Identification of ambiguities in the 1994 chronic fatigue syndrome research case definition and recommendations for resolution

Stouten, Bart

doi:10.1186/1472-6963-5-37

Cited by 23 publications

(23 citation statements)

References 25 publications

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“…The complexities of CFS case ascertainment based on exclusions and self-reported questionnaires have raised issues central to the pathological identity of CFS. It is suggested that unexplained chronic fatigue, including CFS, is a heterogeneous entity composed of different conditions, with fatigue as the prominent symptom [4][5][6].…”

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confidence: 99%

Polymorphisms in Genes Regulating the HPA Axis Associated with Empirically Delineated Classes of Unexplained Chronic Fatigue

et al. 2006

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Chronic fatigue syndrome (CFS) is characterized by persistent or relapsing fatigue that is not alleviated by rest, causes substantial reduction in activities and is accompanied by a variety of symptoms. Its unknown etiology may reflect that CFS is heterogeneous. Latent class analyses of symptoms and physiological systems were used to delineate subgroups within a population-based sample of fatigued and nonfatigued subjects [1] . This study examined whether genetic differences underlie the individual subgroups of the latent class solution. Polymorphisms in 11 candidate genes related to both hypothalamic-pituitary-adrenal (HPA) axis function and mood-related neurotransmitter systems were evaluated by comparing each of the five ill classes (Class 1, n = 33; Class 3, n = 22; Class 4, n = 22; Class 5, n = 17; Class 6, n = 11) of fatigued subjects with subjects defined as well (Class 2, n = 35). Of the five classes of subjects with unexplained fatigue, three classes were distinguished by gene polymorphsims involved in either HPA axis function or neurotransmitter systems, including proopiomelanocortin (POMC), nuclear receptor subfamily 3, group C, member 1 (NR3C1), monoamine oxidase A (MAOA), monoamine oxidase B (MAOB), and tryptophan hydroxylase 2 (TPH2). These data support the hypothesis that medically unexplained chronic fatigue is heterogeneous and presents preliminary evidence of the genetic mechanisms underlying some of the putative conditions.

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Polymorphisms in Genes Regulating the HPA Axis Associated with Empirically Delineated Classes of Unexplained Chronic Fatigue

et al. 2006

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“…Ceiling effects on patient-reported outcome measures are particularly problematic as they have been identified across many patient populations [22–25]. Ceiling effects, defined as a proportion of patients reporting maximum scores on a given measure more than 40% of the time [23, 25], are particularly concerning for evaluating response to intervention as the range of the patient-reported outcome measure may be too restrictive to capture clinically significant change.…”

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The utility of patient-reported outcome measures among patients with myalgic encephalomyelitis/chronic fatigue syndrome

et al. 2016

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Purpose Debilitating fatigue is a core symptom of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS); however, the utility of patient-reported symptom outcome measures of fatigue for ME/CFS patients is problematic due to ceiling effects and issues with reliability and validity. We sought to evaluate the performance of three patient-reported symptom measures in a sample of ME/CFS patients and matched controls. Methods 240 ME/CFS patients and 88 age, sex, race, and zip code matched controls participated in the study. Participants completed the Multidimensional Fatigue Inventory-20, DePaul Symptom Questionnaire, and RAND SF-36. Results The general and physical fatigue subscales on Multidimensional Fatigue Inventory-20, as well as the role of physical health on the RAND SF-36, demonstrated questionable or unacceptable internal consistency and problematic ceiling effects. The DePaul Symptom Questionnaire demonstrated excellent internal reliability, and less than 5% of participants were at the ceiling on each subscale. The post-exertional malaise subscale on the DePaul Symptom Questionnaire demonstrated excellent clinical utility as it was able to differentiate between ME/CFS patients and controls (OR = 1.23, p < .001), and predict ceiling effects on other patient reported outcome subscales. A score of 20 on the post-exertional malaise subscale of the DePaul Symptom Questionnaire optimally differentiated between patients and controls. Conclusions Significant ceiling effects and concerns with reliability and validity were observed among Multidimensional Fatigue Inventory-20 and RAND SF-36 subscales for ME/CFS patients. The DePaul Symptom Questionnaire addresses a number of concerns typically identified when using patient reported outcome measures with ME/CFS patients; however, an improved multidimensional patient reported outcome tool for measuring ME/CFS-related symptoms is warranted.

