Understanding the ME/CFS research: Which CFS?
Jason LA, Najar N, Porter N, Reh C. "Evaluating the Centers for Disease Control's Empirical Chronic Fatigue Syndrome Case Definition". J Disability Policy Studies. September 2009 vol. 20 no. 2 93-100.
The authors research group proposes that selection criteria for CFS cases have broadened and provides data indicating 38% of those with a Major Depressive Disorder were misclassified as having CFS under the new CDC empirical case definition.
Merz S. [Chronic fatigue syndrome. More and more differential diagnoses suggest a new view of this syndrome]. Lakartidningen. 2002 Aug 22;99(34):3282-7. PMID: 12362846
Several CFS definitions have been developed over the years, and it is common for investigators to erroneously compare studies based on different definitions, which nevertheless all use the term CFS. Much of our "understanding" of CFS does not apply to the small group of patients who fulfill the current (1994) CDC definition.
Even the more specific of the commonly used definitions, Fukuda (1994 CDC), does not make the hallmarks of ME, such as post-exertional malaise, required as diagnostic/selection criteria, so one cannot tell how many study participants have ME (myalgic encephalomyelitis, G93.3, the infectious neuro-immune disease the first CFS definition tried to describe). Other conditions besides MDD which can easily be included in a 'CFS' group include missed cases of lupus, MS, simple vitamin D deficiency, rare diseases, and so forth.
Jason et al. Politics, Science, and the Emergence of a New Disease: The Case of CFS Am Psychol. 1997 Sep;52(9):973-83. PMID: 9301342
Is CFS causally related to deconditioning, and does exercise improve ME/CFS? Does CBT?
Is there any evidence to support the use of pacing?
Carruthers et al. "Myalgic Encephalomyelitis/Chronic Fatigue Syndrome: Clinical Working Case Definition, Diagnostic and Treatment Protocols" J CFS, Vol. 11(1) 2003, pp. 7-115 (pp 46-49).
http://cfids-cab.org/MESA/ccpc-1.html
Explains that most CBT/GET (cognitive behavioural therapy/graded exercise therapy) studies are done on fatigued patients using the name but not any specific criteria for CFS. Refusal and drop-out rates may be very high. Assessment is chiefly subjective. When used respectfully, cognitive therapy may be useful as an adjunct therapy. Some CF patients may see modest improvement from increased activity; but ME/CFS patients report adverse effects, consistent with the definitions and clinical and research findings.
Reasons for drop-outs are not typically assessed or recorded in studies on GET and CBT (which is a departure from normal research standards). However, drop-out rates are generally considered a proxy indicator of harm or other difficulty or dissatisfaction for the patients, and patient surveys reveal a high rate of patients advising that GET made them worse. In fact, GET is consistently rated the worst of all treatments tried. Yet GET remains a top recommended treatment.
Kindlon, T. Reporting of Harms Associated with Graded Exercise Therapy and Cognitive Behavioural Therapy in Myalgic Encephalomyelitis/Chronic Fatigue Syndrome. Bulletin of the IACFS/ME, Fall 2011;19(2):59-111.
www.iacfsme.org/LinkClick.aspx?fileticket=Rd2tIJ0oHqk=&tabid=501
51% of survey respondents (range 28-82%, n=4338, 8 surveys) reported that GET worsened their health while 20% of respondents (range 7-38%, n=1808, 5 surveys) reported similar results for CBT.
Issues involving the heterogeneity of subjects and interventions, tracking of adverse events, trial participants compliance to therapies, and measurement of harms using patient oriented and objective outcome measures are discussed.
The recently published PACE (Pacing, graded activity, and cognitive behavior therapy: A randomized evaluation) trial, which explicitly aimed to assess safety, as well as effectiveness, is also analyzed in detail.
Price JR, Mitchell E, Tidy E, Hunot V. Cognitive behaviour therapy for chronic fatigue syndrome in adults. Cochrane Database Syst Rev. 2008 Jul 16;(3):CD001027. Review. PMID: 18646067
Claims effectiveness based on symptoms of fatigue, a purely subjective measure, and states, The evidence base at follow-up is limited to a small group of studies with inconsistent findings. There is a lack of evidence on the comparative effectiveness of CBT alone or in combination with other treatments, and further studies are required to inform the development of effective treatment programmes for people with CFS.
Nez M, Fernndez-Sol J, Nuez E, Fernndez-Huerta JM, Gods-Sieso T, Gomez-Gil E. "Health-related quality of life in patients with chronic fatigue syndrome: group cognitive behavioural therapy and graded exercise versus usual treatment. A randomised controlled trial with 1 year of follow-up." Clin Rheumatol. Epub 2011 Jan 15. PMID: 21234629
At 12 months, the intervention did not improve HRQL scores, with worse SF-36 physical function and bodily pain scores in the intervention group. Multidisciplinary treatment was not superior to usual treatment at 12 months in terms of HRQL. Although some studies cautiously conclude that exercise therapy is a promising treatment for CFS [14], the results of our study tend to support the somewhat controversial findings of Twisk and Maes [43] that the combination of CBT and GET is ineffective and not evidence-based and may in fact be harmful in some patients, a view supported by various surveys carried out by patient advocate groups [41].
