Potential causal factors of CFS/ME: a concise and systematic scoping review of factors researched
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Journal of Translational Medicine Open Access
REVIEW
Potential causal factors of CFS/ME: a concise and systematic scoping review of factors researched Ashley Elizabeth Muller1* , Kari Tveito2, Inger Johanne Bakken3, Signe A. Flottorp1,4, Siri Mjaaland1 and Lillebeth Larun1
Abstract Background: Chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is understood as a complex condition, likely triggered and sustained by an interplay of biological, psychological, and social factors. Little oversight exists of the field of causal research. This systematic scoping review explores potential causal factors of CFS/ME as researched by primary studies. Methods: We searched eight databases for primary studies that examined potential causal factors of CFS/ME. Based on title/abstract review, two researchers independently sorted each study’s factors into nine main categories and 71 subordinate categories, using a system developed with input given during a 2018 ME conference, specialists and representatives from a ME patient advocacy group, and using BMJ Best Practice’s description of CFS/ME etiology. We also extracted data related to study design, size, diagnostic criteria and comparison groups. Results: We included 1161 primary studies published between January 1979 and June 2019. Based on title/abstract analysis, no single causal factor dominated in these studies, and studies reported a mean of 2.73 factors. The four most common factors were: immunological (297 studies), psychological (243), infections (198), and neuroendocrinal (198). The most frequent study designs were case–control studies (894 studies) comparing CFS/ME patients with healthy participants. More than half of the studies (that reported study size in the title/abstract) included 100 or fewer participants. Conclusion: The field of causal hypotheses of CFS/ME is diverse, and we found that the studies examined all the main categories of possible factors that we had defined a priori. Most studies were not designed to adequately explore causality, rather to establish hypotheses. We need larger studies with stronger study designs to gain better knowledge of causal factors of CFS/ME. Keywords: Etiology, Scoping review, Patient involvement, Chronic fatigue syndrome, Myalgic encephalomyelitis Background CFS/ME is a condition characterized by prolonged, significant, and sometimes disabling exhaustion and postexertion malaise, accompanied by symptoms such as *Correspondence: [email protected] 1 Norwegian Institute of Public Health, Skøyen, PO Box 222, 0213 Oslo, Norway Full list of author information is available at the end of the article
generalized pain, sleep disorders, and cognitive problems [13, 24, 28]. Fatigue can be aggravated by physical and mental exertion, and does not decrease after rest [22, 28]. Rather than a single cause, it is likely that multiple biological, psychological and/or social factors may predispose, trigger, and maintain this condition [11]. Identifying potential causal factors is imperative to understanding CFS/ME and to developing more
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