Preprint Underdiagnosis of myalgic encephalomyelitis/chronic fatigue syndrome-like illness in a large integrated healthcare system, 2024, Wood, Unger et al.

SNT Gatchaman

SNT Gatchaman

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Underdiagnosis of myalgic encephalomyelitis/chronic fatigue syndrome-like illness in a large integrated healthcare system — Kaiser Permanente Northern California, 2022-2023
Mariah Wood; Nicole Halmer; Jeanne Bertolli; Laura B. Amsden; Joshua R. Nugent; Jin-Mann S. Lin; Gretchen Rothrock; Joelle Nadle; Shua J. Chai; Jamila H. Champsi; James Yang; Elizabeth R. Unger; Jacek Skarbinski

BACKGROUND
Surveillance of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS), a chronic, debilitating multisystem illness, is challenging because ME/CFS can be under-recognized in healthcare settings.

METHODS
Using a population-based panel study of 9,820 adult members of Kaiser Permanente Northern California (KPNC), a large, integrated healthcare system, we compared survey-defined ME/CFS-like illness with presence of an ME/CFS diagnosis in the electronic health record (EHR) to evaluate ME/CFS underdiagnosis.

RESULTS
Of those with survey-defined ME/CFS-like illness, an estimated 97.8% (95% confidence interval [CI] 97.1%-98.4%) did not have an ME/CFS diagnosis in the EHR. The group without EHR diagnosis was younger, less likely to identify as white non-Hispanic, and more likely to have developed fatigue in the past three years than the EHR diagnosed group. Both diagnosed and undiagnosed ME/CFS-like illness groups had significantly impaired physical, cognitive, and social functioning, and significantly worse mental health and anxiety than those without ME/CFS-like illness.

CONCLUSIONS
ME/CFS is underdiagnosed in the Kaiser Permanente Northern California healthcare system. Enhanced syndromic surveillance that characterizes patients with ME/CFS who have not been diagnosed has the potential to increase timely recognition of ME/CFS.


Link | PDF (Preprint: MedRxiv) [Open Access]
 
The primary author is has a Kaiser Permanente email address. It's very good to see health insurers and providers taking an active interest in quantifying the scale of the problem.

In collaboration with Kaiser Permanente Northern California (KPNC), the California Emerging Infections Program conducts enhanced syndromic surveillance for ME/CFS through the Surveillance to Optimize Protocols for Early Identification and Subgrouping of ME/CFS (STOP ME/CFS) project. STOP ME/CFS involves annual surveys to identify and follow patients with ME/CFS-like illness among KPNC’s 4.5 million members, who are demographically similar to the California population (6).
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A stratified random sample of adult English-speaking KPNC members who had been members for at least one year and who had a valid email address on file were recruited from seven mutually-exclusive strata, including:
1) ME/CFS diagnosis in the electronic health record (EHR);
2) post-COVID conditions diagnosis in the EHR;
3) persons at higher risk for ME/CFS based on an internally-created predictive model;
4) persons who had COVID-19 before July 5, 2021;
5) persons who had COVID-19 between July 5, 2021 and December 14, 2021;
6) persons who had COVID-19 after December 15, 2021 through beginning of recruitment (May 9, 2022); and
7) persons who did not fit into any above strata.
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Sampled persons were invited to participate in an online survey from July 10 to October 17, 2022. Of 98,505 eligible sampled members, 9,825 (10.0%) completed the survey in year one. In year two, all respondents from year one were contacted again from May 1 to November 27, 2023; of those respondents, 6,393 (65.1%) completed the survey again in year two (since recruitment, five respondents requested data removal; the updated number of respondents is 9,820).
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A 10% response rate means of course that there is an enormous amount of bias in the results. Also, the sampling was focussed on the first three strata. There was weighting for non-response, aiming to produce unbiased population level predictions of prevalence.
 
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Of the respondents, 645 in year one and 316 in year two (not mutually exclusive) met criteria for survey-identified ME/CFS-like illness based on IOM ME/CFS diagnostic criteria (Supplemental Table 1). In total, 798 persons had ME/CFS-like illness in either year one, year two, or both.

Results
we estimated that 1.8% (95% confidence interval [CI] 1.3%-2.2%) of adult members had survey-identified ME/CFS-like illness during the two-year study period. Among those with survey-identified ME/CFS-like illness, an estimated 97.8% (CI 97.1%-98.4%) did not have any ME/CFS diagnosis in their EHR, with only
2.2% (CI 1.6%-2.9%) having a ME/CFS diagnosis in their health record.
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There are a lot of issues of course, some of them major - e.g. how accurate is survey identified ME/CFS? But, it is suggesting that any large-scale study relying only on formally diagnosed participants will be missing a large chunk of the disease population.


