Self-management for chronic widespread pain including fibromyalgia: A systematic review and meta-analysis

Adam W. A. Geraghty; Emma Maund; David Newell; Miriam Santer; Hazel Everitt; Cathy Price; Tamar Pincus; Michael Moore; Paul Little; Rachel West; Beth Stuart

doi:10.1371/journal.pone.0254642

Peer Review History

Original SubmissionNovember 7, 2020
17 Nov 2020 Decision Letter - Tim Mathes, Editor PONE-D-20-35048 Self-management for chronic widespread pain including fibromyalgia: A systematic review and meta-analysis PLOS ONE Dear Dr. Geraghty, 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. see below. Please submit your revised manuscript by Jan 01 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, Tim Mathes Academic Editor PLOS ONE Journal Requirements: When submitting your revision, we need you to address these additional requirements. 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 2. Please provide a quality a publication bias assessment on the studies included in the systematic review. 3. Please amend your list of authors on the manuscript to ensure that each author is linked to an affiliation. Authors’ affiliations should reflect the institution where the work was done (if authors moved subsequently, you can also list the new affiliation stating “current affiliation:….” as necessary). 4. Please include captions for your Supporting Information files at the end of your manuscript, and update any in-text citations to match accordingly. Please see our Supporting Information guidelines for more information: http://journals.plos.org/plosone/s/supporting-information Additional Editor Comments: Your wrote that you use a Random-Effects meta-analysis. However, the figures show that often the fixed-effects model was used. Probably only forgotten to change in RevMan. Before we can send the article to peer review this should be revised. [Note: HTML markup is below. Please do not edit.] Reviewers' comments: [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.0254642.r001
Revision 1
6 Jan 2021 Author Response Additional Editor Comments: "Your wrote that you use a Random-Effects meta-analysis. However, the figures show that often the fixed-effects model was used. Probably only forgotten to change in RevMan. Before we can send the article to peer review this should be revised." Response: Thank you for noticing this error. You are quite right that this was a RevMan issue. This has now been corrected: All figures have been amended accordingly, and numbers reported in the text updated where necessary. https://doi.org/10.1371/journal.pone.0254642.r002
16 Feb 2021 Decision Letter - Tim Mathes, Editor PONE-D-20-35048R1 Self-management for chronic widespread pain including fibromyalgia: A systematic review and meta-analysis PLOS ONE Dear Dr. Geraghty, 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 Apr 02 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, Tim Mathes Academic Editor PLOS ONE [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 Reviewer #2: (No Response) Reviewer #3: 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 Reviewer #2: Yes Reviewer #3: Yes ****** 3. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: No Reviewer #2: Yes Reviewer #3: 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 Reviewer #2: Yes Reviewer #3: 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 Reviewer #2: Yes Reviewer #3: 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: Thanks for the invitation for reviewing the paper. I think this revised version is generally good in methodology, and the results and conclusions are also important for the application of self-management in the management of chronic widespread pain including fibromyalgia. I only suggest that it would be better in methodology to assess the grade of evidence body (the pooled effect size of meta analysis) by using GRADE (Gradings of Recommendations Assessment, Development, and Evaluation). Reviewer #2: Thank you for the opportunity to review this paper. It is a well written and comprehensive systematic review and meta-analysis of self-management interventions used to manage chronic pain, including Fibromyalgia. I have a few minor comments for the authors to consider. 1. Lines 177 – it is not clear who first conducted the initial searches. Was this by the research team members or by the librarian? 2. Lines 173-177 are about the search are repetitive. 3. Lines 756- 758. The authors make an interesting point that further research should be conducted in primary care. Therefore, could the authors make it clearer in the delivery modality section where the SM interventions were delivered? For example, the authors highlight that the studies were outpatient-based (line 276) could the authors clarify whether the studies in their review were conducted in a hospital or community outpatient setting? Reviewer #3: Thank you for the opportunity to review this interesting, well-written, and timely systematic review and meta-analysis on self-management interventions for CWP/Fibromyalgia. In my opinion, this review is excellent, but could be improved in a few, minor ways: 1. It would be helpful if the authors define short- and long-term in the abstract. 