Cost-effectiveness of interventions for medically unexplained symptoms: A systematic review, 2018, Wortman et al

Andy

Andy

Retired committee member
Abstract
BACKGROUND:
In primary and secondary care medically unexplained symptoms (MUS) or functional somatic syndromes (FSS) constitute a major burden for patients and society with high healthcare costs and societal costs. Objectives were to provide an overview of the evidence regarding the cost-effectiveness of interventions for MUS or FSS, and to assess the quality of these studies.

METHODS:
We searched the databases PubMed, PsycINFO, the National Health Service Economic Evaluation Database (NHS-EED) and the CEA registry to conduct a systematic review. Articles with full economic evaluations on interventions focusing on adult patients with undifferentiated MUS or fibromyalgia (FM), irritable bowel syndrome (IBS) and chronic fatigue syndrome (CFS), with no restrictions on comparators, published until 15 June 2018, were included. We excluded preventive interventions. Two reviewers independently extracted study characteristics and cost-effectiveness data and used the Consensus on Health Economic Criteria Checklist to appraise the methodological quality.

RESULTS:
A total of 39 studies out of 1,613 articles met the inclusion criteria. Twenty-two studies reported costs per quality-adjusted life year (QALY) gained and cost-utility analyses (CUAs). In 13 CUAs the intervention conditions dominated the control conditions or had an incremental cost-effectiveness ratio below the willingness-to-pay threshold of € 50,000 per QALY, meaning that the interventions were (on average) cost-effective in comparison with the control condition. Group interventions focusing on MUS (n = 3) or FM (n = 4) might be more cost-effective than individual interventions. The included studies were heterogeneous with regard to the included patients, interventions, study design, and outcomes.

CONCLUSION:
This review provides an overview of 39 included studies of interventions for patients with MUS and FSS and the methodological quality of these studies. Considering the limited comparability due to the heterogeneity of the studies, group interventions might be more cost-effective than individual interventions.
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Open access at https://journals.plos.org/plosone/article?id=10.1371/journal.pone.0205278
 
Rather like the PACE claims to be cost effective, this only has any meaning if the treatments actually work.

Certainly [as] with ME it is unlikely that any MUS approach could provide an effective treatment as it is just plain inappropriate. The treatment may drive patients away from the medical service treating them so inappropriately, therefore appearantly in the short term saving that service money, but it will not overall save anything in relation to the overall cost to society, indeed we are all aware of examples such inappropriate care worsening patients' condition and significantly increasing the overall burden of the condition.

[I should really read the article itself, but I just can't face it. However I am fairly confident that the studies included in this review, again like PACE only looked at short term implications of the intervention and did not address longer term issues like how many people had return to work or were still on benefits after one or two years.]
 
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Response Twisk on Wortman review:

CBT / GET is not effective for CVS, let alone ME

Based on a review of 'treatments' Margreet Wortman ,

Joran Lokkerbol , Tim Olde Hartman (member of the Health Council Committee ME / CFS )

that 'behavioral interventions', such as CBT, GET and 'pragmatic rehabilitation',

be effective for 'medically unexplained physical symptoms' in general

and fibromyalgia, irritable bowel syndrome, and 'CFS' in particular.

That message is very actively propagated by the first author ( click here ).

In a commentary I fill in the evidence that behavioral interventions would be effective for CFS.

The letter could not be published as an article (Plos One does not know 'letters'),

but is; posted by the editors of Plos ONE as a comment on the website.

The 'evidence-base' for 'CFS' includes 8 studies (see studies 2 and 9 below),

of which six would relate to chronic fatigue (CV) and two to CFS.

