Why is ME/CFS getting so little research funding?

In a previous blog, Evelien and I argued that ME/CFS is getting less research funding than illnesses with a comparable prevalence and disability. In this article, we will try to get a better understanding of why ME/CFS gets so little research funding. Many causes have been proposed such as stigma, lack of a biomarker, mischaracterization of ME/CFS as a psychosomatic condition, and so forth. 

Our main conclusion is that except for disease burden, it’s quite difficult to identify factors that determine the level of research funding. There aren’t any obvious explanations that stand out. Given the high disease burden of ME/CFS, it remains inexplicable why so little funding has been devoted to research its cause and pathology. The most likely explanation is that there are multiple factors at play and that for each one of these, the odds are stacked against us.

DALY and the Global Burden of Disease Study

Researchers usually compare the level of research funding based on some measure of how impactful a disease is. If one illness is more prevalent or deadlier than another, it makes sense that more funds are devoted to studying its cause and treatment. The term ‘disease burden’ is intended to take these factors into account. There are several measures used to reflect disease burden, but the most popular, and arguably the most accurate one, is DALY.

DALY stands for Disability-Adjusted Life Year. It expresses lost years of “healthy” life. To calculate DALY, researchers take into account the prevalence of the illness, how disabling and long-lasting it is, the number of deaths it causes, and the life expectancy at death. The concept originated from a landmark study by the World Health Organization in 1990, but there have been multiple updates since. The latest version of the Global Burden of Disease (GBD) study presented data on 369 diseases and injuries and was published in October 2020 in The Lancet. Unfortunately, it didn’t provide a DALY estimate for ME/CFS but Mary Dimmock, Art Mirin, and Leonard Jason provided an estimate back in 2017 (with an update in 2020).

The disease burden of ME/CFS

Unfortunately, there wasn’t much data for Dimmock and colleagues to go on. They had to calculate ‘years lost due to disability’ based on data provided in a 2003 Australian disease burden study and a 2015 Danish study that compared the quality of life of ME/CFS to various other diseases. Both studies had major limitations. The first was based on extrapolations and registrations using the International Classification of Diseases (ICD), the second looked at patients who were part of the Danish ME/CFS Association and who self-reported a diagnosis of ME/CFS. Both studies used the EQ-5D for their ME/CFS estimates while the GBD study used a different methodology.

Getting an ME/CFS estimate for ‘years lost due to death’ was even more difficult. Dimmock et al. focused on two papers on premature death due to ME/CFS, published by the research team of Leonard Jason from DePaul University, Chicago (here and here). Both of these were retrospective studies: they asked about deaths due to ME/CFS that had already occurred, so after the persons were deceased.

Other, longitudinal, studies (here and here) that followed patients over time, reported no significant increase in death due to ME/CFS. Therefore, to remain cautious, we have included two estimates for the DALY of ME/CFS: the one estimated by Dimmock and colleagues and one where ‘years of life lost’ was set at 0. As the graph below shows, ME/CFS has a large disease burden, larger than multiple sclerosis, Parkinson’s disease, epilepsy, and autism, even if we assume it causes no excess death. All figures are DALY estimates for the United States in the year 2015 (if we looked at worldwide cases, the disease burden of illnesses such as tuberculosis would be much higher).

NIH funding per disease burden

The next step is to compare DALY estimates with research funding for each disease category. We focus on funding data of the National Institutes of Health (NIH) in the United States, the largest funder of health research worldwide. In 2015, the NIH looked into their funding records and matched their illness categories with those of the GBD study. The results were used in many studies including the one by Dimmock, Mirin, and Jason (who were kind enough to send us their data and calculations).

We divided the amount of NIH funding (in millions of dollars) by the DALY estimates for each illness category. The average (in yellow) indicates the virtual amount of funding that an illness would get if all funding was divided equally based on their disease burden (to clarify: in reality, this isn’t the case: disease burden determines only a third of the variation in NIH funding).

As the table below shows, ME/CFS gets by far the least amount of funding per disease burden, even if we take the cautious estimate of DALY that assumes no excess mortality, ME/CFS simply gets so little funding that it makes hardly any difference whether we assume there are premature deaths due to ME/CFS or not.

Because the NIH funding per DALY is so unevenly distributed we had to take the logarithm of each amount to fit them into the same graph. The length of the bar might therefore be a bit misleading (a one-point difference represents multiplication by 10). More important is the rank: the higher up the bar chart the more funding an illness category is receiving per DALY.