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“…Taylor, Jason, and Torres (2000) compared the FSS with the Fatigue Scale (Chalder et al, 1993) and found for a CFS-like group, the FSS was more closely associated than the Fatigue Scale with severity ratings for the eight core CFS symptoms (Fukuda et al, 1994) and a number of functional outcomes. A ceiling effect in the FSS may limit its utility to assess severe fatigue-related disability, and Stouten (2005) has warned that many fatigue scales do not accurately represent the severe fatigue that is uniquely characteristic of CFS.…”

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Fatigue Scales and Chronic Fatigue Syndrome: Issues of Sensitivity and Specificity

Jason¹,

Evans²,

Brown³

et al. 2011

DSQ

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Few studies have explored issues of sensitivity and specificity for using the fatigue construct to identify patients meeting chronic fatigue syndrome (CFS) criteria. In this article, we examine the sensitivity and specificity of several fatigue scales that have attempted to define severe fatigue within CFS. Using Receiver Operating Characteristic (ROC) curve analysis, we found most scales and sub-scales had either significant specificity and/or sensitivity problems. However, the postexertional subscale of the ME/CFS Fatigue Types Questionnaire (Jason, Jessen, et al., 2009) was the most promising in terms of specificity and sensitivity. Among the more traditional fatigue scales, Krupp, LaRocca, Muir-Nash, and Steinberg's (1989) Fatigue Severity Scale had the best ability to differentiate CFS from healthy controls. Selecting questions, scales and cut off points to measure fatigue must be done with extreme care in order to successfully identify CFS cases. Keywordschronic fatigue syndrome; fatigue; fatigue scales; sensitivity and specificity There have been relatively few studies assessing the sensitivity and specificity of fatigue scales which are frequently used to identify individuals with chronic fatigue syndrome (CFS) and differentiate them from healthy controls. The present investigation consists of two distinct studies, both of which employ samples of individuals with CFS and controls, and to assess the effectiveness of several well known fatigue instruments in discriminating between these two groups by utilizing Receiver Operating Characteristic (ROC) curve analyses. The Fukuda et al. (1994) CFS case definition is the currently accepted case definition internationally, although there is no available "gold standard" to assess fatigue severity. This case definition requires an individual to experience six or more months of persisting or recurring chronic fatigue and the co-occurrence of four of eight additional core symptoms. However, these Fukuda et al. requirements have been criticized as lacking operational definitions and guidelines for accurate identification of CFS cases (Jason, King, et al., 1999;Reeves et al., 2003). For example, these criteria do not specify how to assess fatigue severity or the presence of persisting or recurring fatigue for a period of 6 or more months. Partially in response to these problems with operationalizing the Fukuda et al. definition, the Centers for Disease Control and Prevention (CDC) developed an empiric case definition for CFS that involves assessment of symptoms, disability, and fatigue with standardized instruments and specific cutoff points .Correspondence concerning this article should be addressed to Leonard A. Jason, Ph.D., Director, Center for Community Research, 990 W. Fullerton Ave., Suite 3100, Chicago, Il. 60614. telephone: 773-325-2018; fax: 773-325-4923 et al., 2007), rates that are about ten times higher than prior CDC prevalence estimates (Reyes et al., 2003) and estimates of other investigators . It is plausible that this inflated CFS prevalence...

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Identification of ambiguities in the 1994 chronic fatigue syndrome research case definition and recommendations for resolution

Cited by 23 publications

References 25 publications

Polymorphisms in Genes Regulating the HPA Axis Associated with Empirically Delineated Classes of Unexplained Chronic Fatigue

Polymorphisms in Genes Regulating the HPA Axis Associated with Empirically Delineated Classes of Unexplained Chronic Fatigue

The utility of patient-reported outcome measures among patients with myalgic encephalomyelitis/chronic fatigue syndrome

Fatigue Scales and Chronic Fatigue Syndrome: Issues of Sensitivity and Specificity

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