Justification of research using systematic reviews continues to be inconsistent in clinical health science—A systematic review and meta-analysis of meta-research studies

Jane Andreasen; Birgitte Nørgaard; Eva Draborg; Carsten Bogh Juhl; Jennifer Yost; Klara Brunnhuber; Karen A. Robinson; Hans Lund

doi:10.1371/journal.pone.0276955

Peer Review History

Original SubmissionJanuary 24, 2022
2 Apr 2022 Decision Letter - Andrzej Grzybowski, Editor Transfer Alert This paper was transferred from another journal. As a result, its full editorial history (including decision letters, peer reviews and author responses) may not be present. PONE-D-22-02383Justification of research using systematic reviews continues to be inconsistent in clinical health science - a systematic review and meta-analysis of meta-research studiesPLOS ONE Dear Dr. Andreasen, 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 May 17 2022 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. 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The PLOS ONE style templates can be found at https://journals.plos.org/plosone/s/file?id=wjVg/PLOSOne_formatting_sample_main_body.pdf and https://journals.plos.org/plosone/s/file?id=ba62/PLOSOne_formatting_sample_title_authors_affiliations.pdf 2. We noticed you have some minor occurrence of overlapping text with the following previous publication(s), which needs to be addressed: - https://www.jclinepi.com/article/S0895-4356(22)00016-6/fulltext In your revision ensure you cite all your sources (including your own works), and quote or rephrase any duplicated text outside the methods section. Further consideration is dependent on these concerns being addressed. 3. We note that you have indicated that data from this study are available upon request. PLOS only allows data to be available upon request if there are legal or ethical restrictions on sharing data publicly. 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For a list of acceptable repositories, please see http://journals.plos.org/plosone/s/data-availability#loc-recommended-repositories. We will update your Data Availability statement on your behalf to reflect the information you provide. 4. We note that you have stated that you will provide repository information for your data at acceptance. Should your manuscript be accepted for publication, we will hold it until you provide the relevant accession numbers or DOIs necessary to access your data. If you wish to make changes to your Data Availability statement, please describe these changes in your cover letter and we will update your Data Availability statement to reflect the information you provide. [Note: HTML markup is below. Please do not edit.] 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 Reviewer #2: Yes ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: Yes Reviewer #2: Yes ****** 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: Yes Reviewer #2: Yes ****** 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 Reviewer #2: Yes ****** 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 this interesting meta-research paper, which is part of a series of papers. Basing new research on systematic reviews is clearly important and has been the subject of a number of reviews. This paper essentially reviews the meta-research in this area, to give a global assessment of the issue taking into account all of the evidence The content of the rest of the series was not made clear, but a decision has been made to publish them singly. I think the short description of the rest of the programme could be expanded a little to put the work in context and help the reader understand how the work fits together. How do the different studies relate, and are other papers needed to put the current work in context? The introduction defines meta-research in broad terms, but it is not until the results that the reader is given a sense of the actual designs included and of relevance to the research question. Were these defined a priori, or were these study designs that fit the broad definition which happened to be found in the search? Are there meta-research designs of relevance to the research question which were not found in the searches? Personally, I would bring a description of the range of study design forward into the introduction, as getting a sense of the sorts of approaches to meta-research of relevance will help non-specialists in this area. I was not clear of the likely designs until quite late in the paper The review methods seemed very rigorous, and I had no major comments on those beyond one clarification. When they said, ‘No study was excluded on the grounds of low quality’, did they mean that no studies were considered so bad, or that as a rule no studies were every going to be excluded on that basis? As noted above, there were a number of study designs included, and all were assessed using the generic risk of bias tool. Presumably some designs are just stronger than others? The survey must be considered a weaker design that the others. Again, this links to the earlier comment about the need for more detail on design of the meta research, which I felt was lost in the use of a generic risk of bias assessment. I did not understand the statement ‘The clinical interpretation of the large heterogeneity is seen in a broad prediction interval with a range from 16 to 71%’ and that needs clarification The discussion is balanced, but there are a few significant issues that are given a fairly cursory consideration and would