Doubt, uncertainty and criticism are the driving forces in science. Ensuring that patients are not harmed in research should be a top priority.
The music therapy study was initially published 9th April 2020. (1). Read about the study in Science Norway: Norwegian researchers have tested music therapy for chronic fatigue.
Michiel Tack also commented on the article, and his eLetter response was published in BMJ: Inaccuray in reporting CEBA part II. The eLetter response I submitted was rejected. Read it here: : Switched outcome measures, cherry-picked results and unfounded conclusions.
On October 1st, 2020 BMJ retracted the article. The editors thanked Michiel Tack in the retraction notice for bringing problematic issues in the paper to their attention.
The same day, an amended version of the article was published with a new identification number. (2) The editing history and Tack’s comment is thus lost. It is stated that the new version is peer-reviewed, however the reviews are not published. The results of the study are worse than initially reported. Read more here: The Music therapy-study was retracted, amended and re-published: The results are worse than first reported.
Unfortunately, in the republished version of the paper, most of the issues raised remain unresolved. I submitted a new eLetter response about this which was rejected by BMJ – again. My response summarizes the facts which the researchers themselves have documented in their own article. Furthermore, the conclusions drawn were already pointed out by peer-reviews of the first draft:
“An analysis shows that the article is full of contradictions and illogical conclusions, and that the results disprove the researchers’ hypotheses.” Read the eLetter response here: Serious concerns, by Nina E. Steinkopf
Michiel Tack has also submitted an eLetter response, not only once, but twice. His response was also rejected. Read the eLetter response here: This is a resubmitted version of a retracted paper and many issues remain unresolved, by Michiel Tack.
In the response, Tack argues that there are inconsistencies in the retraction notice, the paper doesn’t explain that the study was designed to test large effect sizes, there is little data to support the conclusion that the intervention is “feasible and acceptable”, the outcome measure for post-exertional malaise was added post-hoc and that the reported recovery rates risk misleading readers.
I have asked Tack about his opinion about BMJ’s decision. He says:
“I’m disappointed that they rejected my comment without clarification. Although my letter highlights inconsistencies, it is constructive in nature. I don’t think that it violates any of the terms and conditions for electronic letter submission the BMJ has specified on its website.
There’s a discrepancy between the retraction notice where the editors of BMJ Paediatrics Open thank readers from bringing issues to their attention, and the refusal to publish my rapid response on the republished version. It gives the impression that BMJ Paediatrics Open does not want further discussion of this paper. The practice of rejecting e-responses without clarification discourages readers from bringing issues to light.
I still hope that BMJ Paediatrics Open will take the concerns expressed in my comment seriously.”
As per their own statement, “BMJ Paediatrics Open will maintain a rigorous and transparent peer review process and adhere to the highest ethical standards for research conduct.” Furthermore, “……the journal will be a strong advocate for children’s health, both within the UK and overseas. Health professionals not only have the right to act on behalf of children, they have a responsibility to try and ensure that children have the best possible health outcomes.”
In the study, adolescents with chronic fatigue after EBV infection were treated with a mental training program which has many similarities with the pseudoscientific method Lightning Process (LP). The treatment did not only reduce the participants activity level, but it is also manipulative and victim-blaming.
The participants and their parents were told that personality, thoughts and feelings influence the development of the disease.
Long-term fatigue after an infection is described by the researchers as negative illness behaviour, disease attribution and «avoidance». The parents were involved in the therapy because the researchers believed that parents’ influence could strengthen negative disease behaviour and attribution, which in turn could have a negative impact on the prognosis. During the program, the focus was aimed at the personality of the children, the parents and family dynamics. Both participants and parents were thus implicitly made responsible for the «situation» and the outcome of the therapy. And even though the outcome was clearly negative, the researchers conclude that “This study provides important baseline data for a larger trial”.
LP is based on the pseudoscientific methods neuro linguistic programming. BMJ has published one article about a study where the method was used (3). This article has been subject to serious concerns and has a 3,000-word correction notice. 55 scientists, academics, and other experts has appealed to BMJ to retract it. Read more in Statnews: BMJ should retract flawed chronic fatigue syndrome research paper.
In this context it is interesting that dr. Esther Crawley of the University of Bristol, was the lead investigator of the LP-trial. Crawley has published many papers about this illness in BMJ journals and is an associate editor at BMJ Paediatrics Open.
Norwegian health-authorities classifies LP as an alternative method. Recently, a planned study where 120 ME-patients are to be treated with LP has been approved: Controversial study on ME-patients receives ethical approval.
The study design shows that the study is cynical, unethical and indefensible.
In the music therapy-study, the LP-organization Recovery Norway contributed to the study design. They will now also contribute with “user involvement” in the planned LP-study. Lead author of the music therapy-study Vegard B. B. Wyller, and Esther Crawley are also contributing.
If this LP-study is completed, what are the odds that the article will be published in BMJ?
This is all happening at the same time as British health authorities publish the draft guidelines for ME/CFS where LP is especially advised against. Read more here: NICE backtracks on graded exercise therapy and CBT in draft revision to CFS guidance. (4).
Doubt, uncertainty and criticism are the driving forces in science. Ensuring that patients are not harmed in research should be a top priority. We can only hope that BMJ’s editors change their minds and see the value in readers getting involved.
Nina E. Steinkopf
Formerly HSE and Quality Director
Now; ME-patient and writer
1. Malik S, Asprusten TT, Pedersen M, et al Retracted: Cognitive–behavioural therapy combined with music therapy for chronic fatigue following Epstein-Barr virus infection in adolescents: a feasibility study BMJ Paediatrics Open 2020;4:e000620. doi: 10.1136/bmjpo-2019-000620
2. Malik S, Asprusten TT, Pedersen M, et al Cognitive–behavioural therapy combined with music therapy for chronic fatigue following Epstein-Barr virus infection in adolescents: a randomised controlled trial BMJ Paediatrics Open 2020;4:e000797. doi: 10.1136/bmjpo-2020-000797
3. Crawley EM, Gaunt DM, Garfield K, et al Clinical and cost-effectiveness of the Lightning Process in addition to specialist medical care for paediatric chronic fatigue syndrome: randomised controlled trial Archives of Disease in Childhood 2018;103:155-164.
4. NICE backtracks on graded exercise therapy and CBT in draft revision to CFS guidance, BMJ 2020; 371 doi: https://doi.org/10.1136/bmj.m4356 (Published 10 November 2020) BMJ 2020;371:m4356.