Illness beliefs and treatment outcome in chronic fatigue syndrome, 1998, Deale, Chalder and Wessely

[Hutan]

Abstract
Longitudinal studies have shown that physical illness attributions are associated with poor prognosis in chronic fatigue syndrome (CFS). Speculation exists over whether such attributions influence treatment outcome.

This study reports the effect of illness beliefs on outcome in a randomized controlled trial of cognitive-behavior therapy versus relaxation. Causal attributions and beliefs about exercise, activity, and rest were recorded before and after treatment in 60 CFS patients recruited to the trial.

Physical illness attributions were widespread, did not change with treatment, and were not associated with poor outcome in either the cognitive-behavior therapy group or the control group.

Beliefs about avoidance of exercise and activity changed in the cognitive behavior therapy group, but not in the control group. This change was associated with improved outcome.

These findings suggest that physical illness attributions are less important in determining outcome (at least in treatment studies) than has been previously thought. In this study, good outcome is associated with change in avoidance behavior, and related beliefs, rather than causal attributions.

https://www.sciencedirect.com/science/article/abs/pii/S002239999800021X
paywall
Edited to add: https://www.simonwessely.com/Downloads/Publications/CFS/88.pdf

 

[Utsikt]

Beliefs about avoidance of exercise and activity changed in the cognitive behavior therapy group, but not in the control group. This change was associated with improved outcome.

Do we know what ‘outcome’ means here? What did they measure?

 

[alktipping]

Do we know what ‘outcome’ means here? What did they measure?

They measured their own hubris and liked what they saw .

 

[rvallee]

I literally have no beliefs. I am a universal agnostic. I don't even understand the concept of believing in something, it strikes me as quite silly (although I do understand and respect that for a lot of people this is definitely important) because how the hell would I just know the answer to complex questions? Makes zero sense to me. I know I'm weird about that.

So this makes those long-standing claims so damn weird to me. I think I said it before, but I literally rate a complete zero on the usual checklist of stereotypes and psychosocial factors. Not a single one of them. Which doesn't even seem possible simply through chance, considering how most of them are completely generic, and the main factor (let's be honest here) literally has a 51% chance.

I'm not a woman. Was not middle aged when I got ill. I did not have a severe acute illness before. Never had trauma, I couldn't even name a single 'traumatic' event in my life. Had and still have a great family. I did well at school. Comfortable middle class, enough to feel its freedom, but not too much that it screwed me up. And I definitely do not have beliefs, about anything, and I am probably one of the least biased people in the world. I don't even understand the idea of thinking I know the answer to things I don't understand, or about wanting specific answers to be correct. I don't want to intuitively guess stuff, I want to know stuff. How it really works. For real.

But on the belief scale, I am definitely in the bottom 0.01%. I simply don't have any. It makes all those claims about my life experience sound completely clownish, especially considering how both (and oddly) specific and generic they are.

 

[Utsikt]

@Hutan found a link to the full version on Wessely’s webpage:
https://www.simonwessely.com/Downloads/Publications/CFS/88.pdf

 

[Utsikt]

This is the paper on the trial:
https://psychiatryonline.org/doi/epdf/10.1176/ajp.154.3.408

 

[Yann04]

I literally have no beliefs. I am a universal agnostic

Same. My entire outlook on the world before getting ME was a sort of external world sceptic, moral non-realist, post-modernist and post-structuralist outlook.
Basically a lot of buzzwords for saying everything is relative and we can’t even really know if there is anything real or such thing as objective truth.

In a philosophical sense, my only belief is the we cannot know if literally anything is true because every information we have is shaped by our percetions which are in turn shaped by cognitive processes (whatever that means and that’s really upto debate).

 

[Utsikt]

This is the paper on the trial:
https://psychiatryonline.org/doi/epdf/10.1176/ajp.154.3.408

The purpose of this study was to test whether cogni- tive behavior therapy (comprising graded activity and cognitive restructuring) was significantly superior to re- laxation, selected to control for nonspecific treatment factors, including support, therapist time and attention, expectations, and homework practice

Diagnosis of chronic fatigue syndrome was made according to U.K. (15) and U.S. (16) case definitions

15 is Oxford. 16 is this: https://pubmed.ncbi.nlm.nih.gov/1322076/

 

[Utsikt]

This is the paper on the trial:
https://psychiatryonline.org/doi/epdf/10.1176/ajp.154.3.408

All measures other than the assessor ratings were self-rated.

This means that we have the issue of an unblinded trial with only subjective outcomes. This makes the results notoriously unreliable.

Outcome assessment depended largely on self-rated outcome measures. However, no objective measures ex- ist for subjectively experienced fatigue, disability, and mood disturbance, which are the areas of interest in chronic fatigue syndrome. It is acknowledged that in- vestigators rely on patient self-report instruments (16); we therefore used recommended, reproducible meas- ures that are sensitive to change in chronic fatigue syn- drome (11, 16, 29).

