After a request for the original data was denied, PLOS ONE editors have flagged a 2012 sub analysis of a controversial clinical trial on chronic fatigue syndrome with an editor’s note.

The editor’s note — which reads like an Expression of Concern — reiterates the journal’s policy that authors make data and materials available upon request, and notes that staff are following up on “concerns” raised about the study.

There have been numerous requests for data from the “PACE” trial, as the clinical trial is known, which the authors say they have refused in order to protect patient confidentiality. On November 13, James Coyne, a psychologist at the University Medical Center, Groningen, submitted a request for the data from the PLOS ONE paper to King’s College London, where some of the authors were based. According to Coyne’s WordPress blog (he also has a blog hosted by PLOS), the journal asked him to let them know if he “had any difficulties obtaining the data.” He did — KCL denied the request last Friday (the whole letter is worth reading):

The university considers that there is a lack of value or serious purpose to your request. The university also considers that there is improper motive behind the request. The university considers that this request has caused and could further cause harassment and distress to staff.

Last author Peter White at Queen Mary University of London, UK, told us the journal had not asked them to release the data, but he would work with PLOS to address any questions:

We understand PLOS One are following up concerns expressed about the article, according to their internal processes. We will be happy to work with them to address any queries they might have regarding the research.

Here’s the editor’s note for “Adaptive Pacing, Cognitive Behaviour Therapy, Graded Exercise, and Specialist Medical Care for Chronic Fatigue Syndrome: A Cost-Effectiveness Analysis,” in full:

Several readers have raised concerns regarding the analyses reported in this article. We are also aware that there have been requests for the data from this study. The article was published in 2012; the PLOS data policy that applies to the article is that for submissions prior to March 3, 2014, which is outlined here: http://journals.plos.org/…. The policy expects authors ‘to make freely available any materials and information described in their publication that may be reasonably requested by others for the purpose of academic, non-commercial research’. The policy also notes that access to the data should not compromise confidentiality in the context of human-subject research. PLOS ONE takes seriously concerns raised about publications in the journal as well as concerns about compliance with the journal’s editorial policies. PLOS staff are following up on the different concerns raised about this article as per our internal processes. As part of our follow up we are seeking further expert advice on the analyses reported in the article, and we will evaluate how the request for the data from this study relates to the policy that applies to the publication. These evaluations will inform our next steps as we look to address the concerns that have been noted. Competing interests declared: PLOS ONE Staff

We weren’t sure what the last line was referring to, so contacted Executive Editor Veronique Kiermer. She told us that staff sometimes include their byline under “competing interests,” so the authorship is immediately clear to readers who may be scanning a series of comments.

The trial followed 641 patients, and concluded that two forms of therapy that focus on mental aspects of the disorder were safe, and might be more effective than the therapy commonly favored by patients.

Patients and advocates have disputed these claims — arguing, among other things, that the findings may prompt some to believe chronic fatigue is a mental, not a physical, disorder. (No one knows what causes CFS, which can leave patients bedridden; some scientists think that there might be multiple causes.) Further, advocates argue, a program focused on cognitive behavioral therapy or graded exercise therapy — the two treatments supported by the PACE trial — could actually be harmful to patients by encouraging too much exercise. There have been calls from patients and academics alike for a re-analysis of the data.

The PLOS ONE paper compared the cost-effectiveness of various forms of therapy for CFS, and concluded that cognitive behavior therapy could be the most cost-effective approach to treatment. It has been cited 10 times, according to Thomson Scientific’s Web of Knowledge.

In September, we spoke to White about the choice to not release the data behind their findings (we were considering publishing an account of the trial by David Tuller; ultimately we could not agree on an approach). During that previous interview, White told us:

We have yet to release any raw data. It’s part of our patient’s consent form. We promised that we would keep it confidential. We think it’s really important for our trial — A, for if we do a long-term follow up, which I hope we will. B — When you make a promise to participants, and say ‘I’m going to protect your data, I’m not going to release it to any member of the public,’ we keep that promise. We made a promise and we have to keep it.”

In his request to KCL, Coyne explained his plans for the data:

I am interested in reproducing your empirical results, as well as conducting some additional exploratory sensitivity analyses. Accordingly, and consistent with PLOS journals’ data sharing policies, I ask you to kindly provide me with a copy of the dataset in order to allow me to verify the substantive claims of your article through reanalysis.

To Coyne, the stakes are higher than this one trial. In PLOS Blogs post titled “Why the scientific community needs the PACE trial data to be released,” Coyne explains that his reasons for requesting the data go beyond his skepticism of the PACE trial’s conclusions:

The crisis in the trustworthiness of science can be only overcome only if scientific data are routinely available for reanalysis. Independent replication of socially significant findings is often unfeasible, and unnecessary if original data are fully available for inspection.

KCL’s response to Coyne’s request notes the controversy surrounding the trial, including allegations that the scientists have received death threats from activists:

There have been significant efforts to publicly discredit the trial following the release of the first article in the Lancet journal in 2011. Among other public campaigns, there is a Wikipedia page dedicated to criticisms of this project. The campaign has included deeply personal criticism to the researchers involved as well as significant comment around the decisions not to disclose data and information about the project.

We asked Kiermer what the journal will do if the authors refuse to release the data:

We are looking into the matter and we cannot speculate at this point.

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