ME/CFS gets the lowest level of funding per disease burden

As the graph shows, ME/CFS is at the very bottom of the list. That could be because the DALY for ME/CFS was calculated differently than for the other illness categories. But even if Dimmock and colleagues overestimated the DALY for ME/CFS tenfold, ME/CFS would still be at the bottom of the list, below pneumonia. The average – the amount of funding that an illness would get if all funding was divided equally based on disease burden – is 115 times the level of funding ME/CFS got in 2015. The conclusion by Dimmock and colleagues that ME/CFS is significantly underfunded, remains robust.

Explanation 1: are psychosomatic conditions getting less funding?

The list of other illnesses might give a clue of why ME/CFS is receiving so little funding per disease burden. One common explanation is that the mischaracterization of ME/CFS as a psychosomatic condition hampered investment in research. The myth that ME/CFS can be successfully treated (or even cured) with graded exercise therapy (GET) and cognitive behavior therapy (CBT) is also thought to be a hindrance to more research into the underlying pathology of ME/CFS.

If we look at the graph, we see that psychosomatic or psychiatric conditions (indicated in green) do receive less funding per DALY than average but they still receive significantly more than ME/CFS. Anorexia, anxiety, and depression are getting more than 10 times the level of funding per DALY than ME/CFS. For each of these conditions, the idea that unhelpful thoughts and behavior are perpetuating symptoms is equally popular as for ME/CFS (whether this idea makes sense or not is outside the scope of this article). For schizophrenia, CBT has also been a popular intervention including a technique called “peripheral questioning” where patients’ delusional beliefs are questioned. As with ME/CFS, there have also been CBT-trials that focus on recovery. Despite all of this, schizophrenia is receiving much more funding and clinical trials than ME/CFS, including trials on pharmacological interventions.

Explanation 2: self-inflicted, less worthy of investment?

Another explanation states that illnesses that are believed (rightly or wrongly) to be self-inflicted are also seen as less worthy of investment. “We tend to underfund things where we blame the victim,” said Claiborne Johnston, Dean of Dell Medical School. That might explain why an illness such as chronic obstructive pulmonary disease (COPD) – which is often caused by lifestyle choices such as smoking – is at the bottom of our list.  But as our graph of NIH data shows, some illness categories that have the strongest perception of being self-inflicted such as sexually transmitted diseases, alcoholism, and drug use disorders get a lot of funding per disease burden.

Explanation 3: women’s diseases downplayed?

Some have noted that many underfunded diseases predominantly affect women. In her book ‘Doing Harm’, Maya Dusenbery highlights that chronically underfunded illnesses such as vulvodynia, endometriosis, interstitial cystitis, temporomandibular joint dysfunctions, and fibromyalgia all disproportionately affect women. Dusenbery also has an explanation for this, namely that women are frequently seen as inaccurate judges of when something is seriously wrong in their bodies. She writes: “often, women’s symptoms are brushed off as the result of depression, anxiety, or the all-purpose favorite: stress. Sometimes, they are attributed to women’s normal physiological states and cycles: to menstrual cramps, menopause, or even being a new mom.” The data on NIH funding and disease burden seem to support this hypothesis with migraine and endometriosis appearing at the bottom of our list. On the other hand, some cancers that primarily affect women receive higher funding per disease burden. And despite being underfunded, in 2015, endometriosis still got more than 10 times the amount of funding per disease burden than the amount ME/CFS received.

Explanation 4: lack of a biomarker, lack of progress?

Another common explanation is that ME/CFS lacks a biomarker and is most likely a heterogeneous construct. ME/CFS might act as an umbrella for multiple poorly understood health conditions that have  symptoms in common but differ greatly in their underlying pathological mechanism. If true, this would mean that research on ME/CFS is more complicated than research on illnesses where a biomarker is available and where this could act as a starting point for further leads.

Funding is mostly determined by the incentives of researchers and they want a return on investment. They generally prefer to study a hypothesis that is likely to be true or interesting so that their expertise is valued. If research hypotheses are no more than a shot in the dark, that would jeopardize a scientist’s career prospects. The problem with ME/CFS might thus be that, because of a lack of a biomarker and its likely heterogeneous nature, the return on investment is too low to attract new researchers. With ME/CFS, every idea is like a toss of the dice. That might explain why the field remains small and populated by academics who are unable to recognize that the data fails to support their hypothesis. It might also explain why progress is mostly driven by scientists who want to make a difference regardless of their career prospects (for example because they have a family member or friend with ME/CFS).

