RETURN TO TABLE OF
in CFIDS: The Role of Research Methodology in Gaining Public Support
By Kim Kenney
Credibility has long been the Holy
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
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
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
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
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
The CDC and DePaul estimates of CFIDS
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
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
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
The CFIDS Association of America.
problems common in CFIDS research
Small numbers of patients/controls
Inconsistent application of case
Poorly selected and/or defined control
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