A review and meta-synthesis of qualitative studies on myalgic encephalomyelitis/chronic fatigue syndrome

Abstract:

OBJECTIVE: To review and synthesize findings across qualitative studies on Myalgic Encephalomyelitis/chronic fatigue syndrome (ME/CFS).

METHODS: Articles were systematically reviewed and analyzed within a meta-analytic framework. Analyses included a multi-perspective examination of ME/CFS, as well as a comparative analysis of ME/CFS versus other chronic conditions.

RESULTS: Thirty-four qualitative studies on ME/CFS were included. Findings include three substantive thematic areas that focus on: (1) experiences of people with ME/CFS, (2) experiences of physicians, and (3) themes that intersect both of these groups. For patients, illness development influenced identity, reductions in functioning, and coping. Physician-specific themes described lack of awareness about ME/CFS and recommended improvement in educational resources. Intersecting themes expressed issues with diagnosis creating tensions and fueling the stigmatization of ME/CFS.

CONCLUSIONS: Findings indicate multilayered, context-specific experiences and ways in which both people with ME/CFS, as well as those involved in their lives (e.g., family or the medical community), interpret this illness. Future qualitative studies should recognize the various facets of the ME/CFS experience, the network members of people with ME/CFS, and the sociocultural environment through which the illness is understood.

PRACTICE IMPLICATIONS: Health care professionals can gain unique insight from patient experiences, allowing for more accurate diagnoses and treatment recommendations.

Copyright © 2011 Elsevier Ireland Ltd. All rights reserved.

 

Source: Anderson VR, Jason LA, Hlavaty LE, Porter N, Cudia J. A review and meta-synthesis of qualitative studies on myalgic encephalomyelitis/chronic fatigue syndrome. Patient Educ Couns. 2012 Feb;86(2):147-55. doi: 10.1016/j.pec.2011.04.016. Epub 2011 May 14. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3229648/ (Full article)

 

XMRV, prostate cancer and chronic fatigue syndrome

Abstract:

BACKGROUND: A new retrovirus, xenotropic murine leukaemia virus-related virus (XMRV), was identified in 2006 and an association was claimed between it and a genetic polymorphism predisposing to cancer of the prostate. In 2009 the same virus was identified in a cohort of patients with chronic fatigue syndrome (CFS). In 2010 a second related virus was identified in a separate group of CFS patients. A series of studies from disparate geographical areas have failed to substantiate this work. Most recently several papers have suggested that the detection of these viruses was explained by laboratory contamination.

SOURCES OF DATA: All papers including the wording XMRV were abstracted from the NIH library of medicine database and included in the analysis.

AREAS OF AGREEMENT: XMRV is a newly described retrovirus whose nucleic acid has been identified in samples from patients with both prostate cancer and CFS.

AREAS OF CONTROVERSY: Opinions differ as to whether the detected nucleic acid indicates infection with this virus in this disease or whether laboratory contamination of samples accounts for its presence.

GROWING POINTS: An increasing number of papers now refute the association of XMRV with human disease in humans although there is some evidence of serological reactivity to the virus. While it is unlikely that XMRV is a major cause of either prostate cancer or CFS, it can infect human cells and might yet have a role in human disease.

AREAS TIMELY FOR DEVELOPING RESEARCH: Further studies to either prove or disprove the disease association of the virus are ongoing.

 

Source: Kenyon JC, Lever AM. XMRV, prostate cancer and chronic fatigue syndrome. Br Med Bull. 2011;98:61-74. doi: 10.1093/bmb/ldr010. Epub 2011 May 6. https://www.ncbi.nlm.nih.gov/pubmed/21551158

 

Chronic fatigue syndrome–a neuroimmunological model

Abstract:

The aetiological and pathophysiological basis of chronic fatigue syndrome (CFS) remains a controversial field of inquiry in the research community. While CFS and similar disease conditions such as fibromyalgia (FM) and post-infectious encephalopathy have been the focus of intense scrutiny for the past 20 years, results of research were often contradictory and a cohesive pathological model has remained elusive. However, recent developments in understanding the unique immunophysiology of the brain may provide important clues for the development of a truly comprehensive explanation of the pathology of CFS.