The lack of a biomarker and research leads might be the reasons why ME/CFS research gets so little funding. Our graph however indicates that some illnesses with no biomarker get much more funding per disease burden, ADHD, and autism being notable examples.

Other explanations: a new diagnosis, stigma, and invisibility

We have tried to think of other explanations for the underfunding of ME/CFS research such as the fact that it’s a relatively new diagnosis.

Firstly, ME/CFS only came to prominence in the US after Holmes and colleagues published their case definition in 1988. But HIV/AIDS is also a new disease and it has been at the top of NIH funding for years.

Secondly, ME/CFS is a stigmatized illness, which might prevent researchers from studying it and patients and their relatives from raising sufficient funds for research. As Tom Kindlon pointed out in blog posts (here and here), much of the research funding consists of private funding, and public funding is often a reflection of that. So one of the problems with ME/CFS research could be that not enough money is being raised privately. This is however a hypothesis that is difficult to test, as data on private funding is hard to retrieve.

Thirdly, it could also be that ME/CFS is an illness that, because of its enormous debility, remains largely hidden in our society. Many severe patients are confined to bed in a quiet darkened room and are therefore invisible to the outside world. Most don’t die or recover in a noticeable way that might get the attention of the general public. They remain ill and invisible for decades.

Conclusion

In this blog post, we have explored multiple explanations of why ME/CFS gets so little research funding.  Our main conclusion is that no explanation is fully persuasive on its own. Looking at the NIH funding per disease burden, there isn’t an obvious reason that stands out. Underfunding of illnesses is likely the result of multiple factors which in the case of ME/CFS, come together into a perfect storm. Identifying the main causes of the lack of funding for ME/CFS research could help us get out of the storm.

If you can think of explanations that weren’t mentioned in this article, feel free to post them in the comments below. We believe more data is needed about this crucial issue, so that progress can be accelerated. For one, it would be interesting to hear the perspective of researchers, both those involved in ME/CFS and those who are not.

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4 Comments

  1. I think a lot of the reason is a combo of CDC staff, insurance companies, Wessely and Strauss/Fauci. I think Osler’s Web covers a lot of it. And Mary Dimmock’s overview is outstanding https://www.dropbox.com/s/fkyl3uyr9x6twyh/Thirty%20years%20of%20Disdain%20-%20Summary.pdf?dl=0
    I’ve got ME-brain quite badly right now but some of the elements are:
    1. When the US had outbreaks in California, Lydonville and then Tahoe – the CDC staff were dismissive and negligent. They didn’t research the outbreak – I think they went skiing instead at Tahoe. Then later had poisoned atmosphere with signs up about the zombie apocalypse (ME patients) and used ME funding to buy furniture etc. Reeves, later DIrector, turned whistleblower on it

    2. It was just after AIDS and insurers didn’t want to pay for another chronic incurable (as AIDS was then) disease. A number of the Wessely school were hired by insurance companies as advisors. Wessely himself contacted the UK’s DWP when it was about to fund aids and support for people with ME and converted it to the bsp model…….. . And Wessely was on the SMC, preventing any reporting on biomedical research for decades He created the Oxford definition of tired people and called that CFS + said CFS was a new version of hysteria. He got Cochrane to classify ME as a mental health condition. Wessely, and SHarpe and Wite as well I think were invited to consult with the US

    3. I forget all the details of the Straus/Fauci work to stop the funding of research. THe is a letter from Straus to Fukuda about “disappearing” CFS by making fatigue the main diagnostic criteria https://forums.phoenixrising.me/threads/nih-strauss-cdc-fukuda-iom-the-cfs-report.29470/ and I also believe Wessely corresponded with Straus. Wessely and SHarpe were part of the Fukuda definition as was Straus. I can’t remember who got CFS assigned to the Office of Research on WOmen’s Health – a small dept with no funding budget! And Fauci at NIAID refused to fund….
    THe bottom line of it all being a culture of no belief with a psychogenic bias, leading to an ensuring of little funding and no medical education, after having engineered watered down definitions of what the americans now called CFS>

    also, Jason documented researchers and educators being threatened with losing their jobs if they tried to work in the ME CFS field. https://www.frontiersin.org/articles/10.3389/fped.2019.00131/full#B49

