AHMF: MP: CDC Addresses 'Ambiguities' in CFS Definition

CDC Addresses "Ambiguities" in CFS Definition

BMC Health Services Research
Research article
Identification of ambiguities in the 1994 chronic fatigue syndrome
research case definition and recommendations for resolution

[See the full article at Biomedcentral.
Please make special note of the pre-publication history of this paper, including reviews by Susanne Mertz and Niloofar Afari, and Editor's comment available on site.]

William C Reeves, Andrew Lloyd, Suzanne D Vernon, Nancy Klimas, Leonard A Jason, Gijs Bleijenberg, Birgitta Evengard, Peter D White, Rosanne Nisenbaum, Elizabeth R Unger and the International Chronic Fatigue Syndrome Study Group

Abstract

Background: Chronic fatigue syndrome (CFS) is defined by symptoms and disability, has no confirmatory physical signs or characteristic laborarory abnormalities, and the aetiology and pathophysiology remain unknown. Difficulties with accurate case ascertainment contribute to this ignorance.

Methods: Experienced investigators from around the world who are involved in CFS research met for a series of three day workshops in 2000, 2001, 2002 intended to identify the problems in application of the current CFS case definition.

The investigators in each focus group relied on their own clinical and scientifiic knowledge, brainstorming within each group and with all investigators when focus group summaries were presented. Relevant literature was selected and reviewed independent of the workshops. The relevant literature was circulated via the list-serves and resolved as being relevant by group consensus. Focus group reports were analyzed and compiled into the recommendations presented here.

Results: Recommendations for use of the definition, standardization of the classification instruments and study design issues are presented that are intended to improve the precision of case ascertainment. The International CFS Study Group also identified ambiguities associated with exclusionary and comorbid conditions and reviewed the standardized internationally applicable instruments used to measure symptoms, fatigue intensity and associated disability.

Conclusion: This paper provides an approach to guide systematic and hopefully reproducible application of the current case definition so that case ascertainment would be more uniform across sites. Ultimately an operational CFS case definition will need to be based on empirical studies designed to delineate the possibly distinct biological pathways that result in chronic fatigue.

The AHMF Comments

The study comprised 33 researchers ( 9 from the CDC) and 2 consumer representatives both from the CFIDS Association of America

Preliminary Questions.
1.What consumer consultation process was undertaken over the 3 years 2000, 2001, 2002 by K. Kenney and V. Walker representing consumer interests worldwide?

2.What was their consumer input to the deliberations of the study group?

3.Were they satisfied with the use of the SPHERE as a screening instrument for potential participants in research studies of CFS?

4.What background information was supplied with regard to the development of the SPHERE in Australia by Ian Hickie? (Hickie I., Davenport TA, Hadzi-Pavlovic D, et al., "Sphere: A National Depression Project .Development of a simple screening tool for common mental disorders in general practice." MedJAust2001:175 Suppl;S10-S17)

While much has been written of the work of Wessely, White, Chalder, Sharpe (UK), the work of Hickie, Lloyd (Australia) has yet to be examined for its pervasive influence.

This research article demands the closest scrutiny. It will have an international impact on all future ME/CFS research and health care provision.

Critical extracts:
"While intended to apply primarily to the research setting many of our
recommendations will be useful for health care providers (and the patient community they serve) because they suggest standardized instruments to record and to measure the key symptom domains and the disability associated with CFS."

"Standardized instruments" and "symptom domains" sound inocuous enough. But what are they precisely, how were they developed, and how have they been tested?

Under the heading "Definition and Evaluation of Accompanying symptoms" the authors state:

"We recommend that research studies use the SPHERE (discussed below) to query subjects (cases and controls) about the occurrence, duration, and severity of the 8 case defining symptoms and other potentially accompanying symptoms."

"We recommend that investigators use the Somatic and Psychological Health Report (SPHERE) as a screening instrument for potential participants in research studies of CFS."

"The SPHERE allows for concurrent measurement of depression, anxiety, somatization disorder and fatigue as independent constructs, hence its utility as a screening instrument in studies of CFS."

The SPHERE program was developed by Ian Hickie in1997 to assist medical practitioners to identify mental illness, using a questionaire filled out in the doctor's waiting room. A study of 46,000 patients screened with the SPHERE led to the headline in the Sydney Morning Herald 16 July 2001, "Six in 10 GP patients have mental illness: study".

The SPHERE has been a significant part of the government "Better Outcomes for Mental Health Initiatives". The 34 questions of the SPHERE are based on the General Health Questionaire, the DSM Somatisation criteria check list, symptoms of fatigue and anergy and neurasthenia checklist. Symptoms include fainting, fevers, diarrhoea, nausea, dizziness, weak muscles, headaches, sore throats, and joint pain.

According to the cluster of symptoms the SPHERE can be used as a screening instrument for the diagnosis of anxiety/depression, somatisation (fibromyalgia or irritable bowel subtypes) or neurasthenia. It can be also used to assess severity.

Ian Hickie was chief author of Psychological Medicine, 1999, 29, 259-268 "Unique genetic and environmental determinants of prolonged fatigue study: a twin study". It concluded in part:

"This study supports the aetiological independence of prolonged fatigue and, therefore, argues strongly for its inclusion in classification systems in psychiatry".

Ian Hickie, Andrew Lloyd and Dennis Wakefield (Australia) have published extensively in CFS. A review of their publications 1988-2003 will demonstrate shifting attributions, blurring of terminology and close allignment to the work of Wessely et al. These researchers have strenuously projected views that the patient's belief system prolongs the illness, and those with fevers, sweats, recurrent diarrhoea, episodic loss of vision, and difficulty swallowing foods, have clinical characteristics of somatoform disorder. As co-authors of Psychological Medicine, 1995,25,925-935, their publication "Can the chronic fatigue syndrome be defined by distinct clinical features?" clarifies this position.

Ian Hickie, Andrew Lloyd and Dennis Wakefield were awarded AUD958,289 per year in 2001 by the Department of Health and Human Services, Centers for Disease Control and Prevention-CDC for a period of up to 3 years. (Program Announcement 01024 Billing code 4163-18-P Notice of Availability of funds).

The conclusions of Hickie, Lloyd, Wakefield about patients' illness beliefs and somatising in the face of stengthening biological evidence are at best speculative hypotheses, and at worst lead to denial of appropriate care and great harm.

Chris Hunter