Shaky Foundation - Part 2
By Craig Maupin at www.cfidsreport.com
Different illnesses yet the same name? - Differences in CFS Research
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” (CFS) is the best way to illustrate the confusing web that has been woven.
The 1994 case definition was intended to broaden CFS. The 1994 case definition is so broad, so non-distinct, 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 chronic fatigue syndrome.
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 this. The case definition for chronic fatigue sydnrome was far less effective in finding answers to the mysteries of CFS than it was in providing a medical and political home to those who suffer from various, unrelated 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 controversial and divisive. The patients who now call the CFS community home are very different, so different that it begs for answers to some important questions. Is mixing these patients effective scientific protocol? And, why are they being diagnosed with the same illness to begin with?
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 Sea Biscuit. 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 being severely disabled by chronic fatigue syndrome. 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, Hillenbrand has found no treatment that addresses her symptoms, and she 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 Seabiscuit, 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," she 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”
Hillenbrand describes a viral onset, 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 to find out.
Patient 2- Fibromyalgia Syndrome - Other patients, also disabled, now call the chronic fatigue syndrome (CFS) community home. Yet, these patients paint a very different picture of CFS. They may not have the differing hallmark post-exertional response experienced by Hillenbrand. The following describes a patient whose symptoms and clinical expression is very different than Hillenbrand. Her most prevalent symptom is a distinct and disabling form of pain or myalgia (muscle pain). Unlike Hillenbrand, this person claims to have received measured benefits from increasing her activity, saying that it has alleviated both 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 chronic fatigue syndrome 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” or often CFS/FM. Many of these patients prefer names for the illness such the popular term CFS/FM. These patients also fit the CDC case definition for chronic fatigue syndrome CFS. And, as you will see, the differences dont' stop there.
Patient 3- Post-traumatic stress disorder - Finally, we have a patient who was also diagnosed with chronic fatigue syndrome, 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 this: “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 to “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 advise 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 her M.E and Chronic Fatigue Syndrome that consists of treating her 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. This patient says patients like Hillenbrand make her “sad and angry”. She also states that charities for chronic fatigue syndrome and M.E are creating a “conspiracy of ignorance” and are responsible for keeping patients like Hillenbrand “remaining ill unnecessarily”. Her views are often repeated by psychiatrists researching “fatiguing syndromes” in the U.K.
While this patient has a wider variety of symptoms, her symptoms were less distinct than the two patients above. She cites stress and psychological trauma, not a viral episode, as the precipitator of her illness. The parameters of her case also may fit 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 (since she has many less-distinct symptoms, perhaps even more so than the severely-disabled Hillenbrand).
Despite extreme clinical differences, all of the above patients meet the CDC’s revised case definition for chronic fatigue syndrome (CFS). Each patient can be combined 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 "integrated" 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 chronic fatigue syndrome (CFS) community responding differently to different treatments and describe very different symptom sets. Despite this, it is difficult to find CFS researchers who will publicly acknowledge this problem. 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 the broad, integrative case definition.
The "integrative" CFS case definition
If the patient community for chronic fatigue syndrome (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 published CFS 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 (low molecular weight 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 DeMeirleir. Researchers in Belgium have observed that their subjects 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. Their research subjects may fit the symptom pattern of Sea Biscuit author, Laura Hillenbrand.
However, at a CFSCRC research center in Seattle a different picture of chronic fatigue syndrome is emerging. Dedra Buchwald has found the immune defects such as the defects observed by Suhadolnik to be less significant and very limited in her research samples, certainly far short of pathological relevance. She forwards a model for CFS that is brain central and very similar to Fibromyalgia. Unlike DeMeirleir and Suhadolnik, Buchwald assert that many of their study participants have been improved by graded exercise. Buchwald's Center has not found any impaired exercise capacity on exercise ergometry tests. She has been one of the most vocal supporters of a broader definition and “integrative” approach to researching CFS. She also has supported a clinical approach that blends and merges Fibromyalgia and CFS samples in research.
Researchers at Buchwald's CFS research center stated that in 2003, they will release results of their research that claim CFS is a disease of pain/sensory amplification. The model for CFS proposed by these researchers is a remarkably similar model to the a widely-held model for Fibromyalgia Syndrome, a prevalently disabling and fatiguing illness with millions of sufferers.