However, one of those eight studies concerns the 'unsurpassed' Prince trial from 2001 ,

which, according to the rules, appears from the extensive study report

people with chronic fatigue, not patients with CFS (as stated), let alone ME .
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https://translate.google.com/transl...Wortman%20Twisk%202019%201497.htm&prev=search

(bit confused about this write up; Prince trial from 2001, is this a different trial from this one?
https://www.s4me.info/threads/the-prince-trial-s-2015-2018-trudie-chalder.7965/ )
 
This review provides an overview of 39 included studies of interventions for patients with MUS and FSS and the methodological quality of these studies. Considering the limited comparability due to the heterogeneity of the studies, group interventions might be more cost-effective than individual interventions
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Does anyone else find it ironic that in a paper on cost-effectiveness (which presumably cost money to research) the only conclusion they could draw is that one therapist to many patients is cheaper than one to one?
 
The logical extrapolation from the conclusion that group therapy is more cost effective is the bigger the group the better.
Look at it the other way, the smaller dose of therapist each patient gets the better. Homeopathic dose of therapist anyone?
 
Trish said:
The logical extrapolation from the conclusion that group therapy is more cost effective is the bigger the group the better.
Look at it the other way, the smaller dose of therapist each patient gets the better. Homeopathic dose of therapist anyone?
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Or we could lose the therapist altogether and form self help groups... Oh wait a minute that just makes people worse.

I demand research into the ideal therapist to patient ratio and I demand it now, I tell you, now!!!
 
Nice comment @Michiel Tack :thumbup:

Are you able to edit it? I'd assume not but just in case, you missed a "t" I think here, "after the cost-effectiveness study and did no show significant differences between CBT and the natural course of the illness."
 
Sly Saint said:
Response Twisk on Wortman review:
(bit confused about this write up; Prince trial from 2001, is this a different trial from this one?
https://www.s4me.info/threads/the-prince-trial-s-2015-2018-trudie-chalder.7965/ )
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He is presumably referring to the Prins et al. 2001 trial. It is also a Dutch word that translates as prince.

That trial is sometimes referred to as a trial of Fukuda et al, but actually weirdly some of the patients didn't satisfy the Fukuda et al criteria!
 
Dolphin said:
He is presumably referring to the Prins et al. 2001 trial. It is also a Dutch word that translates as prince.

That trial is sometimes referred to as a trial of Fukuda et al, but actually weirdly some of the patients didn't satisfy the Fukuda et al criteria!
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Prins 2001 says participants were identified by the CDC criteria except for the criterion that they needed to have four of the eight other symptoms. So it was an Oxford study but they decided to call it a Fukuda study for reasons unknown.
 
Dolphin said:
He is presumably referring to the Prins et al. 2001 trial. It is also a Dutch word that translates as prince.

That trial is sometimes referred to as a trial of Fukuda et al, but actually weirdly some of the patients didn't satisfy the Fukuda et al criteria!
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They were challenged on this in a letter in the Lancet. This was their reply:
We did assess additional symptoms at baseline. In our sample, 252 patients had the diagnosis CFS and 18 idiopathic chronic fatigue.
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There's a lot to say about that trial by Prins et al. 2001.

1) There was an enormous drop-out rate (28-41%, depending on how you define it). From the 93 who were randomized to CBT, only 55 completed the trial.

2) Objective outcomes such as neuropsychological testing and actigraphy were only reported many years later in separate reviews (you have to look at the references to know it's data from that trial). These did not show a significant difference. Work resumption was reported in the 2001 paper, and there were also no differences between groups.

3) The control group failed, it did worse than the natural course of the illness, so the cost-effectiveness compared CBT to the latter.

