By David Tuller, DrPH
Earlier today, I sent the following e-mail to Dr Fiona Godlee, editorial director of BMJ. I cc’d Carol Monaghan MP, Darren Jones MP, and Nicky Morgan MP. I also cc’d Teresa Allen of the Health Research Authority.
Dear Dr. Godlee—
As you know, I have spent some time criticizing a 2011 BMJ Open study involving the use of school absence records to identify children with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME, or what many now refer to as ME/CFS). The investigators exempted the study from ethical review on the false grounds that it was “service evaluation” rather than “research.” To support this claim, they cited an unrelated 2007 letter from the regional research ethics committee.
The school absence study was conducted by investigators from the University of Bristol. As it turns out, members of this team of investigators have cited the same 2007 REC letter to exempt ten other studies (or parts of studies) from ethical review on similar grounds. Five of them, including the school absence study, were published in BMJ journals—two in BMJ Open, and three in Archives of Disease in Childhood. The first was published in 2009, with others following in 2011, 2013, 2015 and 2017.
(I am cc-ing Carol Monaghan MP, Darren Jones MP, and Nicky Morgan MP. All three are involved in parliamentary inquiries into issues related to ME/CFS, including some of the questionable research that has characterized this field. I am also cc-ing Teresa Allen from the Health Research Authority.)
None of these five BMJ papers appear to meet the criteria for “service evaluation” studies, which are generally designed solely to assess the provision of services through the use of anonymized databases. The BMJ Open school absence study, in contrast, featured a formal hypothesis, in-person collection of participant data, and generalizable conclusions. These are all hallmarks of “research” and not “service evaluation,” according to the UK Health Research Authority, the relevant government agency. Studies defined as “research” require ethical review.
Here are the five Bristol studies that cited the 2007 REC letter to exempt themselves from ethical review and were published in BMJ journals:
Association between school absence and physical function in paediatric chronic fatigue syndrome/myalgic encephalopathy. Archives of Disease in Childhood, 2009.
Unidentified Chronic Fatigue Syndrome/myalgic encephalomyelitis (CFS/ME) is a major cause of school absence: surveillance outcomes from school-based clinics. BMJ Open, 2011.
Depression in paediatric chronic fatigue syndrome. Archives of Disease in Childhood, 2013.
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, 2015.
Obesity in adolescents with chronic fatigue syndrome: an observational study. Archives of Disease in Childhood, 2017.
Here is the background:
In 2007, the North Somerset and South Bristol REC approved a request to add questionnaires to those already being used during pediatric care at the Bath CFS/ME clinical service. At the time, the clinical service was conducting assessments with several questionnaires at entry and at twelve months. The REC application sought approval for the Bath CFS/ME clinical service to collect further assessments at six weeks and six months, explaining that the extra data would be useful for measuring and improving the delivery of care.
The title of the research project associated with the application was: “What happens to children with CFS/ME? The study of a longitudinal cohort of children who access a paediatric CFS/ME service.” After reviewing the application, the REC responded in a letter dated May 1, 2007. The letter stated: “Members [of the REC] considered this project to be service evaluation. Therefore it does not require ethical review by a NHS Research Ethics Committee or approval from the NHS R&D office.”
The 2007 REC letter referred to “this project”—i.e. the expanded schedule of questionnaires for children at the Bath CFS/ME clinical service designed to assess health care delivery. The five BMJ studies that cited the letter to exempt themselves from ethical review would seem to fall beyond the scope of that particular “project.”
Besides the school absence study, BMJ Open published a paper including data from adults patients as well as children. It is hard to understand how an REC letter specifically about pediatric questionnaires could be cited to exempt from ethical review a study involving adults. And the three studies published in Archives of Disease in Childhood did not focus on the evaluation of services. Instead, they presented generalizable conclusions about the illness and its links to other factors, such as depression and obesity.
The pediatric depression study in Archives of Disease in Childhood, for example, concluded the following: “Depression is commonly comorbid with CFS/ME, much more common than in the general population, and is associated with markers of disease severity. It is important to screen for, identify and treat depression in this population.” Studies with generalizable conclusions are normally categorized as “research,” not “service evaluation,” and require ethical review.
It is incumbent upon Bristol University to conduct a thorough examination of this matter. Whatever Bristol does, however, BMJ itself must investigate how it published five studies that exempted themselves from ethical review on questionable grounds, with no apparent oversight–all citing the same 2007 REC letter.
BMJ editors need to ask themselves questions such as:
*What lapses in procedures for assessing and reviewing manuscripts might have allowed these studies to slip through? What red flags were missed?
*What is current policy about fact-checking ethics declarations, especially if they are used to exempt studies from ethical review?
*Have editors been particularly lax in their oversight in cases involving this specific illness? Have editors put too much trust in this particular research team?
*In the case of the 2011 school absence study, the abstract itself provided sufficient information to determine that this was “research” requiring ethical review. Why did editors fail to recognize this? Why hasn’t the journal addressed the issue honestly and apologized for this mistake?
*How does BMJ plan to prevent similar embarrassments in the future? After this misstep, how can BMJ ensure public confidence in its editorial decision-making?
Dr Godlee, since I first wrote to BMJ Open about the school absence study more than a year ago, the journal has dissembled, obfuscated, and evaded responsibility for having published research that inappropriately exempted itself from ethical review. Such a lack of transparency and accountability is unacceptable; it is also antithetical to robust scientific debate. Now it appears that your journals have published four other Bristol studies in a similar predicament as the first.
These editorial lapses are especially troubling when issues involve the health and wellbeing of children. To rectify the problems in this instance, BMJ needs to: 1) Review the facts related to the publication of these five papers; 2) take any necessary corrective action in each case to ensure the accuracy and integrity of the published literature; and 3) make the necessary changes in editorial and reviewing processes and policies.
David Tuller, DrPH
Senior Fellow in Public Health and Journalism
Center for Global Public Health
School of Public Health
University of California, Berkeley