By David Tuller, DrPH
In 2019, the University of Bristol and the UK’s Health Research Authority issued a report about their joint investigation of 11 papers. The lead investigator of all 11 papers was Professor Esther Crawley, Bristol’s methodologically and ethically challenged pediatrician and grant queen. The report recommended that Professor Crawley correct the ethics statements in all of the papers. But it absolved Professor Crawley of responsibility for her actions, concluding that everything was all just an unfortunate but understandable misunderstanding.
That absolution was a wrong-headed decision. But at least Professor Crawley was asked to make the corrections. That was already some time ago. Last summer–prompted by comments on the Science for ME forum–I double-checked and, sure enough, only a few of the papers had actually been corrected. I alerted the Health Research Authority, which looked into the matter and pressed Bristol to ensure that the other journals followed suit. Since then, the agency has kept me updated about its progress; the other day I received a final report about the matter. I have posted it below, followed by my response.
After I had alerted the HRA last fall, I also took the liberty of myself writing to the various journals that did not publish corrections to find out whether Professor Crawley had indeed contacted them. The results were inconclusive. An editor at one journal indicated that he had received a message from Professor Crawley, and had responded seeking further information, but then had not heard back again from her. An editor at another journal confirmed that it had received a request and acknowledged having dropped the ball on its end. Others did not provide the requested information.
16 March 2023
Dear David Tuller,
Re: Concerns regarding non-compliance with recommendations from the 2019 review jointly commissioned by the HRA and the University of Bristol
Thank you for your email to the Health Research Authority (HRA) dated 30 August 2022, in which you outlined your concerns regarding compliance with recommendations made by the panel of the 2019 publications review jointly commissioned by the HRA and the University of Bristol. We noted your specific concerns that:
• The review recommended that corrections were made to the ethics statements of eleven research papers. Despite this, only four of the eleven papers appear to have been corrected.
We have now considered your concerns in accordance with the HRA ‘Policy and procedure for managing complaints relating to third parties’. Our response is as follows:
- On 5 June 2019, a panel of experts appointed by the HRA and the University of Bristol conducted a review of eleven publications on chronic fatigue syndrome and myalgic encephalomyelitis, which had been published over a period of several years and cited the same Research Ethics Committee (REC) reference.
- The review found that the REC reference cited in the ethics statements of all eleven publications was either inappropriate or inadequate to cover the activity described. The panel concluded that the ethics statements in every case should be amended.
- The panel made specific recommendations for corrections to the ethics statements for each of the eleven papers. The University of Bristol undertook to notify the respective journal editors of the recommended corrections for each of the publications reviewed.
- Following receipt of your complaint, we contacted the University of Bristol to request an update on the progress of the corrections to the ethics statements. The University of Bristol informed us that they wrote to the respective journal editors prior to 31 October 2019 to notify them of the recommended corrections. The corrections were subsequently published for four of the eleven papers:
- Collin, S.M., Nuevo, R., van de Putte, E.M. et al. Chronic fatigue syndrome (CFS) or myalgic encephalomyelitis (ME) is different in children compared to in adults: a study of UK and Dutch clinical cohorts. BMJ Open 5, e008830 (2015). https://doi.org/10.1136/bmjopen-2015-008830
- Crawley E.M., Emond A.M., Sterne J.A.C. Unidentified Chronic Fatigue Syndrome/myalgic encephalomyelitis (CFS/ME) is a major cause of school absence: surveillance outcomes from school-based clinics. BMJ Open 1, e000252 (2011). https://doi.org/10.1136/bmjopen-2011-000252
- Crawley, E., Collin, S.M., White, P.D. et al. CFS/ME National Outcomes Database, Treatment outcome in adults with chronic fatigue syndrome: a prospective study in England based on the CFS/ME National Outcomes Database. QJM 106, 555–565 (2013). https://doi.org/10.1093/qjmed/hct061
- Norris, T., Hawton, K., Hamilton-Shield, J. et al. Obesity in adolescents with chronic fatigue syndrome: an observational study. Arch Dis Child 102, 35–39 (2017). https://doi.org/10.1136/archdischild-2016-311293
- The University of Bristol agreed to contact the journal editors of the seven papers for which corrections had not been published. The University of Bristol wrote to the journal editors in November 2022, to further notify them of the corrections that were recommended in the 2019 review.