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  2. Notice that the diseases at the top of your list are transmissible? Those are the diseases that get the most funding, full stop. A problem with the ME community world-wide is that it cannot, will not, believe or accept that ME is an infectious disease. I can hear people screaming right now. Their fear of becoming pariahs has always been greater than their desire to be cured. (Irony is that they are already pariahs…) They allowed a handful of health policy people who knew little about the disease turn it into “fatigue,” a category in which it sill resides, 40 years later. In other words, a non-disease that sprang from the Id. Only a handful in medicine even knew about ME until the mid-1980s, when MDs everywhere began to notice large numbers of similar cases of a disease they thought was new in their practices. It was ME. They were assured by respected institutions like CDC, FDA and NIH that the malady was a condition of low-achieving, sympathy seeking women, or high-achieving women, Type A women trying and failing to have it all–take your pick. Recently, one reliable estimate put the number of ME cases at 65 million world wide. ME long ago surpassed AIDS and at least in the US, very likely tuberculosis and numerous other infectious diseases. The immune abnormalities in ME scream infection, and so does everything else about the disease as a matter of fact. But, especially the epidemiology. You don’t go from zero cases to 65 million in a generation unless it’s transmissible. Something occurred in late 70s and early 80s to start the worldwide spread of ME. Once considered epidemic, it is now endemic. What makes NIH throw billions in cash, year after year, at certain diseases and neglect others? If there is genuine fear of an epidemic, and there are bodies on the ground as with Covid or AIDS, the money will never stop flowing. Any effort to calculate a reasonable sum of money for ME by considering issues like “disease burden” or other superfluous categories is a) wasted effort and b) results in a profound underestimate of the actual sums needed. Your list above certainly is familiar to anyone with ME, but the real reason ME gets no money is that governments’ efforts to disappear ME as a non-threatening, questionable disease unable to be transmitted–except via commiseration among its victims–made it easy for everyone to ignore–or laugh at. Rather than facing up to the fact that they had an AIDS-like disease staring them in the face and pouring billions into searching for the causative agent and drug therapies, government health policy makers dumped ME on the side of the road to focus on AIDS, and now Covid. Until patients start fighting for the truth, and give up worrying about being seen as pariahs, staus quo on the money front is a sure thing. What if you could catch ME? That’s what public service announcements should be messaging to the masses, followed by, “You can.”

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    1. Thanks for your response.

      My blog has now changed place to mecfsskeptic.com would it be possible to copy-paste your response there so more people might read it?

      I suspect that most patients and doctors would have no problem accepting that ME is an infectious disease – if that were indeed the case – because that would surely increase the scientific interest into the illness.

      I think the current evidence for ME being infectious isn’t very strong. There isn’t evidence of marked regional differences as is the case for say Lyme Disease and since the 1980s there have been little to no reports of clusters or outbreaks.

      The main argument for this infectious hypothesis is the sudden increases of cases in the 1980s but I think most experts believe that this increase could also be explained by an increase in recognition of the syndrome. The same was true for Multiple Sclerosis at the end of the 20th century –from a rare and recently discovered illness, MS quickly became a frequent diagnosis in neurology clinics.

      There are no reports of physicians who would like to study ME/CFS but couldn’t find it. Basically, everyone who bothered to take a look since the 1980, found ME/CFS patients to study: in Harvard, LA to Miami.

      You mention a figure of 65 million worldwide but that is based on prevalence studies where a whole community was screened. Those studies indicated that the vast majority of patients aren’t diagnosed. So it isn’t the case that suddenly hundred thousands of patients were showing up and getting diagnosed by multiple physicians as one would suspect with an infectious outbreak. It’s more that a few physicians got interested in the illness and that their estimates and community screenings suggest that it is quite prevalent. For all we know, that might have always been the case. The illness might not have been picked up or was disguised under other diagnostic labels as is probably still the case in non-Western countries.

      The prevalence studies that we have do not suggest a stark increase of cases and the regional differences that exist (for example in Europe). They suggest increased medical awareness of ME/CFS rather than an infectious spread.

      Take for example the increased awareness of ME/CFS and the increased number of patients in Germany. I would argue that has simply to do with physicians like Scheibenbogen who are diagnosing patients with ME/CFS, not with ME/CFS becoming more prevalent or a virus hitting Germany.

      If you disagree with what I wrote, it would be interesting if you could summarize your main arguments for an infectious onset of ME/CFS so that the hypothesis can be more adequately discussed.

      Kind regards,

      Michiel Tack

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