PLOS ONE has issued an expression of concern after the authors of a controversial study about chronic fatigue syndrome declined to share some of their data.

In an unusual move, the journal included the authors’ response to the expression of concern (EOC), in which they strongly argue against the notice, and “do not accept that it is justified.”

In 2015, following public requests to review the data, the journal issued an “editor’s note” on the paper, noting the journal’s policy that authors make data and materials available.

There have been numerous requests for data from the “PACE” trial, as the clinical trial is known. Patients and advocates have long disputed the results, arguing that suggesting cognitive behavior and graded exercise therapy could cause harm.

In the latest notice, the journal says it consulted two editorial board members about the paper, a 2012 sub analysis of a controversial clinical trial on chronic fatigue syndrome. The journal then asked the authors to provide the data behind five tables, which would enable researchers to replicate the cost-effectiveness analyses the authors report for different therapies — including graded exercise therapy, which some patient advocates believe could be harmful.

As with previous requests for data, the authors refused to provide it, citing patient confidentiality and consent. The notice explains:

In consideration of the requirements for ethical oversight of data access that may apply to datasets involving human subjects, we contacted the authors and Queen Mary University of London, where the dataset is held, to request that steps be taken to develop a mechanism that would allow requests for data to be independently reviewed and the data released in accordance with our policy while respecting patient privacy. The authors and Queen Mary University of London shared the data policy in place at the institution, however we consider that aspects of the existing framework impose limitations and conditions not aligned with our editorial policy.

The authors proposed a compromise, according to the journal:

The authors have offered to release aggregated data from the study but have reiterated reservations about the public release of individual-level patient data. The journal policy does not require public release of anonymised patient-level data, but does require a suitable framework for data access for the purpose of academic, non-commercial research. While the release of summarized data does not fully comply with the journal requirements, we welcome that the authors are now willing to share summarized data, and we will provide this once it is made available to us. In spite of requests to the authors and Queen Mary University of London, we have not yet received confirmation that an institutional process compatible with the existing PLOS data policy at the time has been developed or implemented for the independent evaluation of requests for data from this study. We conclude that the lack of resolution towards release of the dataset is not in line with the journal’s editorial policy and we are thus issuing this Expression of Concern to alert readers about the concerns raised about this article.

Alongside the journal’s EOC is a strongly worded critique from the authors of the study, including last author Peter White at Queen Mary University of London, UK:

We disagree with the Expression of Concern about our health economic paper that PLOS ONE has issued and do not accept that it is justified. We believe that data should be made available and have shared data from the PACE trial with other researchers previously, in line with our data sharing policy. This is consistent with the data sharing policies of Queen Mary University of London, and the Medical Research Council, which funded the trial. The policy allows for the sharing of data with other researchers, so long as safeguards are agreed regarding confidentiality of the data and consent as specified by the Research Ethics Committee (REC). We have also pointed out to PLOS ONE that our policy includes an independent appeal process, if a request is declined, so this policy is consistent with the journal’s policy when the paper was published. During negotiations with the journal over these matters, we have sought further guidance from the PACE trial REC. They have advised that public release, even of anonymised data, is not appropriate. As a consequence, we are unable to publish the individual patient data requested by the journal. However, we have offered to provide key summarised data, sufficient to provide an independent re-analysis of our main findings, so long as it is consistent with the REC decision, on the PLOS ONE website. As such we are surprised by and question the decision by the journal to issue this Expression of Concern.

A spokesperson for PLOS told us this is the first time the journal has included a statement from the authors in an EOC:

This has been a complex case involving many stakeholders and we wanted to document the different aspects of the case in a fair manner.

He pointed to this post on the PLOS site today, by the editor and managing editor of PLOS ONE, which explains further about the journal’s decision to issue the EOC, and the importance of data sharing.

We asked if the journal plans to retract the paper if the authors fail to provide what it’s asked for; the spokesperson explained:

At this time, PLOS stands by its Expression of Concern. For now, we have exhausted the options to make the data available in accordance with our policy at the time, but PLOS still seeks a positive outcome to this case for all parties. It is our intention to update this notice when a mechanism is established that allows concerns about the article’s analyses to be addressed while protecting patient privacy. PLOS has not given the authors a deadline.

One of the readers who has requested the data is James Coyne, a psychologist at the University Medical Center, Groningen, who submitted his request 18 months ago (and wrote about it on the PLOS blog site). Although some of the data have been released (to one person under the Freedom of Information Act), it’s not nearly enough to conduct an analysis, Coyne told us:

This small data set does not allow recalculation of original primary outcomes but did allow recalculation of recovery data. Release of the PLOS data is crucial for a better understanding of what went on in that trial. That’s why the investigators are fighting so hard.

Eventually, Coyne began suggesting to PLOS that he would organize public protests and scientific meetings attended by journal representatives.

I think it is the most significant issue in psychotherapy today, in terms of data sharing. It’s a flagrant violation of international standards.

That sentiment has been echoed in a 2015 STAT column by our co-founders Ivan Oransky and Adam Marcus, who noted:

If the information Coyne is seeking is harmful and distressing to the staff of the university — and that’s the university’s claim, not ours — that’s only because the information is in fact harmful and distressing. In other words, revealing that you have nothing to hide is much less embarrassing than revealing that you’re hiding something.

“Adaptive Pacing, Cognitive Behaviour Therapy, Graded Exercise, and Specialist Medical Care for Chronic Fatigue Syndrome: A Cost-Effectiveness Analysis” has been cited 19 times since it was published in 2012, according to Clarivate Analytics’ Web of Science, formerly part of Thomson Reuters.

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