On 2014 Jul 01, Frank Twisk commented:

A definition of recovery in myalgic encephalomyelitis and chronic fatigue syndrome should be based upon objective measures. Qual Life Res. 2014 Jun 17. doi: 10.1007/s11136-014-0737-1. Twisk FNM.

Abstract

Introduction

Adamowicz and colleagues recently proposed to use "a consistent definition of recovery that captures a broad-based return to health with assessments of both fatigue and function as well as the patients' perceptions of his/her recovery status" for patients with chronic fatigue syndrome (CFS).

Methods

A qualitative analysis of case definitions for Myalgic encephalomyelitis (ME) and CFS and methods to assess the symptoms and clinical status of ME and CFS patients objectively.

Results

The criteria of CFS define a heterogeneous disorder. ME, often used interchangeably with CFS, is principally defined by muscle weakness, cognitive impairment etc., but above all post-exertional "malaise": a long-lasting increase in symptoms, e.g. muscle pain and cognitive deficits, after a minor exertion.

The principle symptom of CFS however is "chronic fatigue". Since post-exertional "malaise" is not obligatory for CFS, only part of the CFS patients meet the diagnostic criteria for ME, while not all ME patients qualify as CFS patients.

There are several accepted methods to assess characteristic symptoms and the clinical status of ME and CFS patients using objective measures, e.g. (repeated) cardiopulmonary exercise tests.

Conclusion

To resolve the debate about the clinical status, proposed effectiveness of therapies and recovery in ME and CFS, it is crucial to accurately diagnose patients using well-defined criteria for ME and CFS and an objective assessment of various typical symptoms, since subjective measures such as "fatigue" will perpetuate the debate.

On 2014 Jun 04, Joan Crawford commented:

Part 2

I’d have liked to have seen this article proposing solid, objective measures be used in the future – ones that have face validity with patients and doctors. Moreover, future trials need to decide if the researchers are aiming at recovery from ill health to as near/close to pre-morbid health (as in like how you’d recover from a severe, debilitating infection) or if they are aim at improving functioning/quality of life as a goal in its own right. This difference ideally should be clearly identified.

The omission of the obvious mathematical/statistical flaws with the use of SF36 PF scale was notable. They miss that using 1SD below mean is not an OK thing to do to compare HC and patients. Doing this is a statistical nonsense. The HC data is massively skewed with a ceiling effect. The bulk of HCs score the max score of 100. (Bowling, 1999, Figure 1). The HC data is not normally distributed so using the mean is not terribly helpful here when comparing and setting standards for recovery. I think using the mode (the value that appears most often in the data sets) would be much more meaningful in this context. Similar will be true of the fatigue scores. There was also no mention of the lowering of the SF36 PF scale outcome measure cut off level in the PACE trial (White et al., 2011). There may well be good reasons for doing so but there was no critical review as to whether the reasons given by White et al were good enough to justify the changes between the proposed pre-trial paper measures (White et al 2007) and the final Lancet one. I notice that White was a co-author of the Knoop (2007) paper that selected a SF36 PF score of -1SD as recovered but in a later trial (White et al., 2011) this was reduced to 60 (-2SD). This is quite a leap by the same investigator.

I particularly do not like their pathologisation/speculation of the role of pre-morbid patient functioning. Action prone nonsense. No one can or are they ever likely to prove objectively that pwME/CFS were overactive/overambitious/action prone before getting ill. I hear people mourning the loss of activities, relationships and careers they cherished and enjoyed immensely. So now they cannot even talk about their pre-illness time without feeling psychologised? To not take a more critical view of this is a real let down for me. It’s subjective, speculative nonsense that is unprovable. Patients have a right to get annoyed when they are psychologised in this manner. It should be rather obvious that patients have ex-work colleagues and friends who worked as hard (or harder, longer, faster) than them who are still employed, with families, fit and well with fulfilling lives. PwME/CFS cannot do this because they are sick. Not because they overdid it a bit. That is burnout, not ME/CFS. I thought it remiss to not see the other side regarding how the impact of this speculation might affect patients. Moreover, there are plenty of not especially ambitious, couch potatoes who are ill!

How recovery in ME/CFS is operationalised in the future requires more critical thought and this must take into account how the patients define recovery and must be able to be demonstrated objectively (Haywood et al., 2011).