- Following the further notifications by the University of Bristol in November 2022, the recommended corrections have been published for the following three papers:
- Bould, H., Collin, S.M., Lewis, G. et al. Depression in paediatric chronic fatigue syndrome. Archives of Disease in Childhood 98, 425–428 (2013). https://doi.org/10.1136/archdischild-2012-303396
- Brigden,A.,Parslow,R.M.,Gaunt,D.etal.Definingtheminimallyclinically important difference of the SF-36 physical function subscale for paediatric CFS/ME: triangulation using three different methods. Health Qual Life Outcomes 16, 202 (2018). https://doi.org/10.1186/s12955-018-1028-2
- Crawley,E.,Sterne,J.A.C.Associationbetweenschoolabsenceandphysical function in paediatric chronic fatigue syndrome/myalgic encephalopathy. Archives of Disease in Childhood 94, 752–756 (2009). https://doi.org/10.1136/adc.2008.143537
• We note with concern that, as at the date of this letter, corrections have not been published for four of the eleven papers for which the review panel made recommendations:
- Collin, S.M., Crawley, E., May, M.T. et al. The impact of CFS/ME on employment and productivity in the UK: a cross-sectional study based on the CFS/ME national outcomes database. BMC Health Serv Res 11, 217 (2011). https://doi.org/10.1186/1472-6963-11-217
- Collin, S.M., Nikolaus, S., Heron, J. et al. Chronic fatigue syndrome (CFS) symptom-based phenotypes in two clinical cohorts of adult patients in the UK and The Netherlands. J Psychosom Res 81, 14–23 (2016). https://doi.org/10.1016/j.jpsychores.2015.12.006
- Crawley, E., Hunt, L. & Stallard, P. Anxiety in children with CFS/ME. Eur Child Adolesc Psychiatry 18, 683–689 (2009). https://doi.org/10.1007/s00787-009-0029-4
- Webb, C.M., Collin, S.M., Deave, T. et al. What stops children with a chronic illness accessing health care: a mixed methods study in children with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME). BMC Health Serv Res 11, 308 (2011). https://doi.org/10.1186/1472-6963-11-308
- We are satisfied that the University of Bristol have fulfilled their undertaking to notify the relevant journal editors of the recommended corrections. It is not within the remit of the University to implement the corrections; this is an editorial decision for the respective journals.
- We are however concerned that four papers remain published in peer-reviewed journals without the corrections that were recommended in the 2019 review. It is important that people can trust research that has been approved by the HRA and the ethics statements of these papers cite a REC reference which the review panel found was either inappropriate or inadequate to cover the activity described.
- Our Chief Executive, Matt Westmore, has today written to the editors of the three journals for which corrections have not been published:
o BMC Health Services Research
o European Child and Adolescent Psychiatry o Journal of Psychosomatic Research
The letter expresses our concern that the papers have not been corrected and notifies the editors that we intend to publish the letter on our website if the papers have not been corrected by 3 April 2023.
We hope that the recommended corrections to the ethics statements of the remaining four papers will be published promptly. However, we do not regulate the journals and implementation of the correction recommendations is an editorial decision for the journal publishers. If any of the remaining four corrections are not published by 3 April 2023, we will publish the letter from our Chief Executive to the respective journal editor on the HRA website so that our expression of concern is a matter of public record.
We hope that this information is helpful to you, and that it provides reassurance that we have given careful consideration to the concerns you raised and have acted within our remit to address them. Any further concerns regarding compliance with the corrections
recommendations made by the panel of the 2019 publications review should be raised with the publishers of the respective journals.