There is no reason to not using objective measures like walking distance, employment, etc.

 

[Utsikt]

 

[Trish]

If I read this correctly, it seems that they found that what one believes about the cause of illness has no effect on the subjective outcomes of a CBT versus relaxation trial.
And they found an association between some aspects of exercise avoidance beliefs and subjective outcomes. They admit this is only an association, and cannot say anything about causation.

 

[Hutan]

This means that we have the issue of an unblinded trial with only subjective outcomes. This makes the results notoriously unreliable.

The trial is even worse than that.

The exclusion criteria were somatization disorder, severe depression (DSM-III-R melancholic subtype), ongoing physical in-
vestigations, concurrent new treatment, and inability to attend all treatment sessions.

An enormous amount of work was required from those in the CBT group.

There were 13 sessions (CBT vs relaxation). There was homework in daily diaries.
For CBT (which included GET):

Presenting problems were assessed, and patients kept diaries recording hourly details of activity, rest, and fatigue.

Rather than being symptom dependent, activity and rest were divided into small, man-ageable portions spread across the day (for example, three 5-minute walks daily rather than a 45-minute walk once a week). Patients were encouraged to persevere with their targets and not to reduce them on a bad day or exceed them on a good day.

Once a structured schedule was established, activity was gradually increased and rest was reduced, step by step as tolerance developed. Therapist and patient agreed on specific daily targets covering a range of activities (such as walking, reading, visiting friends, or gardening). A sleep routine was established—for example, stopping daytime sleep, rising at a specific time each morning, reducing time in bed, and using stimulus control techniques for insomnia (20).

Cognitive strategies were introduced at session 8 (while the graded activity program continued). Patients recorded any unhelpful or dis-tressing thoughts and, in discussion and as homework, practiced generating alternatives (21). The unhelpful or distressing thoughts
included fears about symptoms and treatment, perfectionism, self-criticism, guilt, and performance expectations.

In the final sessions, strategies for dealing with setbacks were re-hearsed and patients drew up “action plans” to guide them through the coming months. The importance of maintaining the principles of therapy after discharge was reinforced.

So, there was teaching on how to deal with distressing thoughts that would have impacted on how participants reported on their health in the followup period.

 

[Utsikt]

More from the trial:

All patients were seen individually, at weekly or fortnightly inter- vals. Mean therapist time per patient was 15 hours. Information leaf- lets supplemented each phase of treatment. Each session began with a homework review and ended with agreement on homework tasks, which were recorded in daily diaries. The therapist followed detailed session-by-session treatment manuals devised for both cognitive be- havior therapy and relaxation. The research team met fortnightly to review cases and ensure protocol adherence.

There were no attempts at blinding within the research team, even though only one of them acted as the therapist.

Cognitive behavior therapy.

This treatment was collaborative, educative, and negotiated and had a behavioral emphasis. The aim was to show patients that activity could be increased steadily and safely without exacerbating symptoms. Sessions 1 to 3 involved en- gaging the patients in therapy and offering a detailed treatment ra- tionale. Presenting problems were assessed, and patients kept diaries recording hourly details of activity, rest, and fatigue.

The bolded part shows how the intervention might cause the participant to report that they can do more, regardless of any actual changes - because they have been told that that’s how things work and that that’s what they should expect to experience.

Cognitive strategies were introduced at session 8 (while the graded activity program continued). Patients recorded any unhelpful or dis- tressing thoughts and, in discussion and as homework, practiced generating alternatives (21). The unhelpful or distressing thoughts included fears about symptoms and treatment, perfectionism, self- criticism, guilt, and performance expectations.

Any doubts about the effectiveness of the treatment was labeled as unhelpful, and actively discouraged as a part of the treatment.

In the final sessions, strategies for dealing with setbacks were re-
hearsed and patients drew up “action plans” to guide them through
the coming months. The importance of maintaining the principles of
therapy after discharge was reinforced

Patients made action plans for how they would continue to convince themselves that they could do more after the treatment.

The control group:

Relaxation.

The same session structure was followed in the relaxation group. The first three sessions involved engagement, rationale
giving, information gathering, and diary keeping (recording daily events, feelings, fatigue, and muscle tension). No advice about scheduling activity, reducing rest, or altering sleep patterns was given. The
relaxation techniques were adapted from applied relaxation training (22). Progressive muscle relaxation, visualization, and rapid relaxation skills were taught during the 10 treatment sessions and were practiced twice daily as homework.

The control group was seemingly not told how to think or behave.

All in all, the contents of the intervention make the participant’s answers even more unreliable than what they already were as subjective reports.

In short, it is fatally flawed.

