PONE-D-20-16971

Psychological therapies for adolescents with Borderline Personality Disorder (BPD) or BPD features – a systematic review of randomized clinical trials with meta-analysis and Trial Sequential Analysis

PLOS ONE

Dear Dr. Jørgensen,

Congratulations on having conducted an excellent systematic review on a very important topic. As you can see, one reviewer expressed concerns about the co-publication with the recently published Cochrane review on the topic. This review also included the data on adolescents, which technically means that this submission to PlosOne is a co-publication. I would also like to be very clear that you have been transparent about this in your acknowledgements, thank you!

I have then made contact with the Editorial office of PlosOne to clarify the issue of co-publications. Unfortunately, PlosOne does indeed not have a co-publication agreement or policy but only accepts updated reviews or extensions of previous work (including Cochrane reviews). The Editorial office wrote that "they would only consider submissions of this nature if the overlap between the primary results of the submission have not been reported before. We would expect that authors outline how their work advances on their previous publication in the submitted manuscript."

Given these formal reasons, I have to unfortunately send the manuscript back to you. There are two potential options to proceed: (1) you could re-submit the paper and clearly develop an own and less overlapping focus (I do not know whether this is at all possible, and I unfortunately will need to reject the paper if this is not the case, e.g. a simple sub-group analysis seems not sufficient); or (2) you would probably need to withdraw your submission, and submit to a journal with co-publication agreement.

My apologies for not bringing better news on this occasion.

In case you would like to chose option 1, please submit your revised manuscript by Sep 05 2020 11:59PM. If you will need more time than this to complete your revisions, please reply to this message or contact the journal office at plosone@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.

Please include the following items when submitting your revised manuscript:

• A rebuttal letter that responds to each point raised by the academic editor and reviewer(s). You should upload this letter as a separate file labeled 'Response to Reviewers'.
• A marked-up copy of your manuscript that highlights changes made to the original version. You should upload this as a separate file labeled 'Revised Manuscript with Track Changes'.
• An unmarked version of your revised paper without tracked changes. You should upload this as a separate file labeled 'Manuscript'.

If you would like to make changes to your financial disclosure, please include your updated statement in your cover letter. Guidelines for resubmitting your figure files are available below the reviewer comments at the end of this letter.

If applicable, we recommend that you deposit your laboratory protocols in protocols.io to enhance the reproducibility of your results. Protocols.io assigns your protocol its own identifier (DOI) so that it can be cited independently in the future. For instructions see: http://journals.plos.org/plosone/s/submission-guidelines#loc-laboratory-protocols

Kind regards,

Michael Kaess, M. D.

Academic Editor

PLOS ONE

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2. Please provide the full electronic search strategy for at least one database, including any limits used, such that it could be repeated.

3. Please ensure that you refer to Figures 2 and 3 in your text as, if accepted, production will need this reference to link the reader to the figure.

4. We note you have included a table to which you do not refer in the text of your manuscript. Please ensure that you refer to Tables 4 and 5 in your text; if accepted, production will need this reference to link the reader to each Table.

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Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Yes

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2. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

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3. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: No

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4. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

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5. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: Thank you for the opportunity to review your paper. I contacted the PLOS One Editorial Team, concerning the co-publication of this previously published Cochrane review. Unfortunately, (1) PLOS One has no co-publication agreement with Cochrane, and (2) does not accept studies for publication that have already been published, in whole or in part, elsewhere in the peer-reviewed literature. Based on this and my exchange with the Editorial Team, I am very sorry, but I have to reject the paper. Although you present additional data, the overlap between the two manuscripts is obvious. I suggest to check on other journals with existing co-publication agreement, and resubmit your paper there.

Journals with Cochrane Co-Publication agreements

https://documentation.cochrane.org/display/EPPR/Agreements+with+journals+for+the+co-publication+of+Cochrane+Reviews

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6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #1: No

[NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. Please log into your account, locate the manuscript record, and check for the action link "View Attachments". If this link does not appear, there are no attachment files.]

While revising your submission, please upload your figure files to the Preflight Analysis and Conversion Engine (PACE) digital diagnostic tool, https://pacev2.apexcovantage.com/. PACE helps ensure that figures meet PLOS requirements. To use PACE, you must first register as a user. Registration is free. Then, login and navigate to the UPLOAD tab, where you will find detailed instructions on how to use the tool. If you encounter any issues or have any questions when using PACE, please email PLOS at figures@plos.org. Please note that Supporting Information files do not need this step.

PONE-D-20-16971R1

Psychological therapies for adolescents with Borderline Personality Disorder (BPD) or BPD features – a systematic review of randomized clinical trials with meta-analysis and Trial Sequential Analysis

PLOS ONE

Dear Dr. Jørgensen,

Thank you for submitting your manuscript to PLOS ONE. After careful consideration, we feel that it has merit but does not fully meet PLOS ONE’s publication criteria as it currently stands. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process.

Please submit your revised manuscript by Jan 09 2021 11:59PM. If you will need more time than this to complete your revisions, please reply to this message or contact the journal office at plosone@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.

Please include the following items when submitting your revised manuscript:

• A rebuttal letter that responds to each point raised by the academic editor and reviewer(s). You should upload this letter as a separate file labeled 'Response to Reviewers'.
• A marked-up copy of your manuscript that highlights changes made to the original version. You should upload this as a separate file labeled 'Revised Manuscript with Track Changes'.
• An unmarked version of your revised paper without tracked changes. You should upload this as a separate file labeled 'Manuscript'.