4) No follow-up data is reported, which is strange because two people publically stated that such data existed and that these did not show significant results. The first person is Kenneth M Lassesen. He wrote in a letter to the editor, in 2001:

"After hearing a presentation on Judith Prins and colleagues’ study1 at the American Association for Chronic Fatigue Syndrome (AACFS) International Conference, Seattle, in January, 2001, I find disturbing the lack of full disclosure. At the AACFS, a question was asked about the length of benefit for CBT. The presenter stated that the natural course and CBT groups did not differ significantly 3 years after treatment."​

The other person is Elke van Hoof. She wrote in an Editorial in the Journal of Chronic fatigue syndrome, in 2003:

"Most treatment studies that achieve a 1/3rd improved, a 1/3rd no change and a 1/3rd worsened result are observing an ineffective therapy as the changes appear to be a result of the normal course of the disease process. This is consistent with the report by one of the coresearchers that the effects of CBT were no longer present after 3 years (Bleijenberg G, communication, Fifth International Research, Clinical and Patient Conference)."​

5) As noted, there's the issue of selection criteria:

"Patients were eligible for the study if they met the US Centers for Disease Control and Prevention criteria for CFS, with the exception of the criterion requiring four of eight additional symptoms to be present."
​
Which is weird. This research group used to apply to Oxford definition (but that definition differs from the first criteria of the Fukuda-criteria because fatigue has to be the main symptom). Perhaps they realized the CDC criteria had more prestige so it was better to present the results as such. I don't know.

6) The study was funded by "a grant from the Health Insurance Council (College voor Zorgverzekeringen)."


The results were published in the Lancet (for some reason they seem to like these kinds of flawed CBT-trials). When PACE-defenders say there are many other trials who have found similar results, they are talking about flawed trials like this one.
 
dave30th said:
He is presumably referring to the Prins et al. 2001 trial. It is also a Dutch word that translates as prince.

That trial is sometimes referred to as a trial of Fukuda et al, but actually weirdly some of the patients didn't satisfy the Fukuda et al criteria!
Prins 2001 says participants were identified by the CDC criteria except for the criterion that they needed to have four of the eight other symptoms. So it was an Oxford study but they decided to call it a Fukuda study for reasons unknown.
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Michiel Tack said:
5) As noted, there's the issue of selection criteria:

"Patients were eligible for the study if they met the US Centers for Disease Control and Prevention criteria for CFS, with the exception of the criterion requiring four of eight additional symptoms to be present."
Which is weird. This research group used to apply to Oxford definition (but that definition differs from the first criteria of the Fukuda-criteria because fatigue has to be the main symptom). Perhaps they realized the CDC criteria had more prestige so it was better to present the results as such. I don't know.
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@dave30th and @Michiel Tack , you might find this of interest, from the early days of the Dutch "CFS expert" group:


Het Parool, 22nd of July 1989: Jos van der Meer "doubts if the criteria that English and American researchers have drafted are applicable to many of dutch patients. 'I see for example few patients with swollen glands here, to name an example'"


Trouw, 24th of November 1989: Van der Meer: "We are not researching people that are 'a little tired'. It's about patients that are disablingly tired, that is to say: have lost more than half of their energy. Requirement is also that former medical examination has yielded nothing."


In that same newspaper article, actually just before the abovementioned quote, Van der Meer says that because it is at times "very difficult to distinguish" between regular, common occuring fatigue and CFS, they have involved a psychologist in the research. [Which is Bleijenberg. Anyone here thinking he did a good job at that? :cautious: ]

The next interview with this paper shows how the whole biomedical project has derailed, seemingly stemming in large part from Bleijenberg's involvement. Suddenly it mentions McEveredy & Beard, George Beard, Kendell with depression, neaurasthenia and patient's "resistance" against a psych diagnosis, and Straus (who, according to Osler's Web, by this time was already convinced it was hysteria/neurasthenia). (Also DeFreitas and Gunn)


This is what the research team says about the CDC criteria then, Trouw, 9th of February 1991: "The description is too strict. With a definition like that you show too many people with chronic fatigue the door. The majority of our M.E.-patiënts doesn't meet the requirement of having to have 8 of the 11 symptoms. Our starting point is simple, persisting, disabling fatigue that exists longer than a year. The advantage of that limit is that you exclude the 'regular Pfeiffers' with it." (Pfeiffer=mononucleosis, EBV)
 
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Arvo said:
@dave30th and @Michiel Tack , you might find this of interest, from the early days of the Dutch "CFS expert" group:
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Thanks. I think it is sometimes underestimated how influential this Dutch group has been. They have for example published by far the most trials on ME/CFS. I also think they were more radical than Sharpe or Wessely: patients were encouraged to no longer see themselves as CFS patients.