 

[Hutan]

The CBT group was younger (mean age 31), relaxation group (38).
Illness duration was different (CBT 3.4 years....relaxation 4.6 years)

More of the CBT group would have been in the onset range where natural recovery is common.

Patient attribution of symptoms to physical illness was very different (CBT 57%.... relaxation 73%)

I wonder about the exclusion - people with an inability to attend all of the sessions were excluded. That is an ambiguous exclusion, allowing the researchers potentially to exclude people who might have started and decided the intervention was not for them, and not mention them as drop outs. 8 people of the 147 who were screened were diagnosed with somatisation disorder and excluded.

12 were unable to attend sessions regu-larly (seven lived too far away or had work commitments, and five were bed bound or dependent on wheel-chairs). Of the 67 patients eligible for trial entry, seven(10%) refused; two gave no reason for refusing, threedid not wish to be randomized, and two did not wish to have cognitive behavior therapy

The CBT intervention was onerous, with non-symptom contingent GET. The people who got through to the end of the CBT intervention would have been a very select group. I suspect they would have included people who were probably already somewhat recovering at trial start, and wondering if they should be doing more activity.

Table 2 lists the completers as CBT 27/30 and relaxation 26/30.

 

[Hutan]

I think the primary outcome was what we would call the SF-36 Physical Functioning scale.

Physical functioning scale of Medical Outcomes Study Short-Form General Health Survey

CBT: Pre-treatment 25.5; Post-treatment 56.2; 6 months 71.6
Controls: Pre-treatment 27.8; post-treatment 34.6; 6 months 38.4

We've talked before about the problems with SF-36. For many of us, it's hard to know how to answer a question about how easy it is to climb a flight of stairs. It would be very easy to change someone's frame of reference when it comes to answering those questions.

The criterion for improvement was an increase of 50 or more or an end score of 83 or more (which represents the ability to carry out moderate activities, such as lifting a table, carrying pur-chases, or bowling, without limitations).

I have to say, on the face of it, with the crucial exception of the subjective outcomes for an unblinded intervention that trains people to report better outcomes, the study is quite well done. And, although people in the CBT group weren't cured, there were significant reported improvements at 6 months.

At 6-month follow-up, five relaxation patients (but no cognitive behavior therapy patients) rated themselves as worse; none attributed this to treatment.

It is difficult to reconcile the results of this trial with what we know. It is surprising that the improvements continued for so long.

More of the patients receiving cognitive behavior therapy rated themselves as satisfied with their level of improvement, but almost all patients rated the treatments as useful. The assessor ratings of improvement in disability and fatigue were consistent withthe self-rated improvement.

 

[Hutan]

There were no significant differences between the im-proved and unimproved patients on any pretreatment characteristic, including psychiatric disorder and illness attributions. Poor outcome was associated with taking medical retirement or making a new claim for a disabil-ity-related benefit during (but not before) treatment (cog-nitive behavior therapy: χ2=7.9, df=1, p<0.01; entire group: χ2=5.3, df=1, p<0.02). The numbers involved were small and should be interpreted with caution.

Here they imply that people who did not improve had an incentive to not report improvements.

We had only one posttreatment independent assessment, giving a “snapshot” of status at 3-month follow-up. The results of this assessment were consistent with the global self-ratings and the proportions of patients improved, but given the fluctuating nature of chronic fatigue syndrome, more frequent independent assessments (for example, at baseline and posttreatment and each follow-up) and an interview with a relative or significant other may be useful in future studies.

This is interesting. The results of the independent assessment at 3 months are not reported in any detail. This paragraph suggests that the blinded assessor may not have come up with such a clear benefit from the CBT.

The results of this trial are similar to those of the pilot study (11), but two controlled trials (13, 14) showed cognitive behavior therapy to be ineffective. This negative finding could be due to differences in the nature and delivery of the interventions studied. In a nonran-domized comparison of cognitive behavior therapy with a waiting list, graded activity (a key component in the present study) was excluded as it provoked relapse (13). This could reflect a difference in how graded activity was introduced. Often, the first step in our intervention was not to increase activity but to redistribute or even reduce it, interspersing it with sufficient rest. Activity levels were increased only after a consistent, manageable program was established.

A randomized comparison of cognitive behavior therapy and routine clinic attendance (14) produced an unsustained improvement in activity levels (32).

Here they note that not all researchers at this time were finding that the treatment was effective.

 

[Hutan]

I can see how this study would be compelling for doctors and for people considering funding the PACE trial.

For sure, the subjective self-report measures combined with what I would term manipulation, having seen that video by Chalder of an example therapy session would have affected responses. And it is really easy to think about things a bit differently to change the outcome of the SF-36 physical function score from the pre-treatment CBT score to the 6 months score.
See here: What do people think of SF-36?
And its findings were at odds with two other studies undertaken five or so years earlier.

But, there was ongoing improvement after the therapy sessions, out to 6 months. How did they manage that? It seems so much at odds with what we know.

