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Spring 2002

Believing in CFIDS: The Role of Research Methodology in Gaining Public Support
By Kim Kenney

Credibility has long been the Holy Grail of the CFIDS community. Since the 1980s, when research and news publications disparaged the illness as a form of social hysteria that offered stressed overachievers or bored middle class housewives a way to "opt out" of their unfulfilled lives, people with CFIDS (PWCs) have worked to foster better understanding of their illness and more acceptance for the way in which CFIDS devastates lives. A diagnostic marker, a name change, a celebrity spokesman and a CFIDS story line on "ER" or a popular soap opera have all been proposed as instant solutions to close the credibility gap.

With none of the above likely to occur immediately, what can be done to strengthen the case for CFIDS? Improving the quality of CFIDS research holds promise in the short run and long term. In particular, tighter research methods in CFIDS studies would give us a stronger case to take to the media, the government and other quarters of the research community. It can do more to build credibility than PWC Oscar winners Cher and Randy Newman holding a CFIDS benefit concert in Central Park.

Of course, it also would offer hope to thousands of people with CFIDS looking for a cure for the illness that has so devastated their lives. Medical research is an inexact science that requires intellect, logic, reason and artful creativity. Studying a complex and misunderstood topic like CFIDS also demands perseverance, courage and a thick skin. Pioneers in CFIDS research have stretched minimal staff resources and leveraged scarce funds to document what we know about this puzzling condition. But the research literature is replete with studies that contradict one another, offering skeptics plenty of ammunition.

There is broad agreement within the CFIDS research community that many of the differences in study results are likely due to varying methods of defining study participants, and to establishing study protocols. For example, in the mid-to-late 1990s several research groups set out to test a Johns Hopkins University research report that a high percentage of PWCs also had an autonomic nervous system (ANS) condition known as neurally mediated hypotension (NMH). The Hopkins study screened CFS patients for symptoms consistent with NMH and performed formal tilt table tests on those who appeared likely to have a form of ANS dysfunction. When other groups tried to validate the study, they opted not to pre-screen patients, used different protocols for the tilt study or employed different standards to define a positive result. It didn't help that even experts have trouble agreeing on the "right" way to conduct and interpret these tests.

Not surprisingly, research results varied significantly between groups, casting doubt on the benefit of further exploring mechanisms and treatments for ANS dysfunction in CFIDS. The variable findings also doused patients' and clinicians' hopes that objective proof would finally arise and silence the critics.

Another striking case of the impact of methodology dates back to 1990, when the U.S. Centers for Disease Control and Prevention (CDC) launched a study to determine how many U.S. adults had CFIDS. This study used the stringent 1988 CFIDS case definition that, by many practitioners' reports, was difficult to apply. They asked selected clinicians in Reno, Nev., Wichita, Kan., Grand Rapids, Mich., and Atlanta to refer suspected cases of CFIDS to a physician review panel. When the study ended, the CDC estimated that 3,000 to 10,000 American adults had CFIDS. Patient advocates knew this estimate represented only the tip of the iceberg and they pushed for new studies to be done in different ways. Ten years later, both CDC and DePaul University researchers independently did studies that put the estimate at 500,000-800,000 CFIDS cases among U.S. adults (using the broader 1994 case definition). Both groups found that 85-90 percent of CFIDS cases had not been diagnosed by a health care professional, highlighting the critical flaw of the four-city study conducted a decade earlier. It was clear that the higher estimates were due more to improved study design than a surge in the illness itself.

The CDC and DePaul estimates of CFIDS cases became powerful tools in advocacy and public relations efforts. Convincing government officials and media of the scope and severity of CFIDS was made easier using statistics from these two studies that employed similar methodology and reached similar conclusions. It eliminated the need to explain why poor study design vastly underestimated the number of cases. It also allowed advocates to emphasize the impact of the disease on patients' lives and focus lawmakers' attention on how they could help constituent PWCs. In addition, it provided data documenting the dramatic need for health care provider education about CFIDS and the limited access to health care services by PWCs. These compelling numbers helped put CFIDS in context of better-recognized public health concerns, allowing advocates to use comparisons such as, "twice the number of people affected by multiple sclerosis."

A recent joint effort by the Agency for Healthcare Quality and Research and the National Institutes of Health set out to produce an "evidence-based" report on CFIDS treatment studies. This attempt at defining proven therapies actually yielded a fairly dismal report card on CFIDS treatment research. The summary uses language like "scant and insufficient data," "no consistent patterns" and "not well established." It highlights often weak study methodology and underscores the need for more research, larger studies and greater uniformity in designing treatment trials. Although progress has been made (see commentary on p.1), the report casts a long shadow on the entire field of CFIDS research.

Bringing CFIDS out of that shadow and into the light requires a major effort employ-ing numerous complementary strategies. Rock-solid research results are the best way to attract widespread media attention, so strengthening research methodology must be one of those strategies. Good research attracts greater funding, which lures more top-flight researchers, who in turn can produce headline-catching research findings. Good research begets more good research - and good research creates opportunities to shape public opinion and public policy, improving circumstances for all those affected by CFIDS.

We do need a celebrity spokesman and "chronic fatigue syndrome" is a woefully inadequate and derisive term. We must continue efforts to attract influential advocates and rename the illness to speed our work. Generating meaningful momentum in CFIDS research will bring more resources and credibility to our cause and, ultimately, a better quality of life to PWCs.

Kim Kenney is president & CEO of The CFIDS Association of America.

Methodologic problems common in CFIDS research

• Small numbers of patients/controls
• Inconsistent application of case definition
• Poorly selected and/or defined control groups
• Failure to sub-group patients by length of illness, age at onset, severity, etc.
• Variable lab and testing protocols and standards · No standardization of outcome measures
• Lack of confirmatory studies