2. It would be helpful if the authors define what pain construct they are referring to in the abstract. Pain has many constructs and its only after reading the paper that the reader understands the focus is on pain intensity/severity rather than another construct. 3. I would recommend that the authors proofread the entire review. I found several small editing errors that can distract from an important message of the review. Examples: page 6, line 152; page 10, line 222; page 11, line 227; page 11, line 229; page 13, line 294; page 21, line 375; page 22, line 393 (awkward wording); page 23, line 425, page 31, line 608; 4. Unclear sentence: Page 13, lines 283-285. Please clarify. ****** 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:** Shizheng Du Reviewer #2: No Reviewer #3: 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.0254642.r003
Revision 2
25 Jun 2021 Author Response Response to reviewers Reviewer #1: Thanks for the invitation for reviewing the paper. I think this revised version is generally good in methodology, and the results and conclusions are also important for the application of self-management in the management of chronic widespread pain including fibromyalgia. Response: We thank the reviewer for their positive comments. I only suggest that it would be better in methodology to assess the grade of evidence body (the pooled effect size of meta-analysis) by using GRADE (Gradings of Recommendations Assessment, Development, and Evaluation). Response: We agree, the use of GRADE would improve our methodology. We have now employed the GRADE framework to rate the quality of evidence for our primary outcomes (physical function and pain) and our two comparisons: Self-management vs. usual care/no treatment controls and self-management vs. active comparison. Importantly, in the present review we included studies that both could and could not be meta-analysed. We did this to avoid bias that may occur when review teams only include trials with data suitable for meta-analysis. Murad et al. provide guidance on how to include studies without estimates of effect into the GRADE framework. We followed this approach to assess the quality of evidence that we summarised narratively, as well as our meta-analyses. We now include summary of findings tables (Table 2 and Table 3) on pages 24 to 27, along with full evidence profiles as supporting information (S4 and S5). We have also included the following text in the methods section on pages 10 and 11: “We used the GRADE (Grading of Recommendations Assessment, Development, and Evaluation) approach to rate the quality of evidence in the review for our primary outcomes (29). When using GRADE, evidence on outcomes from RCTs starts as high quality, and reviewers then rate down for limitations (risk of bias), inconsistency, indirectness, imprecision, and publication bias (ratings range from ‘high’, to ‘very low’). Evidence for outcomes can be rated up considering factors such as very large effects and evidence of dose response gradients (29). Whilst this approach is frequently coupled with meta-analysis, Murad et al. (30) show how it can also be used in absence of a quantitative estimate of effect. As such, we included both meta-analysed and narratively reviewed studies when grading the quality of evidence for the primary outcomes. Where estimates and confidence intervals were absent in narratively reviewed studies, we took a cautionary approach and rated down for imprecision. We prepared evidenced profiles and summary of findings tables for our two comparisons: self-management vs. usual care/no treatment, and self-management vs. active comparison. GRADE ratings were agreed though consensus by a sub-team comprising AG, BS, CP and EM” The following text has been included in the results section on page 23: “The quality of the evidence for the review outcomes was rated as low in most cases. Table 2 and Table 3 show our summary of findings for our two main comparisons (self-management vs. usual care and self-management vs. active comparison). Most outcomes were rated down with a combination of serious limitations (risk of bias) and either serious inconsistency, or serious imprecision. Further details can be found in the full evidence profiles provided as supporting information (S4 and S5). Reviewer #2: Thank you for the opportunity to review this paper. It is a well written and comprehensive systematic review and meta-analysis of self-management interventions used to manage chronic pain, including Fibromyalgia. Response: We are grateful for the positive comments on our review. I have a few minor comments for the authors to consider. 1. Lines 177 – it is not clear who first conducted the initial searches. Was this by the research team members or by the librarian? Response: We apologise for this lack of clarity. We have now clarified that systematic review specialist, EM, conducted all searches. On page 6, line 126 and 127 we have added the following: “All searches were conducted by systematic review specialist, EM.” 2. Lines 173-177 are about the search are repetitive. Response: We are not clear on the source of the repetition mentioned. These lines refer to the screening and selection of appropriate titles and abstracts, then the further screening and selection of full text papers. Although similar, both are important parts of the review process. Thus, we have left this section for now. We are happy to amend further following editorial guidance if necessary. 