I wonder if the participants had ongoing contact with Chalder? Did any of the participants in this study ever provide any commentary about it?

Those two studies mentioned as not finding CBT effective are interesting:

13. Freidberg F, Krupp LB: A comparison of cognitive behavioral treatment for chronic fatigue syndrome and primary depression.
Clin Infect Dis 1994; 18(suppl 1):S105–S110

14. Lloyd A, Hickie I, Brockman A, Hickie C, Wilson A, Dwyer J, Wakefield D: Immunologic and psychologic therapy for patients
with chronic fatigue syndrome: a double blind, placebo con-trolled trial. Am J Med 1993; 94:197–203

There are some very recognisable names on those papers. They would be worth a closer look.

 

[Utsikt]

But, there was ongoing improvement after the therapy sessions, out to 6 months. How did they manage that? It seems so much at odds with what we know.

Oh, that’s easy:

After 6 months of LC I spent 5 weeks at a rehab center. I exercised every week day, sometimes multiple sessions. They never taught me about PEM or delayed symptoms. I was told to rest between the exercise sessions, but I had to rest way more than the 75 year old open heart surgery patient.

I went from 400 to 600 meters on the 6MWT. I ran uphill intervals the last week. I had loads of fun and terrible symptoms (but that had been my life for 6 months anyways so at least I got to do something now). And I would have said myself that I could do a lot more. I crashed for a few months after getting home to my parents again.

It took me 18 months to realise that I could in fact not keep up with exercising 2-3 times a week. My health had deteriorated to the point of becoming bedridden, which took me another 6 months to realise and accept (so I crashed repeatedly from overexertion from trying to make my own food and walk a total of 3-400 meter daily.

All of this is to say that you might not need any direct brainwashing to get people to answer better on SF36 or any other subjective scale. All you need is to tell them that they have permission to do more - and they will happily oblige simply because they want to. And the perceived effect can last for a long, long time.

From the SF36 thread:
https://www.s4me.info/threads/what-do-people-think-of-sf-36.31504/page-5

 

[Arvo]

But, there was ongoing improvement after the therapy sessions, out to 6 months. How did they manage that? It seems so much at odds with what we know.

That fits with the theoretical idea at the time, when they first started to promote CBT for ME as treatment: that the treatment was cost-effective as it would have ongoing positive effect; they would help the patient over the first bump, after which the improvement would go on because of the Operant Conditioning (so basically the ongoing discovery that continuing activity while feeling sick worked and getting positive reward, expanded activity ability, from it) and the patient having been taught to "correct" their wrong cognitions when they occured.

See Sharpe et al, published just the year before. They also saw ongoing improvement after 4 months, 16 sessions, of CBT:

"The improvement in day to day functioning in the group who received cognitive behaviour therapy continued after treatment had ended. Such a long term effect of cognitive behaviour therapy is consistent with its aim of teaching patients to help themselves and has been observed with depression and with the chronic fatigue syndrome. An increasing effect of treatment after it has been completed is a more unusual finding but has been reported in the treatment ofchronic back pain."

"Evaluations took place before randomisation, after the initial treatment period (five months), at eight months, and, finally, at 12 months after entry.":

 

[Evergreen]

I have to say, on the face of it, with the crucial exception of the subjective outcomes for an unblinded intervention that trains people to report better outcomes, the study is quite well done. And, although people in the CBT group weren't cured, there were significant reported improvements at 6 months.

It is difficult to reconcile the results of this trial with what we know. It is surprising that the improvements continued for so long.

But, there was ongoing improvement after the therapy sessions, out to 6 months. How did they manage that? It seems so much at odds with what we know.

I wonder if the participants had ongoing contact with Chalder? Did any of the participants in this study ever provide any commentary about it?

I have often wondered about these early studies. Fulcher and White 1997 is similarly promising - the exercise group had a 20 point increase in SF36 PF score at 12 weeks (compared to 8 point increase in the control group). Compare this to the GET group in PACE who had an increase of about 11 points at 12 weeks. Though none of the PACE arms got to the 72 reported for CBT in Deale et al. 1997.

My interpretation is referral/selection bias: these were small studies with 30-ish people per group who had been referred to psychiatrists/CBT therapists with fatigue. While those with current psychiatric disorders were excluded from Fulcher and White, and those with severe depression or somatization disorder were excluded from Deale et al. 1997, I suspect that the group of patients they saw, and see, is quite different to the group of patients who would be referred by their GP to, say, an infectious disease doctor or an immunologist.

The booklet for Chalder and Wessely's clinic can be downloaded here:
https://www.yumpu.com/en/document/v...atigue-service-booklet-slam-national-services

There are two case studies/testimonials at the back, written by patients who benefitted. I don't identify with them at all. I did not benefit from CBT or GET.