A Shaky Foundation: Different Illnesses Yet the Same Name? Differences in the Chronic Fatigue Syndrome Community Today
March 10, 2003
By Craig Maupin at www.cfidsreport.com
It isn’t difficult to see the problems with the 1994 CDC revised case definition. In fact, a quick look at some of the discrete differences in those who now are diagnosed with “Chronic Fatigue Syndrome” is the best way to illustrate the confusing web that has been woven. For emerging illnesses such as Chronic Fatigue Syndrome (CFS), quality medical care and research will start with clear distinctions. But unfortunately for those with CFS, it is those distinctions that the CFS research community often seems to want to blur, downplay, and integrate.
Since the 1994, case definition was intended to broaden CFS, this comes as no surprise. The 1994 case definition is so broad, so nondistinct, that it has changed the face of the CFS community – creating a diverse representation of many differing illnesses. There are many patients whose symptoms appear so different yet all are diagnosed as CFS. This begs the question of both how and why the CFS research community is encouraging combining them in research and treatment.
So far the CFS research community has kept its cards to its vest, rarely taking pains to justify the fast and loose research and diagnostic standards they have formulated to govern themselves. Many CFS researchers claim that the conflicting results are proof that CFS could have many causes and symptoms - that it is not distinct. Phrases like “heterogeneous disorder”, “symptom overlap”, and “co-morbidity” are the trendy phrases of the day. CFS researchers toss these phrases out frequently, trying to justify a research pattern that blurs distinctions, has led to conflicting results, is uninterpretable, and has achieved little respect.
The truth is, the case definition was far less effective in finding answers to the mysteries of CFS than in providing a medical and political home to those who suffer from various emerging illnesses. And, for those crafting this definition, with their own differing ideologies and political needs, that may have been the point.
By taking a look at some of the different patients who now fit the 1994 CDC revised case definition for CFS, one can see why CFS research is so controversial and divisive. The patients who now call the CFS community home are so different that it begs some important questions. Is mixing these patients effective scientific protocol? And, why are they being diagnosed with the same illness to begin with? Is the CFS research community encouraging illnesses that already had previous definitions to be integrated into CFS research samples? And, if so, why would they do this?
Clear differences – Different patients diagnosed with Chronic Fatigue Syndrome
Patient 1 - Post-viral/infective flu-like illness
In 2001, PWC and author Laura Hillenbrand released a successful book on the legendary racehorse Seabiscuit. The book eventually climbed the New York Times bestseller list. While promoting the book, Hillenbrand spoke frequently about what it was like to write the book while severely disabled by CFS. She painted an intense and vivid picture of the CFS hallmark symptom of post-exertion fatigue. Often bedridden, Hillenbrand spoke of having to ration the amount of effort each day that she could apply to writing her book. Activity and exercise are activities that she must pay a price to participate in. Her recovery time for even a simple shower was often as much as three hours.
To date, she has found little that seems effective in treating her symptoms, and has simply succumbed to treating her illness by pacing herself and restructuring her life to live within her limits. She had to sacrifice to write her book, giving up for a time treasured activities. Despite the sacrifice, it gave her a sense of accomplishment. She has decided to learn as best as she can how to adjust to her illness after trying a host of treatments unsuccessfully.
"For the first few years I tried everything, and hoped for a possible cure," Hillenbrand says. "Nothing helped, and a lot of things set me back. Emotionally, you put a big stake in everything you try. And it hurts every time a treatment doesn't work." The picture Hillenbrand painted of CFS in interviews is very similar to what was seen in the 1980’s outbreaks at Incline Village and Lyndonville that gave birth to what has become the broader CFS community. She described severe fatigue, postexertional exhaustion, nausea, flu-like symptoms, and neurological problems such as vision difficulties. So, does Hillenbrand have CFS? Yes. She fits the case definition set up by the CDC. Does she have the same illness as everyone else who meets the 1994 case definition? Read on.
