[N.B. Title link takes you to the research thread on the forum]