Generally, persons with ME/CFS-like illness and an EHR diagnosis had lower well-being scores than those with ME/CFS-like illness without an EHR diagnosis; however, persons without an EHR ME/CFS diagnosis had a significantly lower SF-36 mental health T-score (30 vs. 34) and significantly higher anxiety score on GAD-2 (worse anxiety) (4 vs. 3) than persons with an EHR ME/CFS diagnosis.
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Persons with ME/CFS-like illness without an EHR diagnosis had worse anxiety than persons with ME/CFS-like illness and an EHR diagnosis, suggesting a possible effect of unrecognized illness on mental health among people with ME/CFS symptoms.
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Interesting although there are many possible interpretations of those findings. Diagnosing a person with ME/CFS doesn't seem to increase well-being, contrary to the suggestions that diagnosis improves prognosis (unless of course medical attention results in people not having ME/CFS). The increased mental health scores in people without an ME/CFS diagnoses might be partly because they have been told that they are anxious and depressed, and they have internalised those diagnoses.
 
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In an integrated healthcare system with 2.7 million adult members, an estimated 1.8% had symptoms consistent with ME/CFS-like illness during a two-year period. Of those with ME/CFS-like illness, 97.8% had no documentation of an ME/CFS diagnosis, despite a high level of primary care utilization; 53% of those with ME/CFS-like illness and no EHR diagnosis reported fatigue began less than three years prior.
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There might be an unusual increase in ME/CFS prevalence due to covid-19 - so a bubble of undiagnosed people with ME/CFS are working their way through the diagnosis process. Possibly, people who meet ME/CFS criteria are being diagnosed with Long Covid rather than ME/CFS. Oddly, Long Covid was not mentioned at all in the paper, nor PASC.

I think this paper provides evidence of a substantial increase in the prevalence of ME/CFS in the last three years (i.e. in the Covid years), although there are issues with it.
 
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This report documents potential underdiagnosis of ME/CFS by healthcare providers, even in an integrated healthcare system with accessible primary care services. Numerous factors may contribute to the high level of underdiagnosis of ME/CFS, including the lack of a diagnostic biomarker, a low level of dissemination of standardized screening and assessment tools, limited uptake of clinician training on
ME/CFS, a low-level of patient awareness of ME/CFS, misconceptions among clinicians that the illness is psychogenic in nature or does not exist, and the fact that ME/CFS symptoms can overlap with other illnesses (1,19). The symptoms of the undiagnosed group may provide insight into screening for and monitoring ME/CFS earlier in the illness course.

These findings indicate the importance of enhanced symptom-based surveillance as an initial step in recognizing undiagnosed ME/CFS, as those with undiagnosed ME/CFS-like illness would not have been identified without this project’s symptom-based surveillance efforts. Enhanced syndromic surveillance has the potential to increase timely recognition of ME/CFS. Public communication campaigns could also promote earlier recognition of ME/CFS symptoms, which could lead to earlier care-seeking and care provision.
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Kaiser Permanente have, I think, a relatively good track record on ME/CFS, and it's good to see them investigating the prevalence and still noting that ME/CFS does exist and isn't psychogenic.

I just hope the outcome of the study isn't a big effort to diagnose people with ME/CFS and then deny those people further investigations because they have a diagnosis. Although, even that could be a step forward - people given a label but no treatments are likely to be dissatisfied and join advocacy movements calling for research.
 
I'm not sure how this fits with the reported prevalence in the latest CDC update:
Hutan said:
STOP ME/CFS project
Overall, the researchers estimated that 1.67%, or 45,892, of 2.7 million adult KPNC members of the Kaiser plan in Northern California had ME/CFS-like syndrome during the study period, which was July to October 2022. Of those, 14% developed the illness after COVID-19.
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CDC is working with the Emerging Infections Program in California and Kaiser Permanente Northern California. Work on quantifying the incidence of ME/CFS has been published. The study is ongoing.
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This study suggests that a lot of people have developed ME/CFS in the last three years. I'm not sure how you would know what percentage of those new cases are due to Covid-19 when asymptomatic and repeat infections are thought to cause ME/CFS.
 