If you would like to make changes to your financial disclosure, please include your updated statement in your cover letter. Guidelines for resubmitting your figure files are available below the reviewer comments at the end of this letter.

If applicable, we recommend that you deposit your laboratory protocols in protocols.io to enhance the reproducibility of your results. Protocols.io assigns your protocol its own identifier (DOI) so that it can be cited independently in the future. For instructions see: http://journals.plos.org/plosone/s/submission-guidelines#loc-laboratory-protocols

We look forward to receiving your revised manuscript.

Kind regards,

Michael Kaess, M. D.

Academic Editor

PLOS ONE

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Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #2: All comments have been addressed

Reviewer #3: (No Response)

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2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #2: Yes

Reviewer #3: Partly

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3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #2: Yes

Reviewer #3: No

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4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #2: Yes

Reviewer #3: Yes

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5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #2: Yes

Reviewer #3: Yes

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6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #2: I appreciate your work that is based on a sound methodology and focused on an area of research with high clinical significance.

One critical comment from my side: You have made extreme efforts to demonstrate potential biases of the publications included in this meta-analysis. This is very helpful to classify the results. However, I would appreciate if you would have been more critical concerning the question which trial should be included in the analysis. You have included three trials (Santisteban, Salzer and McCauley) with no structured assessment of relevant constructs, especially BPD severity (see table 3). Even if the number of trials in the field of BPD in adolescents is sparse, having conducted a RCT should not be the predominant criterion. To give an example: The trial of Salzer et al. (2014) was focused on mixed disorders of conduct and emotions (F92) according to DSM-IV, and some of the patients were retrospectively classified as suffering from BPD. No results of this trial are mentioned in your paper except GAF, and there are even no data on attrition. Why didn´t you exclude this trial from your meta-analysis?

As the inclusion of these three trials does not affect your results, I recommend publication without further revision.

Reviewer #3: The manuscript deals with a highly relevant question of the efficacy of BPD-specific psychological therapies for adolescents. The review is carried out at the highest standard possible at the moment and most welcome to inform researchers and cinicians about how to proceed in research and treatment.

There is a striking lack in suffiently powered studies and studies of at least good quality. The authors make a very convincing analysis of the flaws in the current data about this topic especially with regard to risk of bias and attrition. This is a kind of warning for future research in this area to plan for high drop-out and obviously small effect sizes if any (not very encouraging..).

Although I highly value this scientific approach, I would question some central decisions made by the authors that I have problems to follow.

First of all, it remained unclear to me, how the final data set was selected. Maybe going back and forth to the just published cochrane is confusing but also in the flow chart some confusion remains about that. The search resulted in 6 studies fulfilling criteria (flow chart) and then you added another 4 and one because of the main author.. What was the rationale on this additional inclusion process and why did those studies not appear in your search and did those also fulfill inclusion criteria (should be changed in the flow chart as it sounds as if those would not fulfill criteria).

Regarding your statistical analysis: Did you include only the subsamples of BPD patients or the whole samples once the study was included. This goes a bit back and forth in your description and thus remains unclear, e.g. the attrition rates: are they related to the full samples or only the BPD/ BPD feature patients? Some studies had very low BPD percentages.. This is also not mentioned in the results part at all and not mentioned in the limitations.

My biggest problem is the pooling of data on the basis of 2 studies and with such high heterogeneity (I2>50%). I would strongly recommend to reconsider this analytic strategy as you are comparing apples and pears here. Thus, I would highly recommend to report only on single outcomes which could be justified by the high heterogeneity of studies and also settings. MBT-A in Roussow & Fonagy compared to MBT-G in Beck is very different and to pool these together is in my eyes misleading (content-wise and from a statistical point of view).

As you are very critical with the former review that pooled data with high heterogeneity, you should apply this to your data as well.

Thus, I would recommend to point out in the summary more strongly that conclusions on the efficacy of BPD specific psychological therapies can not be derived from such highly biased, low attrition and underpowered studies.. But it would be helpful to learn if changes pre-post were present in both treatments (experimental and control) which would mean that both treatments were equally effective and not that nothing works (what it looks like now). Maybe in adolescence many things work? I just want you to reflect in the discussion a bit more on problematic conlusion that could be drawn from a false or biased reading of these results.

Thank you for this important work, I hope that my comments help the improve the manuscript.

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7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #2: No

Reviewer #3: Yes: Svenja Taubner

[NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. Please log into your account, locate the manuscript record, and check for the action link "View Attachments". If this link does not appear, there are no attachment files.]

While revising your submission, please upload your figure files to the Preflight Analysis and Conversion Engine (PACE) digital diagnostic tool, https://pacev2.apexcovantage.com/. PACE helps ensure that figures meet PLOS requirements. To use PACE, you must first register as a user. Registration is free. Then, login and navigate to the UPLOAD tab, where you will find detailed instructions on how to use the tool. If you encounter any issues or have any questions when using PACE, please email PLOS at figures@plos.org. Please note that Supporting Information files do not need this step.

Psychological therapies for adolescents with Borderline Personality Disorder (BPD) or BPD features – a systematic review of randomized clinical trials with meta-analysis and Trial Sequential Analysis

PONE-D-20-16971R2

Dear Dr. Jørgensen,

We’re pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it meets all outstanding technical requirements.

Within one week, you’ll receive an e-mail detailing the required amendments. When these have been addressed, you’ll receive a formal acceptance letter and your manuscript will be scheduled for publication.

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Kind regards,

Michael Kaess, M. D.

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Reviewers' comments: