ME Research UK — Energising ME Research

Severely overlooked by science — an overview of research on severely-ill people with ME

Author

Dr Neil C Abbot, for ME Research UK and the 25% ME Group

Publication

A short version of this article originally appeared in the August 2004 issue of the magazine InterAction.

Part 2

A scandalous situation overall

As regards biomedical research overall, there is a serious mismatch between published studies and the extent of the human problem. While we know very little about the incidence and prevalence of ME generally, the proportion of patients with severe illness has been variously estimated at 25% (hence, the “25% ME Group for Severe Sufferers”), one third who experience “a severe and debilitating downhill course” (Dr Melvin Ramsay), and 34% as reported by Action for ME’s Members Survey of November 2000. This evidence, conjoined with findings from the CMO’s report that “a minority… remain permanently, severely disabled and dependent on others”, illustrates the scale of the problem that still needs to be addressed.

To put it bluntly, if 30% of ME patients in the UK are severely affected at any one time, this means that between 36,000 and 72,000 people are severely ill. For such numbers to be so under-investigated by scientists, albeit for practical reasons, is surely scandalous — yet this is the situation. The CMO’s report concludes that, “Current provision of services falls well below what is needed for the vast majority of severely and very severely affected patients” (Section 3.4.3.1).

Stabbing in the dark

It’s sobering to consider that in 2004 we still understand very little about the origin and outcome of severe illness in ME. Yes, we know that severe illness (and the other medical conditions that accompany it) complicates matters. We know that the prospects for recovery tend to be worse for those most severely affected, whether adults or children, and that specific viral triggers, duration of illness and socioeconomic status have all been associated with the severity of the clinical picture. Most importantly, we know that the cumulative impact of severe illness over many years, where there is no sense of improvement, is profound.

Yet, even these rather unsurprising findings are based on a relatively small number of investigations, and as such cannot be called definitive. The truth is that, after some stabbing in the dark, the complexity, severity and longevity of the illness are still only dimly perceived. The most meaningful information often comes from the dedicated efforts of specialist groups that have collected data on their severely ill members or clients (see below).

Sources of community-based information on severely affected ME patients

25% ME Group for Severe Sufferers — Severely affected analysis report, March 2004

Action for ME Report — Severely Neglected: ME in the UK, March 2001

Case History Research on Myalgic Encephalomyelitis (CHROME)

Soft data and hard experience

Clearly, community-based surveys can be very useful for describing the experiences of people with severe ME, and might be important for hypothesis generation; i.e., they may uncover areas of concern (such as the lack of community care provision), and highlight areas where new research is needed (such as the urgent need for pain relief). In short, they can provide a systematic record of individual suffering, and point to ways to alleviate it.

However, there is a very real problem about the meaning of survey data generally to medical/scientific professionals outside of ME patient circles. Strange as it may seem, surveys come low (grade III or lower) in the hierarchy of research designs, since they are not valuable for determining causation, or the ‘specific effect’ of treatment. In addition, charities’ in-house patient surveys have many limitations. First, most of the data has usually not been collected using validated, standardised outcome questionnaires that can be used for comparison with other studies or illnesses. Then there is the problem of poor response rates that range from 66% down to 31% in recent ME group surveys, raising the whole question of the meaning of non-responses, a statistical minefield (fortunately) beyond the scope of this article.

But the greatest flaw in the eyes of suspicious outsiders is the ‘Christine Keeler’ effect — “they would say that, wouldn't they”. Since the survey data emanates from a so-called self-selecting group of people with self-reported symptoms, a question mark hangs over the veracity of the data, especially if — as some psychosocial professionals maintain — claiming to be severely ill can help maintain a sickness role and acquire state benefits.

A good example of the difficulties of getting such information published came in 2002 when the 25% ME Group and ME Research UK tried to publish a small table of data as a letter in the Journal of the American Medical Association (read it here). To say there was a lack of interest is an understatement: in the end, the data was presented in ME Research UK’s report Unhelpful Counsel (read it here), and so was seen by, at most, a few hundred people instead of the 200,000+ members of the American Medical Association, the sort of audience the most severely affected patients, and the data, deserved. These problems with survey data are not specific to ME, by the way, but this illness — with its peculiar combination of public scepticism and professional uncertainty or outright distain — is probably more at risk than most.

Preparing to climb the mountain

Given that surveys are things that patient charities are well-positioned to undertake, there are in fact some simple things that groups representing the severely ill can do to facilitate the acquisition of valid information. One is to include with their custom-designed questionnaires at least one recognised validated outcome measure, such as the Medical Outcomes Study SF36, a short, easily-completed measure of quality of life that allows direct comparison with other illnesses and other types of ME patients. Again, groups could be willing to supply data for analysis by independent organisations though, of course, the ideal situation is for a university department or other professional group to be involved from the outset.

However, a most useful advance would be a national epidemiological investigation of the true prevalence and impact of severe ME. Indeed, as the CMO report made clear, the organisation of primary care services in the UK offers a unique opportunity to undertake prevalence studies on the national scale needed to generate the requisite data.

To end where we began, the same report stated in 2002 that “care of people who are severely affected is an urgent challenge”. Scientific investigation of the severely affected — their clinical status, experience of illness and treatment — is also a challenge that should be swiftly met.

Ignored and invisible, maybe — but it doesn’t have to be like that. Energy, vision and funding can transform any situation.