Patient 2 - Fibromyalgia Syndrome
Other patients, also disabled, now call the CFS community home and paint a very different picture of CFS. These patients may not have the differing hallmark post-exertional response of CFS, but still are being diagnosed with CFS. Below, is a patient whose symptoms and clinical expression is very different than Hillenbrand, both in hallmark symptoms and in her response to treatments. Her most prevalent symptom is a distinct and disabling form of pain or myalgia. Unlike Hillenbrand, she claims to have received measured benefits from increasing her activity, saying that it has alleviated her pain and general symptoms.
Here she describes her clinical improvement from her CFS/FM following exercise. "Regular exercise benefits everyone, but especially those with chronic pain conditions. Muscles that are well conditioned and well toned can reduce chronic pain". She goes on to say that if you are diagnosed with both CFS and fibromyalgia you "should exercise regularly, and gradually increase your duration and frequency."
Another person diagnosed with CFS and fibromyalgia adds similar sentiments, "I have to exercise, and "My muscles were withering and hurting until I began doing so...it definitely helps me."
These patients’ main symptoms are not exercise activity intolerance or flu-like symptoms, but myalgia, or muscle pain. Yet, like Hillenbrand, they are diagnosed as having "chronic fatigue syndrome." Many of these patients prefer names for the illness such the popular term CFS/FM. These patients also fit the CDC case definition for CFS.
Patient 3 - Post-traumatic Stress Disorder
Finally, we have a patient who was also diagnosed with CFS, and has been cured by psychological counseling and exercise. This patient describes her main clinical feature as a phobia and avoidance of activity following a traumatic and acute illness. She also views her illness as a psychological response to a stressful event, which eventually led to physical deconditioning.
In an article in British magazine Mail on Sunday, the patient’s mother describes her illness as: "She had fainting fits, and woke up every day feeling exhausted." She also described her illness as "feeling giddy" and "terrible all the time," along with headaches, dizziness, and muscle aches. Her physician advised her that, "the best way to recover" was "by doing a little more each day" and, according to the patient, diagnosed her with M.E, or Myalgia Encephalitis.
Eventually, according to this patient, her doctor's advice led to a physical and emotional turnaround that she describes as a cure. She was referred to psychiatrists at King’s College. She is resolute that psychiatrists there have found a cure for M.E and Chronic Fatigue Syndrome that consists of treating patients with gradual increases in exercise and psychological counseling for perpetuating and avoidance behaviors. She also generally feels that patients who are not cured through gradual exercise such as Hillenbrand are rejecting a cure that is already available to them and this behavior makes her "sad and angry." She states that charities for CFS and M.E are creating a "conspiracy of ignorance" and are responsible for keeping patients like Hillenbrand "remaining ill unnecessarily." Her view toward PWC's like Hillenbrand is often repeated by psychiatrists researching "fatiguing syndromes" in the U.K.
While this patient had perhaps a wider variety of symptoms, they were less distinct than the two patients above. She also fits preexisting definitions for post traumatic stress disorder. Since the 1994 case definition weights numbers of symptoms over either severity or distinctness, she has comfortably found a home in the 1994 CDC case definition for CFS (perhaps even more so than the severely disabled Hillenbrand).
All three of these patients are so different that it begs the question of whether it is justifiable to continue treating and researching them as if they had one illness. Despite extreme clinical differences, all of the above patients meet the CDC’s revised case definition for CFS. Each patient can be combined or funded in research samples for CFS, with no requirement for researchers to define or reveal these differences in their research. In fact, the case definition encourages research that integrates these patients claiming, "A comprehensive and integrative approach" toward CFS is "desirable." Most CFS researchers champion this view, and they have crafted a set of new rules and theorems to support it.
Could it be that there are political and financial, rather than pure scientific motives, behind the rationale of including vastly different patients under a broadened nondistinct case definition? It very well could.