benefit from greater detail I was interested in the issue of the ‘quality’ of the reviews used. I accept that the data here was not enough for analysis, but felt that the authors (as experts in this area) could be pushed to provide a stronger statement about what criteria should be used by further studies (for example, how do we judge if a review used as the basis for research is a strong basis. How long before a quoted review is too ‘old’?) They acknowledge that ‘the checklist is not yet optimal and a validated risk-of-bias tool for meta-research studies is needed’. Given their experience and expertise, what would that look like, and how would it be best developed and tested? How would it take into account the role of different designs noted above, given variation in the approaches to meta-research they found? I appreciate the simple and elegant assessment of the main findings, but they present only vague statement on the role of design and medical specialities. Is it not possible for them to say more on this, or explore the data more fully? What about change over time, which seems very relevant. I did feel the authors could be pushed a little more here, given that they have a programme of work and must be in a position to present more substantive statements. I think that would add to the contribution of the paper Reviewer #2: The article is on interesting topic but several points needs emphasis: the inclusion criteria should be defined more clearly in the text Systematiic review and meta analysis are relatively new and first papers go to late seventies in previous century. This should be considered when reviewing papers. The risk of redundancy could not be well defined from the meta search papers rather it should be from the original articles . This would not be possible unless a focused issue is chosen as an example. The different disciplines have different research out puts as the basis for systematic reviews which makes the comparison difficult . I realize some studies are based on the disclosure of the authors whether they have used the previous systematic reviews or not . This should be confirmed by evidence . These should be mentioned as the limitations of this work . ****** 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: Yes:** Peter Bower Reviewer #2: 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. https://doi.org/10.1371/journal.pone.0276955.r001
Revision 1
25 Apr 2022 Author Response Response letter to the editor and reviewers, Thank you for the opportunity to revise the manuscript. Thank you to the reviewers for the positive and constructive comments concerning the manuscript. We have now revised the manuscript in accordance with these comments by addressing all issues from the editor and from the reviewers below. Journal Requirements: When submitting your revision, we need you to address these additional requirements. Answer: we have addressed the requirements, see our answers below. 1. Please ensure that your manuscript meets PLOS ONE's style requirements, including those for file naming. The PLOS ONE style templates can be found at https://journals.plos.org/plosone/s/file?id=wjVg/PLOSOne_formatting_sample_main_body.pdf and https://journals.plos.org/plosone/s/file?id=ba62/PLOSOne_formatting_sample_title_authors_affiliations.pdf Answer: We believe we meet the style requirements, including correct file naming. 2. We noticed you have some minor occurrence of overlapping text with the following previous publication(s), which needs to be addressed: - https://www.jclinepi.com/article/S0895-4356(22)00016-6/fulltext In your revision ensure you cite all your sources (including your own works), and quote or rephrase any duplicated text outside the methods section. Further consideration is dependent on these concerns being addressed. Answer: We agree that there are overlap in parts of the methods section with the mentioned publication. The paper was published in the period of this manuscript being in review, we have therefore now referred to the publication in this manuscript. This manuscript and the publication are both part of a series of papers assessing the global status of evidence-based research in clinical health research and therefore the overlap in the methods section was expected. We have thoroughly scrutinized the full manuscript and found no full sentences that are overlapping, except for the methods section. To be sure of this, we further have conducted a legal comparison in MS Words with the mentioned publication and again found no full sentences except in the methods section. This is to our sincere knowledge only in the methods section, please let us know if we are mistaken. 3. We note that you have indicated that data from this study are available upon request. PLOS only allows data to be available upon request if there are legal or ethical restrictions on sharing data publicly. For more information on unacceptable data access restrictions, please see http://journals.plos.org/plosone/s/data-availability#loc-unacceptable-data-access-restrictions. In your revised cover letter, please address the following prompts: a) If there are ethical or legal restrictions on sharing a de-identified data set, please explain them in detail (e.g., data contain potentially sensitive information, data are owned by a third-party organization, etc.) and who has imposed them (e.g., an ethics committee). Please also provide contact information for a data access committee, ethics committee, or other institutional body to which data requests may be sent. b) If there are no restrictions, please upload the minimal anonymized data set necessary to replicate your study findings as either Supporting Information files or to a stable, public repository and provide us with the relevant URLs, DOIs, or accession numbers. For a list of acceptable repositories, please see http://journals.plos.org/plosone/s/data-availability#loc-recommended-repositories. We will update your Data Availability statement on your behalf to reflect the information you provide. Answer: We have uploaded the data set necessary to replicate our study findings in a supplementary file and described the changes to the “Data Availability statement” in the cover letter. 