Nijs J, Zwinnen K, Meeusen R, de Geus B, De Meirleir K. "Comparison of two exercise testing protocols in patients with chronic fatigue syndrome." J Rehabil Res Dev. 2007;44(4):553-9. PMID: 18247252
Symptoms are typically worsened after modest amounts of exercise [2], after increased daily physical activity [3], and after a maximal exercise stress test [4-5]. A delayed recovery from exercise typically occurs in patients with CFS [6].... [further discussion of findings and problems of exercise in ME/CFS and of CFS assessment]
Clapp LL, Richardson MT, Smith JF, Wang M, Clapp AJ, Pieroni RE. "Acute effects of thirty minutes of light-intensity, intermittent exercise on patients with chronic fatigue syndrome." Phys Ther. 1999;79(8):749-56 PMID: 10440661
Although clinical findings and self-reported symptoms vary among patients with CFS, a worsening of symptoms, especially of fatigue, after previously well-tolerated levels of exercise continues to be the hallmark of this incapacitating disorder.[25]... Patients with CFS typically feel much worse after modest amounts of physical exercise. Some investigators have suggested the onset of severe symptoms to be 6 to 48 hours following exercise[25] and to last from 2 days to 2 weeks.[2,26]... some individuals with CFS may be able to use low-level, intermittent exercise [3 min sessions] without exacerbating their [condition].
Nijs J, Almond F, De Becker P, Truijen S, Paul L. "Can exercise limits prevent post-exertional malaise in chronic fatigue syndrome? An uncontrolled clinical trial." Clin Rehabil. 2008 May;22(5):426-35. PMID: 18441039
It was shown that the use of exercise limits (limiting both the intensity and duration of exercise) prevents important health status changes following a walking exercise in people with chronic fatigue syndrome, but was unable to prevent short-term symptom increases.
Note that both of the above are time-limited trials and do not measure the effect of an ongoing program over time. Nor do they asses how adding an exercise program might affect ability to carry out important ADLs such as meal preparation. And again, this would be mild and moderate patients, not severe ones.
Black CD, McCully KK. Time course of exercise induced alterations in daily activity in chronic fatigue syndrome. Dyn Med. 2005 Oct 28;4:10. PMID: 16255779
Over the first 4-10 days of walking the subjects with CFS were able to reach the prescribed activity goals each day. After this time, walking and total activity counts decreased. Sedentary controls subjects were able to maintain their daily walking and total activity goals throughout the 4 weeks. Unlike our previous interpretation of the data, we feel this new analysis suggests that CFS patients may develop exercise intolerance as demonstrated by reduced total activity after 4-10 days. The inability to sustain target activity levels, associated with pronounced worsening of symptomology, suggests the subjects with CFS had reached their activity limit.
Bazelmans E, Bleijenberg G, Van Der Meer JW, Folgering H. "Is physical deconditioning a perpetuating factor in chronic fatigue syndrome? A controlled study on maximal exercise performance and relations with fatigue, impairment and physical activity." Psychol Med. 2001 Jan;31(1):107-14. PMID: 11200949
There were no statistically significant differences in physical fitness between CFS patients and their controls. Nine CFS patients had a better fitness than their control.... Physical deconditioning does not seem a perpetuating factor in CFS.
VanNess JM, Stevens SR, Bateman L, Stiles TL, Snell CR. "Postexertional malaise in women with chronic fatigue syndrome." J Womens Health (Larchmt). 2010 Feb;19(2):239-44. PMID: 20095909
The results of this study suggest that PEM is both a real and an incapacitating condition for women with CFS and that their responses to exercise are distinctively different from those of sedentary controls.
Sorensen, et al. "Complement activation in a model of chronic fatigue syndrome." J Allergy and Clin Immunol. 2003 Aug;112(2):397-403. PMID: 12897748
Activity produces immune response, which is associated with symptom flare (PEM).
De Becker P, Roeykens J, Reynders M, McGregor N, De Meirleir K. "Exercise capacity in chronic fatigue syndrome." Arch Intern Med. 2000 Nov 27;160(21):3270-7.
When compared with healthy sedentary women, female patients with CFS show a significantly decreased exercise capacity. This could affect their physical abilities to a moderate or severe extent. Reaching the age-predicted target heart rate seemed to be a limiting factor of the patients with CFS in achieving maximal effort, which could be due to autonomic disturbances.
VanNess MJ, Snell CF, Stevens SR. "Diminished Cardiopulmonary Capacity During Post-Exertional Malaise." J CFS Vol 14(2) 2007. haworthpress.com doi:10.1300/J092v14n02_07
Reduced functional capacity and post-exertional malaise following physical activity are hallmark symptoms of Chronic Fatigue Syndrome (CFS). That these symptoms are often delayed may explain the equivocal results from clinical cardiopulmonary exercise testing with CFS patients. ... For test 2 the CFS patients achieved significantly lower values for both VO2 peak and AT (anaerobic threshold). ... A second test may be necessary to document the atypical recovery response and protracted malaise unique to CFS.