A summary of your complaint and the outcome of our investigation will be published on the HRA website. In line with the principles of the General Data Protection Regulation and the Data Protection Act, we will not publish your name or any other information that is personal to you.
Thank you for taking the time to raise your concerns with us. Yours sincerely,
Complaints Support Manager
Dear Mr Adams–
Thanks for this report. I really appreciate the HRA’s diligent efforts to track this down.
It is of course unfortunate that Professor Crawley did not herself follow up with the journals that did not respond to an initial request from her to correct the ethics statement. Apparently she believed–wrongly–that her obligation ended there and that it was fine to let the entire matter drop at that point. It is also unfortunate that Bristol and the HRA had no follow-up mechanism in place to ensure that the recommendations of their joint investigation of Professor Crawley’s work were implemented.
However, I’m glad it’s now been taken care of and that the record has been corrected–at least in more papers than was the point before the HRA’s efforts.–
David Tuller, DrPH
Senior Fellow in Public Health and Journalism
Center for Global Public Health
School of Public Health
University of California, Berkeley
8 thoughts on “Trial By Error: An Update on the Missing Ethics Corrections in Multiple Esther Crawley Papers”
David, I just gotta say… fuck yeah
Thank you David for your perseverance.
It is just a shame that Prof Crawley did not feel any urgency in retrospectively addressing her own lack of understanding of research ethics. Namely misusing a letter, saying one specific analysis of clinical outcomes from current service provision did not require ethical approval, to unilaterally exempt eleven distinct projects, published between 2009 and 2018, experimenting with novel service developments from any ethical evaluation.
Given these eleven projects were effectively undertaken without any ethical approval you would have thought anyone with a commitment to scientific integrity would have gone back to the relevant ethics committee for retrospective ethical oversight, informed participants that their consent had been obtain under false pretences, allowing them to withdraw approval for use of their data if they felt it necessary, and insisted herself any papers describing projects not gaining that approval and participant consent be withdrawn.
However it would seem that Prof Crawley has left the matter entirely in the hands of her University, who only demanded minimal action, not herself accepting any personal responsibility even now some four years after the matter was drawn to her attention.
Why does everything take so long? It’s like drawing teeth. I agree with David – the HRA and research establishments should have mechanisms in place to ensure that HRA recommendations are fully and rapidly implemented. Patients and patients advocates shouldn’t have to chase these things for them.
From memory, wasn’t this all about data being extracted on quite a scale from vulnerable ME patients – we’re told for ‘service evaluation’ purposes – and then being used for research studies/papers without the informed consent of those patients? What’s to stop other doctors/researchers doing similarly and requesting ‘service evaluation’ data from vulnerable patients, even before they’ve set foot in a clinic, and then using it for their research? The HRA may not agree, but I see this this as a particularly dark chapter in the history of ME.
Thank you, David. Thanks for being on the ball
Our society (in the UK at least) still appears to fawn over doctors, particularly white ones, I think. I seem to remember that one got away with murder -https://pmj.bmj.com/content/80/944/303 . In that article, the editor indicated that medicine and nursing should not be regarded as exceptions when it comes to the potential for bad behaviour, but has anything really changed since? Have the right lessons been learned from that terrible case? I can’t see any evidence that they have – the fawning still goes on and people and key organizations still refuse to accept that medicine and nursing will have bad apples like any other sector. The HRA strikes me as one such organization – it seems so immersed in its fawning and self-congratulation that it has completely lost sight of its original mission – to protect patients and maintain public trust. I’m sure it’s not alone. Medical scandals don’t seem to be enough to end this entrenched obeisance.
Thank you for not letting go and trying to hold her to account.
It looks as though she may have thought after the initial corrections it wouldn’t be followed up any further.
She must be seething. A lesson she needed to learn though.
Thank you David !
Thank you so much, David, for your hard work and follow-up.
Now, to get the PACE Study retracted!
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