4. We note that you have stated that you will provide repository information for your data at acceptance. Should your manuscript be accepted for publication, we will hold it until you provide the relevant accession numbers or DOIs necessary to access your data. If you wish to make changes to your Data Availability statement, please describe these changes in your cover letter and we will update your Data Availability statement to reflect the information you provide. Answer: We have uploaded the data set necessary to replicate our study findings in a supplementary file and described the changes to the “Data Availability statement” in the cover letter. 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 comments Reviewer #1: 1. Thank you for the opportunity to review this interesting meta-research paper, which is part of a series of papers. Basing new research on systematic reviews is clearly important and has been the subject of a number of reviews. This paper essentially reviews the meta-research in this area, to give a global assessment of the issue taking into account all of the evidence Response: Thank you for this response and that is exactly the purpose. 2. The content of the rest of the series was not made clear, but a decision has been made to publish them singly. I think the short description of the rest of the program could be expanded a little to put the work in context and help the reader understand how the work fits together. How do the different studies relate, and are other papers needed to put the current work in context? Response: We have expanded the text and especially regarding how the work fits together and shows our purpose of taking a global assessment of the on evidence-based research in the following six papers: 1. Meta-research evaluating redundancy and use of systematic reviews when planning new studies in health research – a scoping review 2. A Systematic Review on the Use of Prior Research in Reports of Randomized Clinical Trials 3. Justification 4. Design 5. Context 6. The problem of citation bias – a scoping review We do not have other papers in pipeline at the moment, but we are currently working on a Handbook for Evidence-Based Research to provide tools and models to make it easier for researchers to work evidence- based in their research. Changes to text: This study is one of six ongoing meta-syntheses (four systematic reviews and two scoping reviews) planned to assess the global state of evidence-based research in clinical health research. These are; a scoping review mapping the area broadly to describe current practice and identify knowledge gaps, a systematic review on the use of prior research in reports of randomized controlled trials specifically, three systematic reviews assessing the use of systematic reviews when justifying, designing [14] or putting results of a new study in context, and finally a scoping review uncovering the breadth and characteristics of the available, empirical evidence on the topic of citation bias . Further, the research group is working with colleagues on a Handbook for Evidence-based Research in health sciences. 3. The introduction defines meta-research in broad terms, but it is not until the results that the reader is given a sense of the actual designs included and of relevance to the research question. Were these defined a priori, or were these study designs that fit the broad definition which happened to be found in the search? Are there meta-research designs of relevance to the research question which were not found in the searches? Response: We get your point. A very broad and inclusive definition was defined a priori in the published protocol: “Types of study to be included: We will include meta-research studies (or studies performing research on research)” in order not to miss out on relevant studies, because the research field was quite new and further, we did not identify other meta-research studies to guide our process. Due to our very broad and sensitive search strategy we believe we identified all relevant meta-research studies. Only data regarding justification from original papers were included in our meta-analysis as the study design of a survey of delegates use of systematic reviews to justify their studies, was assessed as seriously subjected to a social desirability bias. Changes to text: Introduction: The present systematic review aimed to identify and synthesize results from meta-research studies, regardless study type, evaluating if and how authors of clinical health research studies use systematic reviews to justify a new study. Methods section, eligibility criteria: Studies were eligible for inclusion if they were original meta-research studies, regardless study type, that evaluated if and how authors of clinical health studies used systematic reviews to justify new clinical health studies. 