Buchwald's descriptions of chronic fatigue syndrome suggest that she, like many researchers, could be researching Fibromyalgia under the "integrative" CFS research rubric. . Her center tends to downplay flu-like symptoms and exercise intolerance of CFS, and she seems to prefer that myalgia and brain-centered symptoms (central processing, HPA axis, and neuroendocrine) be emphasized. Her research samples often look more like patient number 2 (above).
Finally, bringing up the rear, are a group of controversial researchers who employ a wholly psychiatric model to explain CFS. Because of their defense of the controversial “integrative” research methods utilized by UK psychiatrist Simon Wessely, these psychiatrists are often referred to as the Wessely School. The Wessely School’s research samples typically look like patient 3 above (post traumatic stress or post trauma avoidance), and their subjects often find complete cures for their fatigue from behavioral and mental modification, typically exercise. These researchers emphasize CFS as a predominantly women's illness, catastrophizing and poor stress adaptation being key features.
There are questions as to whether Wessely is simply using the CFS funding structure to fund post-traumatic stress disorder (PTSD) research, a disorder already defined before CFS. According to Wessely, most of his research subjects have been effectively cured through simple exercise and counseling, dispensed to correct what he claims are ‘avoidance behaviors and attitudes’.
Wessely and his followers feel that clean distinctions and subgroups for emerging illnesses like CFS 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 by individual separated by clinical differences can lead to a “risk of overinvestigation” and increase the “potential for iatrogenic harm”. Focus on gender is a key componment in this research.
Other researchers who are strong proponent of FM integration into CFS research, such as Buchwald, Natelson, and Mohammed Yunus, have often cheered this Wessely's “integrative” view toward research methods, saying it is powerful, innovative and having different fatiguing illnesses in research samples leads to better scientific outcomes. Wessely's view that strong clinical standards and distinctions can be counterproductive to research is very popular among CFS researchers, even though his conclusions toward CFS may remain more controversial and less accepted.
Funds, special interests, and sloppy research methods
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 chronic fatigue syndrome has contributed to a loss of respect, opened a Pandora’s box of conflicting results, and created confusion about what defines CFS. Things probably won't improve much when the CDC will base a new "empirical" definition on clustering from such diverse samples. The confusion will simply continue.
Today, interpreting CFS research is difficult. Sadly, many researchers stringently support keeping the definition for CFS broad, loosely defined, and with very few standards, all the while whispering about the need for "reform". Could it be that they are aware that their bread is buttered by continuation of the broader definition? In other words, do they need the broader definition to obtain funding for a broad array of personal interests and projects? Clinical distinctions have always formed a foundation for medical research. CFS research seems to support ignoring such distinctions.
The broad definition keeps no one perfectly happy, but the keep funds rolling in for a wide variety of interests and needs, leading to a comfortable satiation. One researcher interested in reforming chronic fatigue syndrome 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 create a new "empirical definition" for chronic fatigue syndrome (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" and "symptoms".
However, one issue was not discussed. Is this new integrative “science” championed by the CFS research community actually based on reliable and time-tested scientific methodology? Or, are trendy funding needs and differing political desires causing CFS researchers to advocates broad, loose, integrative research methods?
One thing is for sure. The broad “integrative” approach to CFS research carved out by the CDC has not been effective. Contrary to the claims of the revised CDC case definition, the integration of differing fatiguing illnesses into the CFS research stream has not produced a “clarification” of the disease. Years after the broadened definition, CFS research results are conflicting,and contested.
Yet, not only does the CFS research community still bravely utilize broad loose definition for CFS, they have created a whole new set of rationalizations to prop it up. New questionaires, new complex tests, and a new definition have been created by an Intentional CFS Working Group in 2001. However, producing reform? To rebuild a reliable foundation for CFS research, the greater scientific community will have to reexamine the philosophies and ideologies behind their loose, integrative approach. It is these philosophies that are at the root of the shaky ideological foundation for CFS research. Integration is the "Shaky Foundation".
A new group of courageous scientists and researchers who are willing to use clinically distinct research subjects and unique biomarkers to separate subgroups of people will have to lead the charge for reform. Until this happens, what is touted as "reform"will only be the same old system