Currently, patients in the CFS community are responding differently to different treatments and sometimes describing very different symptoms. Even so, it is difficult to find CFS researchers who will publicly acknowledge this possibility. Instead, a set of porous philosophies has been designed by the CFS research community to put a positive and healthy spin on the conflicting results yielded by broad case definition.
Looking at the state of CFS research – funding different research under a broad definition
If the patient community for CFS looks dissimilar, the research community simply mirrors the differences. Today, researchers receiving government funding for CFS have such diverse ideas, research methods, and models for CFS that it is difficult to interpret CFS published research.
At Temple University, Robert Suhadolnik has found a distinct immune defect in a group of patients that have the hallmark flu-like fatigue and activity intolerance associated with CFS. He named the molecule LMW RNase L. He hasn't found the same defect in patients with fibromyalgia or in healthy controls.
Suhadolnik’s research has been confirmed and extended by other talented microbiologists, such as Belgium's Kenny De Meirleir. Researchers in Belgium have observed that patients with the defect exhale less CO2 during exercise, giving clear evidence that these patients' severe activity intolerance has a microbiological rationale. These researchers paint a flu-like illness of severe fatigue, and they have drawn some clear clinical and microbiological distinctions between their research samples and other fatiguing illnesses. These researchers' patients may fit the symptom pattern of Seabiscuit author Laura Hillenbrand.
However, at research centers under the CFSCRC in New Jersey and Seattle, a different picture of CFS is emerging. Dedra Buchwald and Benjamin Natelson head both centers. Both researchers have found the immune defects in their research samples to be less significant and very limited, certainly far short of pathological relevance, such as the defects observed by Suhadolnik. others have forwarded models for CFS that are brain central and very similar to fibromyalgia. Unlike De Meirleir and Suhadolnik, Buchwald and Natelson assert that many of their study participants have been improved by graded exercise.
Unlike De Meirleir, Buchwald and Natelson’s Centers have not found any impaired exercise capacity on exercise ergometry tests in their research samples. These two researchers have been vocal supporters of a broader definition and an "integrative" approach to researching CFS.
But perhaps more representative of researchers such as Buchwald and Natelson is a strong desire to integrate fibromyalgia or myalgia predominant samples into the CFS research structure. Natelson was instrumental in integrating fibromyalgia into the AACFS (American Association for Chronic Fatigue Syndrome) during his tenure as its president. Buchwald has stated that she has observed few if no clinical differences in the fatigue for FMS and her samples for CFS. Also, her researchers from her Seattle research center have stated that in 2003, they will release results of their research that claim CFS is a disease of pain/sensory amplification. This is a remarkably similar model for Fibromyalgia Syndrome, a prevalently disabling fatiguing illness with millions of sufferers.
Buchwald's descriptions of CFS suggest that their research samples look like the more prevalent fibromyalgia. Her center tends to downplay flu-like symptoms and exercise intolerance, and seems to prefer that myalgia and brain-centered symptoms be emphasized. Her research samples often look more like patient number 2 above.
Finally, bringing up the rear are a class of psychiatrists who use a completely psychiatric model to explain CFS. These psychiatrists are often referred to as the Wessely School, due to their often politically-heated defense of the controversial "integrative" research methods utilized by UK psychiatrist Simon Wessely. The Wessely School's research samples typically look like patient 3 above. There are questions as to whether Wessely is simply using the CFS funding structure to fund PTSD research, which was already defined before CFS. According to Wessely, most of his patients can be effectively cured through exercise and counseling dispensed to correct what are ‘avoidance behaviors and attitudes.’
Wessely and his followers feel that clean distinctions and subgroups for emerging illnesses are actually counterproductive to effective scientific methodology. In a paper published in 1998, Wessely urged researchers to avoid classifying patients according to symptoms, claiming he and his associates in the U.K have discovered novel proof that classifying clinically differing patients is "outdated" and "misguided." He strongly feels that treating and researching patients individually by clinical differences can lead to a "risk of over investigation" and increase the "potential for iatrogenic harm."