4. Personally, I would bring a description of the range of study design forward into the introduction, as getting a sense of the sorts of approaches to meta-research of relevance will help non-specialists in this area. I was not clear of the likely designs until quite late in the paper Response: We agree and have made it clear that all meta-research studies regardless design was included. Changes to text: see above. 5. The review methods seemed very rigorous, and I had no major comments on those beyond one clarification. When they said, ‘No study was excluded on the grounds of low quality’, did they mean that no studies were considered so bad, or that as a rule no studies were every going to be excluded on that basis? Response: The latter, as a rule no studies were excluded, as our intention was not to guide clinical practice. This is stated in the manuscript as the last sentence in the Risk-of-Bias Assessment section. No changes are therefore made. 6. As noted above, there were a number of study designs included, and all were assessed using the generic risk of bias tool. Presumably some designs are just stronger than others? The survey must be considered a weaker design that the others. Again, this links to the earlier comment about the need for more detail on design of the meta research, which I felt was lost in the use of a generic risk of bias assessment. Response: We agree on this point, but we did take a very open approach to monitor the field of justification. And we did not range the study designs in a hierarchical order in our “premature” Risk of Bias tool, as we aimed to assess the area and not to provide any clinical recommendations. However, the author group and colleagues are currently working on an improved checklist tool. No further changes to text. 7. I did not understand the statement ‘The clinical interpretation of the large heterogeneity is seen in a broad prediction interval with a range from 16 to 71%’ and that needs clarification Response: We agree that an explanation is appropriate. Changes to text: The clinical interpretation of the large heterogeneity is seen in a broad prediction interval with a range from 16 to 71%, meaning that there is 95% confidence that the results of the next study will be between a prevalence of 16 to 71%. 8. The discussion is balanced, but there are a few significant issues that are given a fairly cursory consideration and would benefit from greater detail Response: We have addressed the issues mentioned below and provided more detail 9. I was interested in the issue of the ‘quality’ of the reviews used. I accept that the data here was not enough for analysis, but felt that the authors (as experts in this area) could be pushed to provide a stronger statement about what criteria should be used by further studies (for example, how do we judge if a review used as the basis for research is a strong basis. How long before a quoted review is too ‘old’?) Response: Very interesting topic to address further, which we have continuously discussed in the author group, but this is both complex and context dependent in specific topics. Therefore, we have chosen not to elaborate further on the topic in the manuscript, to give an appropriate consideration more space is needed. Instead, we have mentioned these considerations as important to address further in future publications as to guide researchers when using systematic reviews to justify. As mentioned earlier, the research group is working with colleagues on a Handbook for Evidence-based Research in health sciences, which will elaborate on the topics in detail. Changes to text in Discussion section: General recommendations in the use of systematic reviews as justification for a new study are difficult as these will be topic specific, however researchers should be aware to use the most robust and methodologically sound of recently published reviews, preferably with á priori published protocols. 10. They acknowledge that ‘the checklist is not yet optimal and a validated risk-of-bias tool for meta-research studies is needed’. Given their experience and expertise, what would that look like, and how would it be best developed and tested? How would it take into account the role of different designs noted above, given variation in the approaches to meta-research they found? Response: We fully agree with you on this topic and the author group and colleagues are currently working on an improved checklist tool. Your suggestion about ranging the study designs is very relevant and will be considered in the author group in this thorough work that we expect to publish in the near future. We find the work requires space and thorough analysis and we therefore have decided this should be published in an independent paper. 