Hutan said:
Diagnosing a person with ME/CFS doesn't seem to increase well-being, contrary to the suggestions that diagnosis improves prognosis (unless of course medical attention results in people not having ME/CFS).
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There’s probably a severity bias in who gets diagnosed. And also, with the majority of doctors in the healthcare system, having an ME diagnosis basically at best is an equivalent of having no diagnosis, at
worst an equivalent to having a “psychosomatic disease” diagnosis.
 
"All of questions 7 through 10 must be answered with one of the responses in bold to meet inclusion criteria for post-exertional malaise"

7. During the past 4 weeks, have you been unusually fatigued or unwell for at least one day after exerting yourself in any way?
  • Yes
  • No
8. During the past 4 weeks, how often have you had unusual fatigue after exertion?
  • A little of the time
  • Some of the time
  • A good bit of the time
  • Most of the time
  • All of the time
9. During the past 4 weeks, how bad was your unusual fatigue after exertion?
  • Very mild
  • Mild
  • Moderate
  • Severe
  • Very severe

10. How long have you had unusual fatigue after exertion?
  • Less than 6 months
  • 6 months up to 1 year
  • 1 year up to 3 years
  • 3 years up to 5 years
  • 5 years up to 10 years
  • 10 or more years
 
According to the questionnaire used by this research team.

The IOM defines it as "Post-exertional malaise (prolonged exacerbation of a patient’s baseline symptoms after physical/cognitive/orthostatic stress; may be delayed relative to the trigger)".
 
I think they were wrong to use the term "fatigue", since that's poorly defined, and the definition or recognition differs between people. My journal doesn't use the word "fatigue". "Lethargy", "sluggish", "lousy" and "bleh" are more common. Note that I can't even find one word that fits every occurrence. Their acceptance of "unwell" for question 7 was good, and should have been applied to the others. Hmmm, I suppose I could have used "unwell" in my journal, but the subtle differences in my choices might be useful data.
 
Andy said:
"All of questions 7 through 10 must be answered with one of the responses in bold to meet inclusion criteria for post-exertional malaise"
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Yeah, that's hopeless. I wonder if they had any expert patient involvement. Aside from the focus on fatigue rather than unwellness, there's the requirement that PEM has to happen at least 'a good bit of the time'. If a person is able to manage their exertion levels so as to largely avoid PEM, then PEM may have only happened over 'a little of the time'.
 
I have no direct insight but from my perspective of being a physician/scientist trying to define what makes ME/CFS a useful concept this sort of questionnaire approach also seems hopeless.

My understanding of the PEM phenomenon has nothing to do with it having to be regular or often. I tend to think of the person with ME/CFS struggling day by day to achieve a few things they have got used to managing and then, unpredictably, 'crashing' at intervals of weeks or months. There may be daily payback from doing the things that can be managed but I don't see that as necessarily key to ME/CFS. And perhaps the most crucial thing is the unpredictability either for the individual or between individuals. Each person may tell a different story but there is something common to all. Also, the requirement for 6 months is to me less about needing to be ill for 6 months to qualify and more about it taking 6 months observation to be sure that the fluctuating pattern of the illness is of the ME/CFS sort.
 
I'm a patient at CA Kaiser Permanente. My doctor is Dr. James Yang, one of the people credited in the paper. This is my understanding of the situation:

WebDog had extensive contact with Dr. Steve Olson. Dr. Steve Olson created an "ME/CFS Consultative Service" and also caused some administrative changes. He then retired in about 2019-2020. He has an interview with David Tuller where he talks a little bit about what he accomplished.

Currently, this Consultative Service is 5 or 6 part-time doctors. It only serves people in CA. Wait times are too long, 3 months for an initial appointment. It is now run by Dr. James C Yang. I would say that the care that patients have received at the Consultative Service is kind of decent. They definitely understand the basics of pacing and are willing to prescribe basic medications. They won't do anything super out-there or expensive though.

But most CA Kaiser ME/CFS patients don't know this Consultative Service exists. Their doctors don't know it exists and don't refer their patients there. And the service is obviously not big enough to see the amount of patients they need to. I've talked to a lot of ME/CFS Kaiser patients and they usually describe horrible care, rheumatologists who tell them it's basically the same as fibromyalgia, being denied a diagnosis, being referred to psych, being encouraged to try physical therapy, no recognition of dysautonomia or MCAS or other co-occurring conditions, etc.

I could conceivably talk to Dr. James Yang about this study the next time I see him, not sure what would be useful to remark upon though.
 
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