Researchers who are strong proponents of FM integration into CFS research, such as Buchwald, Natelson, and Mohammed Yunus, have often cheered this "integrative" view toward research methods as incredibly innovative and even a step forward in scientific research methods. Wessely's view that standards and distinctions can be counterproductive to research is very popular among CFS researchers, even his conclusions toward CFS remain more controversial and less accepted.
Funding, special interests, and sloppy methods...A connection?
Aside from the differences in both the patients and researchers now covered under the 1994 CDC CFS case definition, one thing is for certain. It is obvious that the CFS research community has laid a confusing and shaky foundation for CFS research. The revised case definition for CFS has opened a Pandora’s Box of conflicting results, created confusion about what defines CFS, and has contributed to a loss of respect. But when one looks at the differences in patients and research standards being applied under the 1994 CDC revised case definition, should this come as a surprise? I recall one CFS activist saying that the CDC’s approach to CFS research is "garbage in, garbage out." And, often what the CFS research community is failing to see, is the loss of credibility to CFS research.
Without a previous knowledge of the research methods, philosophies, and viewpoints of given CFS researchers, there is no way to meaningfully interpret CFS research. Sadly, the CFS research community tends to be insulated. So not surprisingly, justifications for the conflicting results are often repeated with little challenge or debate.
Many researchers in the CFS community stringently support keeping the definition for CFS broad, loosely defined, and with very few standards, all the while whispering about the need for "reform." Often, a few words are whispered in support of reform, while quite a few rationalizations are trumpeted in favor of the current system. Could it be that they are aware that their bread is buttered by continuation of the broader definition? In other words, they need the broader definition to obtain funding for a broad array of personal interests and projects.
The broad definition keeps no one perfectly happy, but it keeps funds rolling in for a wide variety of interests and needs, leading to a comfortable satiation. One researcher interested in reforming CFS research standards wisely sums up the prevailing philosophy in the CFS research community, "This situation has resulted in conflicting clinical and laboratory observations that in all likelihood is due to different populations of patients being studied in different centers." This researcher has it correct!
Summing it up
At a recent CDC meeting to determine new approaches to CFS research, participants gave presentations on Gulf War Illness, fibromyalgia, and predefined psychiatric disorders, all under the "fatigue syndrome" rubric.
Some presenters at the conference seemed to strive to illustrate that their research was the true "CFS", using the broad 1994 case definition as their instrument of measurement. Others strived to proselytize the attendees with a newly formulated religion of "one illness", "one cause", many conflicting "results." Is this "science" actually based on trendy funding needs and building political consensus rather than reliable and time-tested methods? If not, what is it based on? Many say it is based on funding. Others say a lot of what is being funded is not quality research, and the support for the low standards are simply needed to allow low quality research to continue.
One thing is for sure, the broad "integrative" approach to CFS research carved out by the CDC has not been effective. And, despite a set of oft-repeated rationalizations, it is not based on effective scientific methods or ideas. Contrary to the claims of the revised CDC case definition, the integration of differing fatiguing illnesses into CFS research is not "clarifying" the disease. Years after the broadened definition, CFS research results have become as conflicting as the definition itself. Should this turn of events really surprise anyone? Not really! It should have been predictable!
Yet, not only does the CFS research community still bravely utilize low standards, they have created a whole new set of rationalizations to prop them up. To rebuild a reliable foundation for CFS research, the greater scientific community will have to reexamine the philosophies and ideologies that are used to do the "propping." It is these philosophies that are at the root of the shaky ideological foundation of CFS research. The rationalizations of the low standards are just as much a hindrance to true reform as the low standards themselves. They are now often habitually repeated by many patients, researchers, and even activists in the CFS community with little or no examination.
In order for CFS research to find itself on more reliable ground, this flimsy philosophy, not just the conflicting results it has produced, will have to be challenged and rethought.
Source: www.cfidsreport.com. Reprinted with permission of the author. (c) The CFIDS Report.