References:

Anthony, W. A. (1993) Recovery from mental illness: the guiding vision of the mental health service system in the 1990s. Psychosocial Rehabilitation Journal, 16, 11-23.

Bowling A., Bond, M., Jenkinson, C., & Lamping, D.L. (1999). Short Form 36 (SF-36) Health Survey questionnaire: which normative data should be used? Comparisons between the norms provided by the Omnibus Survey in Britain, the Health Survey for England and the Oxford Healthy Life Survey. Journal of Public Health Medicine. 21(3):255-70.

Haywood, K.L., Staniszewska, S., & Chapman, S. (2011). Quality and acceptability of patient-reported outcome measures used in chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME): A systematic review. Quality of Life Research, In press.

Knoop, H., Bleijenberg, G., Gielissen, M. F. M., van der Meer, J.W. M., & White, P. D. (2007). Is a full recovery possible after cognitive behavioral therapy for chronic fatigue syndrome? Psychotherapy and Psychosomatics, 76, 171–176.

Lester, H., & Gask, L. (2006). Delivering medical care for patients with serious mental illness or promoting a collaborative model of recovery. British Journal of Psychiatry, 188, 401–402.

White, P.D., Sharpe, M.C., Chalder, T., DeCesare, J.C., Walwyn, R; on behalf of the PACE trial group. (2007). Protocol for the PACE trial: a randomised controlled trial of adaptive pacing, cognitive behaviour therapy, and graded exercise, as supplements to standardised specialist medical care versus standardised specialist medical care alone for patients with the chronic fatigue syndrome/myalgic encephalomyelitis or encephalopathy. BioMed Cent Neurology, 7:6.

White, P.D., Goldsmith, K.A., Johnson, A.L., Potts, L., Walwyn, R., DeCesare, J.C., et al. (2011). Comparison of adaptive pacing therapy, cognitive behaviour therapy, graded exercise therapy, and specialist medical care for chronic fatigue syndrome (PACE): A randomised trial. Lancet, 5, 377(9768), 823-836.

On 2014 Jun 04, Joan Crawford commented:

Part 1

This paper has a great description of the differences between ‘recovery’ versus ‘adaptation/feeling better’. As a patient I describe this as the difference between ‘feeling better’ and ‘being better’ (i.e. recovered to point comparable with pre-existing health given age increase with ability to return to work/social life without the presence of limiting symptoms). These things appear to get muddled in many clinical trials. I suspect anyone feeling poorly will feel a bit or a lot better after a psychological intervention with a concerned, empathic individual, however, whether this has any impact on their underlying condition or activity levels needs to be shown by objective measures.

It was a disappointment that in this paper there was no push for more objective measures of patient functioning to be used in future research work. For example, pedometers, actigraphy, neuropsychological tests, 2 day exercise testing (if well enough at baseline), using simple dynamometers taking readings over 2 days, return to work/school (or ability to do if the person wished), move from incapacity/sickness welfare payments to job seeking benefits and so on. I’m perplexed when researchers claim that patients are recovered if they continue to receive incapacity/ill health payments. Perhaps in this condition it might be worth measuring whether saliva cortisol levels and NK cell functioning normalise. At a push even the simple 6 minute walking test could be helpful. Objective measures need not be expensive. Simple, reliable and cheap equipment such as pedometers are available for around $30 and can show really well if a patient who is doing better over time. This could be used quite simply to get around the issue of is the patient feeling better because they are actually doing less that is discussed in the review. It’s a shame that straightforward solutions like this were not suggested.

Within the paper the authors refer to Lester & Gask (2006), which includes a popular definition of recovery from within mental health context by William Anthony as: “‘a way of living a satisfying, hopeful and contributing life even with the limitations caused by illness. Recovery involves the development of a new meaning and purpose in one’s life as one grows beyond the catastrophic effects of mental illness’ (Antony, 1993: p. 21). I had not seen this definition before for recovery so it was educating to be made aware that this was a widely used concept. To me that defines adaptation, not recovery. I would not be happy with researchers who decided that that was a good place to start defining recovery from ME/CFS. I’d want the goal to be at a minimum to not feel ill or sick or debilitated and able to be free of disease symptoms and normal functioning for the patient for their age.