We argue that CFS pathogenesis lies in the influence of peripheral inflammatory events on the brain and the unique immunophysiology of the central nervous system. There is also evidence that CFS patients have a relative immunodeficiency that predisposes to poor early control of infection that leads to chronic inflammatory responses to infectious insults.

The neurological and endocrine changes have been described in CFS patients support the view that CFS has an inflammatory pathogenesis when considered as a whole. An inflammatory model of disease also provides an explanation for the marked female sex bias associated with CFS.

This review therefore posits the hypothesis that CFS as a disease of long-term inflammatory processes of the brain. We will also provide an investigative framework that could be used to justify the use of anti-TNF biological agents as a reliable and effective treatment approach to CFS, a syndrome that to date remains frustratingly difficult for both patients and health care professionals to manage.

Copyright © 2011 Elsevier Ltd. All rights reserved.

 

Source: Arnett SV, Alleva LM, Korossy-Horwood R, Clark IA. Chronic fatigue syndrome–a neuroimmunological model. Med Hypotheses. 2011 Jul;77(1):77-83. doi: 10.1016/j.mehy.2011.03.030. Epub 2011 Apr 6. https://www.ncbi.nlm.nih.gov/pubmed/21474251

 

Chronic fatigue syndrome/myalgic encephalomyelitis: an update

Abstract:

Chronic Fatigue Syndrome (CFS), also referred to as Myalgic Encephalomyelitis (ME), is a disease of unknown origin. It is classified as Post Viral Fatigue Syndrome (PVFS) in the WHO International Classification of Diseases (ICD) and listed as sub-category at G93.3 under chapter G93, other disorders of the brain. ME/CFS is primarily an endemic disorder but occurs in both epidemic and sporadic forms. It affects all racial-ethnic groups and is seen in all socioeconomic strata. A diagnosis of CFS is a diagnosis of exclusion, meaning other medical conditions, including psychiatric disorders, must be first ruled out. CFS is diagnosed if there is no other explanation for the fatigue and if the other symptoms did not develop before the fatigue. The estimated worldwide prevalence of CFS is 0.4?1 percent. The disease predominantly affects young adults, with a peak age of onset of between 20 and 40 years, and women, with a female to male ratio of 6:1. Mean illness duration ranges from 3 to 9 years.

The patho-physiological mechanism of CFS is unclear but the immunological pattern of CFS patients gleaned from various studies indicates that the immune system is chronically activated. Besides the role of environmental insults (xenobiotics, infectious agents, stress) the genetic features of patients are studied to evaluate their role in triggering the pathology. At present there are no specific pharmacological therapies to treat the disease but a variety of therapeutic approaches have been described as benefiting patients. Treatment programs are directed at relief of symptoms, with the goal of the patient regaining some level of preexisting function and well-being.

 

Source: Capelli E, Zola R, Lorusso L, Venturini L, Sardi F, Ricevuti G. Chronic fatigue syndrome/myalgic encephalomyelitis: an update. Int J Immunopathol Pharmacol. 2010 Oct-Dec;23(4):981-9. https://www.ncbi.nlm.nih.gov/pubmed/21244747

 

Tired of being inactive: a systematic literature review of physical activity, physiological exercise capacity and muscle strength in patients with chronic fatigue syndrome

Abstract:

A systematic review was undertaken to examine whether patients with chronic fatigue syndrome (CFS) differ from healthy sedentary controls in physiological exercise capacity, physical activity level and muscle strength. From the available literature, it can be concluded that patients with CFS perform less physical activity during daily life, and have less peak isometric muscle strength compared to healthy sedentary control subjects. Conflicting data in relation to physiological exercise capacity of patients with CFS have been reported, but the weighted available evidence points towards a reduced physiological exercise capacity in CFS. Future studies should use a wash-out period for medication use, blinded assessments, a priori power calculation and a sedentary control group comparable for age, gender, body weight, body length and current physical activity level.