11. I appreciate the simple and elegant assessment of the main findings, but they present only vague statement on the role of design and medical specialities. Is it not possible for them to say more on this, or explore the data more fully? What about change over time, which seems very relevant. I did feel the authors could be pushed a little more here, given that they have a programme of work and must be in a position to present more substantive statements. I think that would add to the contribution of the paper Response: The role of design is only considered in relation to that the studies has done meta - research on the topic “justification”. We do not find it was appropriate to explicate more about the roles of medical specialties as the approach in the different studies were very diverse ranging from participants in the survey, to specialties or to specific journals (mostly high ranking) or more broad aimed journals or databases. Change over time is an important and relevant question. We did not address the issue for two reasons. Firstly, most of the papers are published after 2012 and it would be a short timeline to assess. But most importantly, as most of the included studies in our meta-research study were cross-sectional, we would not be able to validly assess change over time with the data at hand. Reviewer comments Reviewer #2 : 1. The article is on interesting topic but several points needs emphasis Response: Thank you. We have answered each point above. 2. The inclusion criteria should be defined more clearly in the text Response: Methods section: we have clarified the inclusion criteria in the methods section. Changes to text: Methods section, eligibility criteria: Studies were eligible for inclusion if they were original meta-research studies, regardless study type, that evaluated if and how authors of clinical health studies used systematic reviews to justify new clinical health studies. 3. Systematic review and meta analysis are relatively new and first papers go to late seventies in previous century. This should be considered when reviewing papers. Response: Yes, it is a fairly new discipline, however it has been recommended to be evidence-based by the use of systematic reviews and meta-analyses for many years. Our aim was therefore to look at meta-research in a broad sense by using previously published studies investigating how large a percentage are using systematic reviews as justification when initiating new health science. 4. The risk of redundancy could not be well defined from the meta search papers rather it should be from the original articles . This would not be possible unless a focused issue is chosen as an example. Response: Risk of redundancy can, in our perspective, be thoroughly assessed by the use of systematic reviews with meta-analyses included, and especially cumulative meta-analyses can pinpoint this in a specific research topic. Therefore, we agree that we cannot point it to a specific field but have taken this meta-research perspective to provide a more global status on the topic. We hope you can follow our reasoning. 5. The different disciplines have different research out puts as the basis for systematic reviews which makes the comparison difficult Response: In this paper, we did not look for the output, but the “input” so to speak, as we assess whether the authors have used justification by using systematic reviews, when initiating a new study in health science. We agree, it is important to define the aim and approach and the outcomes more specifically, if you look into a specific topic. No changes to text. 6. I realize some studies are based on the disclosure of the authors whether they have used the previous systematic reviews or not. This should be confirmed by evidence. These should be mentioned as the limitations of this work. Response: We agree on this point and have clarified in the limitations that we have taken “the face value” reported by the authors in the included studies. Changes to text: Discussion, Strengths and Limitations section: This may raise questions as to the validity of our findings, as the majority of the meta-research studies only provide an indication of the citation of systematic reviews to justify new studies, not whether the systematic review was relevant, recent or of high-quality, or even how the systematic review was used. We did not assess this further either. Attachments Attachment Submitted filename: Response letter_25042022.docx https://doi.org/10.1371/journal.pone.0276955.r002
19 Sep 2022 Decision Letter - Andrzej Grzybowski, Editor PONE-D-22-02383R1Justification of research using systematic reviews continues to be inconsistent in clinical health science - a systematic review and meta-analysis of meta-research studiesPLOS ONE Dear Dr. Andreasen, 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 Nov 03 2022 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: https://journals.plos.org/plosone/s/submission-guidelines#loc-laboratory-protocols. Additionally, PLOS ONE offers an option for publishing peer-reviewed Lab Protocol articles, which describe protocols hosted on protocols.io. Read more information on sharing protocols at https://plos.org/protocols?utm_medium=editorial-email&utm_source=authorletters&utm_campaign=protocols. We look forward to receiving your revised manuscript. Kind regards, Andrzej Grzybowski Academic Editor PLOS ONE Journal Requirements: Please review your reference list to ensure that it is complete and correct. If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references. Any changes to the reference list should be mentioned in the rebuttal letter that accompanies your revised manuscript. If you need to cite a retracted article, indicate the article’s retracted status in the References list and also include a citation and full reference for the retraction notice. [Note: HTML markup is below. Please do not edit.] 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 #1: All comments have been addressed ******** 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 #1: Yes ****** 3. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: Yes ****** 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 #1: Yes ****** 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 #1: Yes ****** 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 #1: I am happy with the responses and thank the authors for their detailed replies, but just had 2 minor issues This probably reflects my ignorance so apologies to the authors, but I still do not understand the relationship between the 95% CI around the pooled percentage, and the 'broad prediction interval' which follows it. Could they add a line to explain? There are some typos remaining. The phrase 'regardless study type' should read 'regardless of study type'. There are some rogue apostrophes in the tables (SR's, RCT's) which need to be edited ****** 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 #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. https://doi.org/10.1371/journal.pone.0276955.r003