References and Part 2 above

On 2014 Jun 04, Joan Crawford commented:

Part 1

This paper has a great description of the differences between ‘recovery’ versus ‘adaptation/feeling better’. As a patient I describe this as the difference between ‘feeling better’ and ‘being better’ (i.e. recovered to point comparable with pre-existing health given age increase with ability to return to work/social life without the presence of limiting symptoms). These things appear to get muddled in many clinical trials. I suspect anyone feeling poorly will feel a bit or a lot better after a psychological intervention with a concerned, empathic individual, however, whether this has any impact on their underlying condition or activity levels needs to be shown by objective measures.

It was a disappointment that in this paper there was no push for more objective measures of patient functioning to be used in future research work. For example, pedometers, actigraphy, neuropsychological tests, 2 day exercise testing (if well enough at baseline), using simple dynamometers taking readings over 2 days, return to work/school (or ability to do if the person wished), move from incapacity/sickness welfare payments to job seeking benefits and so on. I’m perplexed when researchers claim that patients are recovered if they continue to receive incapacity/ill health payments. Perhaps in this condition it might be worth measuring whether saliva cortisol levels and NK cell functioning normalise. At a push even the simple 6 minute walking test could be helpful. Objective measures need not be expensive. Simple, reliable and cheap equipment such as pedometers are available for around $30 and can show really well if a patient who is doing better over time. This could be used quite simply to get around the issue of is the patient feeling better because they are actually doing less that is discussed in the review. It’s a shame that straightforward solutions like this were not suggested.

Within the paper the authors refer to Lester & Gask (2006), which includes a popular definition of recovery from within mental health context by William Anthony as: “‘a way of living a satisfying, hopeful and contributing life even with the limitations caused by illness. Recovery involves the development of a new meaning and purpose in one’s life as one grows beyond the catastrophic effects of mental illness’ (Antony, 1993: p. 21). I had not seen this definition before for recovery so it was educating to be made aware that this was a widely used concept. To me that defines adaptation, not recovery. I would not be happy with researchers who decided that that was a good place to start defining recovery from ME/CFS. I’d want the goal to be at a minimum to not feel ill or sick or debilitated and able to be free of disease symptoms and normal functioning for the patient for their age.

References and Part 2 above

On 2014 Jun 04, Joan Crawford commented:

Part 2

I’d have liked to have seen this article proposing solid, objective measures be used in the future – ones that have face validity with patients and doctors. Moreover, future trials need to decide if the researchers are aiming at recovery from ill health to as near/close to pre-morbid health (as in like how you’d recover from a severe, debilitating infection) or if they are aim at improving functioning/quality of life as a goal in its own right. This difference ideally should be clearly identified.

The omission of the obvious mathematical/statistical flaws with the use of SF36 PF scale was notable. They miss that using 1SD below mean is not an OK thing to do to compare HC and patients. Doing this is a statistical nonsense. The HC data is massively skewed with a ceiling effect. The bulk of HCs score the max score of 100. (Bowling, 1999, Figure 1). The HC data is not normally distributed so using the mean is not terribly helpful here when comparing and setting standards for recovery. I think using the mode (the value that appears most often in the data sets) would be much more meaningful in this context. Similar will be true of the fatigue scores. There was also no mention of the lowering of the SF36 PF scale outcome measure cut off level in the PACE trial (White et al., 2011). There may well be good reasons for doing so but there was no critical review as to whether the reasons given by White et al were good enough to justify the changes between the proposed pre-trial paper measures (White et al 2007) and the final Lancet one. I notice that White was a co-author of the Knoop (2007) paper that selected a SF36 PF score of -1SD as recovered but in a later trial (White et al., 2011) this was reduced to 60 (-2SD). This is quite a leap by the same investigator.

I particularly do not like their pathologisation/speculation of the role of pre-morbid patient functioning. Action prone nonsense. No one can or are they ever likely to prove objectively that pwME/CFS were overactive/overambitious/action prone before getting ill. I hear people mourning the loss of activities, relationships and careers they cherished and enjoyed immensely. So now they cannot even talk about their pre-illness time without feeling psychologised? To not take a more critical view of this is a real let down for me. It’s subjective, speculative nonsense that is unprovable. Patients have a right to get annoyed when they are psychologised in this manner. It should be rather obvious that patients have ex-work colleagues and friends who worked as hard (or harder, longer, faster) than them who are still employed, with families, fit and well with fulfilling lives. PwME/CFS cannot do this because they are sick. Not because they overdid it a bit. That is burnout, not ME/CFS. I thought it remiss to not see the other side regarding how the impact of this speculation might affect patients. Moreover, there are plenty of not especially ambitious, couch potatoes who are ill!