 

Source: Nijs J, Aelbrecht S, Meeus M, Van Oosterwijck J, Zinzen E, Clarys P. Tired of being inactive: a systematic literature review of physical activity, physiological exercise capacity and muscle strength in patients with chronic fatigue syndrome. Disabil Rehabil. 2011;33(17-18):1493-500. doi: 10.3109/09638288.2010.541543. Epub 2010 Dec 20. https://www.ncbi.nlm.nih.gov/pubmed/21166613

 

A review of the definitional criteria for chronic fatigue syndrome

Abstract:

RATIONALE, AIMS AND OBJECTIVES: The research community has for more than three decades tried to unravel the diagnostic mystery that is Chronic Fatigue Syndrome (CFS). This has resulted in considerable amounts of time and money being invested in attempts aimed at establishing the aetiology and pathogenesis of CFS. All of this investment has produced evidence of an interesting variety of endocrine, immune, infectious, muscular and neurological abnormalities in CFS; however, the cause remains elusive. The absence of a known causative agent or diagnostic test for CFS has resulted in the development of a number of CFS case definitions. As such, the main objectives of this paper are to provide a critical review of the similarities and differences between the varying approaches to CFS case definition. The conflicts and controversies that have emerged as a result of the differing definitional criterion for CFS are highlighted and the potential impact on future research is identified.

METHODS, RESULTS AND CONCLUSIONS: This paper presents a critical review of the most frequently used case definitions in CFS. There are currently five case definitions of CFS; however, the most prominent and widely used of these definitions is the 1994 Centre for Disease Control and Prevention Case Definitions. However, the pre-eminence of this definition over the others has never been substantiated and it has been widely criticized for its lack of specificity. Furthermore, none of the above case definitions have produced evidence to demonstrate their accuracy or precision at defining cases of CFS. A summary description of the symptom profile included in each of the case definitions is provided. The inconsistencies that have emerged in CFS research as a consequence of differing approaches to case definition are also highlighted and discussed.

© 2010 Blackwell Publishing Ltd.

 

Source: Christley Y, Duffy T, Martin CR. A review of the definitional criteria for chronic fatigue syndrome. J Eval Clin Pract. 2012 Feb;18(1):25-31. doi: 10.1111/j.1365-2753.2010.01512.x. Epub 2010 Oct 4. https://www.ncbi.nlm.nih.gov/pubmed/21029269

 

The HPA axis in the pathogenesis of chronic fatigue syndrome

Abstract:

Chronic fatigue syndrome (CFS) is a clinical syndrome characterized by profound disabling chronic fatigue associated with a wide array of other physical symptoms. Its etiology is currently unknown. Among the various hypotheses, considerable interest has been placed in the hypothalamus-pituitary-adrenal axis as a possible target of the pathogenesis of CFS. This article reviews the available scientific evidence about a role of hypothalamic-pituitary-adrenal axis in the pathogenesis of chronic fatigue syndrome.

 

Source: Ursini F, Succurro E, Grembiale A, Gagliardi DA, Arturi F. The HPA axis in the pathogenesis of chronic fatigue syndrome. Clin Ter. 2010;161(5):461-4. [Article in Italian] https://www.ncbi.nlm.nih.gov/pubmed/20949245

 

Daily physical activity of patients with the chronic fatigue syndrome: a systematic review

Abstract:

OBJECTIVE: To give an overview of the physical activity level of patients with chronic fatigue syndrome in comparison with asymptomatic controls.

DATA SOURCES: MEDLINE, Web of Science, EMBASE, PsycINFO, Picarta, the Cochrane Controlled Trial Register that is included in the Cochrane Library and reference tracking.

REVIEW METHODS: A systematic literature search was conducted focusing on studies concerning physical activity levels of patients with chronic fatigue syndrome compared to controls. A meta-analysis was performed to pool data of the studies.

RESULTS: Seventeen studies were included with 22 different comparisons between patients with chronic fatigue syndrome and controls. Fourteen studies, including 18 comparisons, showed lower physical activity levels in patients with chronic fatigue syndrome as compared to controls. Four studies, including four comparisons, showed no differences between both groups. The meta-analysis included seven studies and showed a daily physical activity level in patients with chronic fatigue syndrome of only 68% of the physical activity level observed in control subjects. The pooled mean coefficient of variation in patients with chronic fatigue syndrome was higher as compared to control subjects (34.3% versus 31.5%), but this difference did not reach significance.