Revision 2
21 Sep 2022 Author Response Response letter Thank you for the opportunity to revise the manuscript. Thank you to the reviewer for the relevant comments concerning the manuscript. We have revised the manuscript in accordance with these comments by addressing all issues from the editor and from the reviewers below. 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 #1: I am happy with the responses and thank the authors for their detailed replies, but just had 2 minor issues Response: Thank you very much. This probably reflects my ignorance so apologies to the authors, but I still do not understand the relationship between the 95% CI around the pooled percentage, and the 'broad prediction interval' which follows it. Could they add a line to explain? Response: We have revised and explained more in detail and hope the revised text explains this more clearly. Changes to text: Where the confidence interval showed the precision of the pooled estimate in a meta-analysis, the prediction interval showed the distribution of the individual studies. The heterogeneity in the meta-analysis assessed by I2 was 94%. The clinical interpretation of this large heterogeneity is seen in a the very broad prediction interval ranging from 16 to 71%, meaning that based on these studies there is 95% chance that the results of the next study will show a prevalence between 16 to 71%. There are some typos remaining. The phrase 'regardless study type' should read 'regardless of study type'. Response: Thank you, we have revised as suggested. There are some rogue apostrophes in the tables (SR's, RCT's) which need to be edited Response: Thank you for pointing this out. We have edited this now. On behalf of the author group, Jane Andreasen Attachments Attachment Submitted filename: Response letter 20092022.docx https://doi.org/10.1371/journal.pone.0276955.r004
18 Oct 2022 Decision Letter - Andrzej Grzybowski, Editor Justification of research using systematic reviews continues to be inconsistent in clinical health science - a systematic review and meta-analysis of meta-research studies PONE-D-22-02383R2 Dear Dr. Andreasen, 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. An invoice for payment will follow shortly after the formal acceptance. To ensure an efficient process, please log into Editorial Manager at http://www.editorialmanager.com/pone/, click the 'Update My Information' link at the top of the page, and double check that your user information is up-to-date. If you have any billing related questions, please contact our Author Billing department directly at authorbilling@plos.org. If your institution or institutions have a press office, please notify them about your upcoming paper to help maximize its impact. If they’ll be preparing press materials, please inform our press team as soon as possible -- no later than 48 hours after receiving the formal acceptance. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information, please contact onepress@plos.org. Kind regards, Andrzej Grzybowski Academic Editor PLOS ONE Additional Editor Comments (optional): 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 #1: All comments have been addressed ******** 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 #1: Yes ****** 3. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: Yes ****** 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 #1: Yes ****** 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 #1: Yes ****** 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 #1: (No Response) ****** 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 #1: Yes: Peter Bower ******** https://doi.org/10.1371/journal.pone.0276955.r005
Formally Accepted
21 Oct 2022 Acceptance Letter - Andrzej Grzybowski, Editor PONE-D-22-02383R2 Justification of research using systematic reviews continues to be inconsistent in clinical health science - a systematic review and meta-analysis of meta-research studies Dear Dr. Andreasen: I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department. If your institution or institutions have a press office, please let them know about your upcoming paper now to help maximize its impact. If they'll be preparing press materials, please inform our press team within the next 48 hours. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information please contact onepress@plos.org. If we can help with anything else, please email us at plosone@plos.org. Thank you for submitting your work to PLOS ONE and supporting open access. Kind regards, PLOS ONE Editorial Office Staff on behalf of Dr. Andrzej Grzybowski Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0276955.r006

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