Nijs J, van Eupen I, Vandecauter J, et al. "Can pacing self-management alter physical behavior and symptom severity in chronic fatigue syndrome? A case series." J Rehabil Res Dev. 2009;46(7):985-96. PMID: 20104421
When comparing pre- versus post-treatment data, we found that the patients' ability to perform daily activities and the severity of their symptom complexes were improved (p = 0.043). Concentration difficulties, mood swings, muscle weakness, and intolerance to bright light improved as well.... We found that 3 weeks of pacing self-management was accompanied by a modest improvement in symptom severity and daily functioning.
Lehman AM, Lehman DR, Hemphill KJ, Mandel DR, Cooper LM. "Illness experience, depression, and anxiety in chronic fatigue syndrome." J Psychosom Res. 2002 Jun;52(6):461-5. PMID: 12069870
Those who believed that limiting their physical exertion was the path to recovery (55%) had lower depression and anxiety scores (P's<.01) than their counterparts.
Jason L, Benton M, Torres-Harding S, Muldowney K. "The impact of energy modulation on physical functioning and fatigue severity among patients with ME/CFS." Patient Educ Couns. 2009 Nov;77(2):237-41. PMID: 19356884
Those who were able to stay within their energy envelope had significant improvements in physical functioning and fatigue severity. Health care professionals that treat patients with ME/CFS might incorporate strategies that help patients self-monitor and self-regulate energy expenditures.
So what is the patient experience like?
Asbring P, Nrvnen AL. Ideal versus reality: physicians perspectives on patients with chronic fatigue syndrome (CFS) and fibromyalgia. Soc Sci Med. 2003 Aug;57(4):711-20. PMID: 12821018
Encountering patients with chronic fatigue syndrome (CFS) or fibromyalgia can cause dilemmas for physicians due to the uncertainty inherent in these illnesses. [as per the PC narrative] ... Different strategies are developed to handle the encounters with these patients. The results also illuminate the physician's interpretations of patients in moralising terms. Conditions given the status of illness were regarded, for example, as less serious by the physicians than those with disease status. Scepticism was expressed regarding especially CFS, but also fibromyalgia. Moreover, it is shown how the patients are characterised by the physicians as ambitious, active, illness focused, demanding and medicalising. The patient groups in question do not always gain full access to the sick-role, in part as a consequence of the conditions not being defined as diseases.
[despite abundant evidence of biomedical pathology, which unfortunately is not provided to doctors and nurses through usual channels]
Gilje AM, Sderlund A, Malterud K. "Obstructions for quality care experienced by patients with chronic fatigue syndrome (CFS)--a case study." Patient Educ Couns. 2008 Oct;73(1):36-41. Epub 2008 May 16. PMID: 18486415
RESULTS: CFS patients said that lack of acknowledgement could be even worse than the symptoms. They wanted their doctors to ask questions, listen to them and take them seriously, instead of behaving degrading. Many participants felt that the doctors psychologized too much, or trivialized the symptoms. Participants described how doctors' lack of knowledge about the condition would lead to long-term uncertainty or maltreatment. Even with doctors who were supportive, it would usually take months and sometimes years until a medical conclusion would be reached, or other disorders were ruled out. Increased physical activity had been recommend, but most of the informants experienced that this made them worse.
CONCLUSION: Current medical scepticism and ignorance regarding CFS shapes the context of medical care and the illness experiences of CFS patients, who may feel they neither get a proper assessment nor management.
PRACTICE IMPLICATIONS: CFS patients' reports about patronizing attitudes and ignorance among doctors call for development of evidence based strategies and empowerment of patients, acknowledging the patients' understanding of symptoms and the complex nature of the disease.
Drachler Mde L, et al. "The expressed needs of people with chronic fatigue syndrome/myalgic encephalomyelitis: a systematic review." BMC Public Health. 2009 Dec 11;9:458. Review. PMID: 20003363
The review has shown that the lack of recognition of needs and poor support from the health and social systems further compromise socioeconomic status, activities of daily living, social participation and personal development, thereby exacerbating the impact of the illness in their lives.
Larun L, Malterud K. Identity and coping experiences in Chronic Fatigue Syndrome: a synthesis of qualitative studies. Patient Educ Couns. 2007 Dec;69(1-3):20-8. Epub 2007 Aug 14. PMID: 17698311
RESULTS: Twenty qualitative studies on CFS experiences were identified. Symptom experiences and the responses from significant others could jeopardise the patients' senses of identity. They felt severely ill, yet blamed and dismissed. Patients' beliefs and causal attributions oppose the doctors
CONCLUSION: The identities of CFS patients are challenged when the legitimacy of their illness is questioned. This significant burden adds to a loss of previously established identity and makes the patient more vulnerable than just suffering from the symptoms. CFS patients work hard to cope with their condition by knowing more, keeping a distance to protect themselves and learning more about their limits.
PRACTICE IMPLICATIONS: Doctors can support patients' coping by supporting the strong sides of the patients instead of casting doubt upon them.
and nurses can also support the strong sides of the patients