CONCLUSION: Patients with chronic fatigue syndrome appear to be less physically active compared with asymptomatic controls. There is no difference in variation of physical activity levels between patients with chronic fatigue syndrome and healthy control subjects, but the validity and reliability of some methods of measuring physical activity is questionable or unknown.

 

Source: Evering RM, van Weering MG, Groothuis-Oudshoorn KC, Vollenbroek-Hutten MM. Daily physical activity of patients with the chronic fatigue syndrome: a systematic review. Clin Rehabil. 2011 Feb;25(2):112-33. doi: 10.1177/0269215510380831. Epub 2010 Oct 13. https://www.ncbi.nlm.nih.gov/pubmed/20943713

 

Review part 2: Human herpesvirus-6 in central nervous system diseases

Chronic fatigue syndrome (CFS) is a debilitating chronic illness [Fukuda et al., 1994] that often begins suddenly with a “flu-like” illness. Patients with CFS have great functional impairment [Komaroff et al., 1996]. The cost to the U.S. economy from lost productivity alone (not including medical care costs) is $9 billion annually [Reynolds et al., 2004].

While the pathogenesis of CFS is unknown, there is abundant evidence of an underlying biological process. In comparison to various health and disease control groups, patients with CFS have abnormal findings in the CNS and autonomic nervous system, evidence of chronic activation of various parts of the immune system, and disordered energy metabolism.

CNS abnormalities have been found using MRI [Buchwald et al., 1992; Schwartz et al., 1994a; Lange et al., 2001; de Lange et al., 2005], functional MRI [Tanaka et al., 2006], SPECT [Schwartz et al., 1994b; Schmaling et al., 2003], and positron-emission tomography (PET) [Yamamoto et al., 2004]. Neuroendocrine studies reveal hypofunction of corticotropin releasing hormone (CRH) neurons in the hypothalamus [Demitrack et al., 1991], disruption of both serotonergic and noradrenergic hypothalamic pathways [Demitrack et al., 1992; Cleare et al., 1995], and of growth hormone secretion [Moorkens et al., 2000]. Typically, these abnormalities are in patterns opposite to those seen in major depression. Cognitive testing has revealed abnormalities [Tiersky et al., 1997; Daly et al., 2001; Deluca et al., 2004] that are not explained by concomitant mood disorders [Marcel et al., 1996]. Autonomic nervous system testing has found abnormalities—particularly postural orthostatic tachycardia syndrome, neurally mediated hypotension, and heart rate variability during head-up tilt testing [Bou-Holaigah et al., 1995; Freeman and Komaroff, 1997; Stewart, 2000; Naschitz et al., 2002].

The immunological findings described most commonly in CFS are impaired function of natural killer cells, increased numbers of CD8+ cytotoxic T cells that bear antigenic markers of activation on their cell surface, and increased production of various pro-inflammatory and TH2 cytokines [Komaroff, 2006]. Many of these cytokines can produce symptoms characteristic of CFS: fatigue, fevers, adenopathy, myalgias, arthralgias, sleep disorders, cognitive impairment, and mood disorders.

Many recent studies of patients with CFS have identified disorders of energy metabolism [Myhill et al., 2009], increased allostatic load [Maloney et al., 2009], and increased oxidative and nitrosative stress [Maes and Leunis, 2008].

Cases of CFS can follow in the wake of well-documented infection with several infectious agents, and may be more likely when the symptoms of acute infection were most severe [Hickie et al., 2006]. The first large study on the possible role of HHV-6 in CFS included 259 patients with a “CFS-like” illness (the case definition had not yet been developed) and age- and gender-matched healthy control subjects. Primary culture of lymphocytes showed active replication of HHV-6 in 70% of the patients versus 20% of the control subjects (P < 10 −8) [Buchwald et al., 1992].