How recovery in ME/CFS is operationalised in the future requires more critical thought and this must take into account how the patients define recovery and must be able to be demonstrated objectively (Haywood et al., 2011).

References:

Anthony, W. A. (1993) Recovery from mental illness: the guiding vision of the mental health service system in the 1990s. Psychosocial Rehabilitation Journal, 16, 11-23.

Bowling A., Bond, M., Jenkinson, C., & Lamping, D.L. (1999). Short Form 36 (SF-36) Health Survey questionnaire: which normative data should be used? Comparisons between the norms provided by the Omnibus Survey in Britain, the Health Survey for England and the Oxford Healthy Life Survey. Journal of Public Health Medicine. 21(3):255-70.

Haywood, K.L., Staniszewska, S., & Chapman, S. (2011). Quality and acceptability of patient-reported outcome measures used in chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME): A systematic review. Quality of Life Research, In press.

Knoop, H., Bleijenberg, G., Gielissen, M. F. M., van der Meer, J.W. M., & White, P. D. (2007). Is a full recovery possible after cognitive behavioral therapy for chronic fatigue syndrome? Psychotherapy and Psychosomatics, 76, 171–176.

Lester, H., & Gask, L. (2006). Delivering medical care for patients with serious mental illness or promoting a collaborative model of recovery. British Journal of Psychiatry, 188, 401–402.

White, P.D., Sharpe, M.C., Chalder, T., DeCesare, J.C., Walwyn, R; on behalf of the PACE trial group. (2007). Protocol for the PACE trial: a randomised controlled trial of adaptive pacing, cognitive behaviour therapy, and graded exercise, as supplements to standardised specialist medical care versus standardised specialist medical care alone for patients with the chronic fatigue syndrome/myalgic encephalomyelitis or encephalopathy. BioMed Cent Neurology, 7:6.

White, P.D., Goldsmith, K.A., Johnson, A.L., Potts, L., Walwyn, R., DeCesare, J.C., et al. (2011). Comparison of adaptive pacing therapy, cognitive behaviour therapy, graded exercise therapy, and specialist medical care for chronic fatigue syndrome (PACE): A randomised trial. Lancet, 5, 377(9768), 823-836.

On 2014 Jul 01, Frank Twisk commented:

A definition of recovery in myalgic encephalomyelitis and chronic fatigue syndrome should be based upon objective measures. Qual Life Res. 2014 Jun 17. doi: 10.1007/s11136-014-0737-1. Twisk FNM.

Abstract

Introduction

Adamowicz and colleagues recently proposed to use "a consistent definition of recovery that captures a broad-based return to health with assessments of both fatigue and function as well as the patients' perceptions of his/her recovery status" for patients with chronic fatigue syndrome (CFS).

Methods

A qualitative analysis of case definitions for Myalgic encephalomyelitis (ME) and CFS and methods to assess the symptoms and clinical status of ME and CFS patients objectively.

Results

The criteria of CFS define a heterogeneous disorder. ME, often used interchangeably with CFS, is principally defined by muscle weakness, cognitive impairment etc., but above all post-exertional "malaise": a long-lasting increase in symptoms, e.g. muscle pain and cognitive deficits, after a minor exertion.

The principle symptom of CFS however is "chronic fatigue". Since post-exertional "malaise" is not obligatory for CFS, only part of the CFS patients meet the diagnostic criteria for ME, while not all ME patients qualify as CFS patients.

There are several accepted methods to assess characteristic symptoms and the clinical status of ME and CFS patients using objective measures, e.g. (repeated) cardiopulmonary exercise tests.

Conclusion

To resolve the debate about the clinical status, proposed effectiveness of therapies and recovery in ME and CFS, it is crucial to accurately diagnose patients using well-defined criteria for ME and CFS and an objective assessment of various typical symptoms, since subjective measures such as "fatigue" will perpetuate the debate.