One caveat is that the quotes in those articles probably refer to to the Holmes criteria (the Fukuda criteria were only published in 1994) and I would agree that these were too strict in requiring 8 out of 11 additional symptoms.
 
Arvo said:
In that same newspaper article, actually just before the abovementioned quote, Van der Meer says that because it is at times "very difficult to distinguish" between regular, common occuring fatigue and CFS, they have involved a psychologist in the research. [Which is Bleijenberg. Anyone here thinking he did a good job at that? :cautious: ]
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Ok, so this *points up* wasn't true, given Bleijenberg's past and interests. It was an excuse. Bleijenberg was already doing behavioral treatments for things like "speaking stomach syndrome"(basically if you had a noisy belly) or (I think the english term is) anismus (When you can't relax your pelvic floor muscle to relieve your bowels), the first by teaching a different way of breathing, the second with biofeedback and teaching people (women) how to poop using porridge.*
And he wrote pieces that already carry the root of what is currently known as MUS, like how IBS should be renamed "functional stomach complaints", or even more desired: "non-organically explained stomach complaints", so the diagnosis becomes "per exclusionem", and doesn't lead to expensive, non-helping treatments (in reaction to a guy who knew those treatments to have effect b/c he was actually following the latest research), while it gives the patient the unhelpful thought that the bowel is ill. Giving up the concept of IBS would lead to behavioural intervention (a good thing in his opinion) and the main treatment method of effectively reassuring the patient.

Both with IBS and the anismus there were more clearheaded physicians contradicting him (&colleague), saying he shouldn't rule out physical causes. It's very sad to see what has become of treatment and research of those two items. I haven't looked into it further, but Bleijenbergs views have prevailed above those of well-read, experienced physicians who cared for providing good patient care.

He also wrote a much-referenced paper on how organic and functional stomach complaints can't be told apart by anamnesis and psychological examination. (So, according to him, it means there is no actual difference between organic and functional.)

B/c of all this, I don't believe he was added to the Nijmegen CFS research team because, as they claimed, he was there to tell CFS and common occuring fatigue apart.




*Yes, you read that right. It fits with the whole patronising attitude he has regarding CFS, where he thinks teaching patients how to go to bed on time is a big, fruitful part of the treatment. (He tells it like he's some grand provider of truth or something, instead of "teaching" adults banal stuff that they and everyone knows since they were kids.) I often joke that he thinks patients are "too stupid to poop"(a dutch expression meaning you think someone is very dumb), because that's how he acts and it also captures everyday banal things adults know. So imagine my dark amusement when I found out that he actually taught people how to poop. By letting them stick porridge up there so they could practise "pooping" it out. You can't make this stuff up.
 
Arvo said:
*Yes, you read that right. It fits with the whole patronising attitude he has regarding CFS, where he thinks teaching patients how to go to bed on time is a big, fruitful part of the treatment. (He tells it like he's some grand provider of truth or something, instead of "teaching" adults banal stuff that they and everyone knows since they were kids.) I often joke that he thinks patients are "too stupid to poop"(a dutch expression meaning you think someone is very dumb), because that's how he acts and it also captures everyday banal things adults know. So imagine my dark amusement when I found out that he actually taught people how to poop. By letting them stick porridge up there so they could practise "pooping" it out. You can't make this stuff up.
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Reality literally outdoing satire. This Simpsons joke is a totally childish gag and this guy actually seriously does something that is above and beyond even the most satirical interpretation of that joke, because in the Simpsons episode it's actually followed by Homer writing it all down and saying it makes sense, where instead most patients here would think, even say, that this guy is off his rockers.

rzgbxh6kqke51.jpg
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