Some subsequent studies have employed only serological techniques that do not distinguish active from latent infection. The results have been mixed: a slight preponderance has showed an association between CFS and HHV-6 infection [Ablashi et al., 2000; Reeves et al., 2000; Hickie et al., 2006].

In contrast, other studies have employed assays that can detect active infection: PCR of serum or plasma, IgM early antigen antibodies, and primary cell culture. Most of these studies have shown an association between CFS and active HHV-6 infection [Patnaik et al., 1995; Secchiero et al., 1995; Wagner et al., 1996; Zorzenon et al., 1996; Ablashi et al., 2000; Nicolson et al., 2003], whereas a few have not [Koelle et al., 2002; Reeves et al., 2000]. The number of patients in the studies that have found an association between CFS and active HHV-6 infection (N = 717) is much larger than the number in studies that have failed to find an association (N = 48).

Several observations, summarized above, together suggest that active infection with HHV-6 may cause some cases of CFS. First, active infection with HHV-6 is present in a substantial fraction of patients with CFS. Second, HHV-6 is tropic for the nervous system and immune system cells, and CFS is characterized by neurological and immunological abnormalities. Clinical studies with antiviral drugs that have in vitro activity against HHV-6 could provide strong evidence in favor of, or against, the hypothesis that HHV-6 may trigger and perpetuate some cases of CFS.

You can read the full article here: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4758195/

 

Source: Yao K, Crawford JR, Komaroff AL, Ablashi DV, Jacobson S. Review part 2: Human herpesvirus-6 in central nervous system diseases.J Med Virol. 2010 Oct;82(10):1669-78. doi: 10.1002/jmv.21861. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4758195/ (Full article)

 

The genetics and epigenetics of fatigue

Abstract:

Fatigue is a common symptom and includes both physical and mental components. It can be associated with a variety of different syndromes and diseases, but in many cases is not associated with other comorbid conditions. Most humans have experienced acute fatigue in relation to different stressors. Acute fatigue typically decreases as the effect of the triggering factor is reduced and a normal homeostatic balance is restored. Fatigue that persists for 6 months or more is termed chronic fatigue. Chronic fatigue (CF) in combination with a minimum of 4 of 8 symptoms and the absence of diseases that could explain these symptoms, constitute the case definition for chronic fatigue syndrome. In spite of its prevalence, the biology of fatigue is relatively poorly understood and biological markers have not yet been identified.

This literature search was performed in PubMed to identify research on the genetics and epigenetics of fatigue. Publications were included if fatigue was a major topic and the topic was combined with genetic and/or epigenetic measurements in adult humans. A total of 40 publications were identified.

Although altered functioning in the hypothalamic-pituitary-adrenal axis, the serotonergic system, and associations with infectious agents have been identified, the search for genetic or epigenetic markers of fatigue, either in the context of CF or chronic fatigue syndrome (CFS) has been relatively unproductive or, in the case of epigenetics, nonexistent. Although several studies, both hypothesis-testing and hypothesis-generating, have been performed to search for biomarkers, they have mostly been underpowered, restricted by the heterogeneity of the phenotype, or limited by an unsystematic study design.

To be able to confirm the hypothesis that risk for, or levels of, fatigue are influenced by the genetic or epigenetic background of an individual, studies need to be based on larger sample sizes with a more clearly defined phenotype. Studies need to focus not only on the influence of a single aspect such as single nucleotide polymorphisms (SNPs) or differential gene expression on disease risk or state, but also on the systems biology behind the disease in combination with information on environmental influences and validation of findings in functional studies.

Copyright (c) 2010 American Academy of Physical Medicine and Rehabilitation. Published by Elsevier Inc. All rights reserved.

 

Source: Landmark-Høyvik H, Reinertsen KV, Loge JH, Kristensen VN, Dumeaux V, Fosså SD, Børresen-Dale AL, Edvardsen H. The genetics and epigenetics of fatigue. PM R. 2010 May;2(5):456-65. doi: 10.1016/j.pmrj.2010.04.003. https://www.ncbi.nlm.nih.gov/pubmed/20656628