3. Lines 756- 758. The authors make an interesting point that further research should be conducted in primary care. Therefore, could the authors make it clearer in the delivery modality section where the SM interventions were delivered? For example, the authors highlight that the studies were outpatient-based (line 276) could the authors clarify whether the studies in their review were conducted in a hospital or community outpatient setting? Response: Unfortunately, the included studies rarely provided detail on the nature of the outpatient setting (where the groups took place). We have now clarified this in the Table of Characteristics (S3 in supporting information), noting that the setting location is not clear in the majority of cases. A small number of studies did list ‘leisure centre’ or a ‘clinic’ with regard to location, which we have now added to the Table of Characteristics. To include further relevant details related to this reviewer’s helpful point regarding primary care, we now include details of where participants were recruited from. For example, from primary care, hospital rheumatology clinics or community advertisements. We include the following in the ‘Participants’ section on page 13: “Broadly, 46.2% of participants were recruited from rheumatology clinics or hospital specialist settings; 12.8% were recruited from primary care; 15.4% were recruited from a mixture of primary care and rheumatology clinics; 12.8% were recruited from community advertisements, and in a further 12.8% of cases it was not clear where participants were recruited from.” Reviewer #3: Thank you for the opportunity to review this interesting, well-written, and timely systematic review and meta-analysis on self-management interventions for CWP/Fibromyalgia. In my opinion, this review is excellent, but could be improved in a few, minor ways. Response: We appreciate this reviewer’s positive comments. 1. It would be helpful if the authors define short- and long-term in the abstract. Response: We have now done this where short and long term are first used in the abstract on page 2: “Despite some variability in studies narratively reviewed, in studies meta-analysed self-management interventions improved physical function in the short-term, post-treatment to 3 months (SMD 0.42, 95% CI 0.20, 0.64) and long-term, post 6 months (SMD 0.36, 95% CI 0.20, 0.53), compared to no treatment/usual care controls.” 2. It would be helpful if the authors define what pain construct they are referring to in the abstract. Pain has many constructs and its only after reading the paper that the reader understands the focus is on pain intensity/severity rather than another construct. Response: We agree, this is a helpful addition. We now refer to pain intensity in the methods section in the abstract when listing our primary outcomes, on page 2: “Primary outcomes included physical function and pain intensity.” In addition, this useful point regarding constructs led to us reflecting on physical function, our other primary outcome. We have now provided further detail of how physical function was measured in the studies on pages 28-29: “Studies that presented subjective physical function outcomes used a range of measures. The most commonly used included the Fibromyalgia Impact Questionnaire (FIQ) physical functioning subscale [48, 49, 52, 54, 58, 71], and the physical functioning item from the SF-36 [46, 68]. Some studies used the SF-36/8 physical component summary as a primary measure of physical function [32, 55]. Consequently, for consistently, where study authors did not present alternative physical functioning measures, but did present an SF-36/12/8 physical component summary score this was used as a measure of physical functioning.” 3. I would recommend that the authors proofread the entire review. I found several small editing errors that can distract from an important message of the review. Examples: page 6, line 152; page 10, line 222; page 11, line 227; page 11, line 229; page 13, line 294; page 21, line 375; page 22, line 393 (awkward wording); page 23, line 425, page 31, line 608; 4. Unclear sentence: Page 13, lines 283-285. Please clarify. Response: We apologise for these editing errors. We have now carefully worked through the manuscript and corrected or clarified these issues. Additional minor amendments: In addition to the above amendments, as part of the revision process we undertook a further full accuracy check on all data included. As part of this process, we noted some minor corrections were needed to the study sample sizes in the meta-analyses. These corrections were made, and all relevant meta-analyses were rerun. All relevant figures have been updated. We also noted that two studies needed to come out of the FIQ long term analysis; on further consideration we felt they did not meet our definition of long-term. This reduced the number included in the analysis to below the number required for funnel plot asymmetry and applying Egger’s test. We had included McBeth et al.’s 2012 results on physical function in our narrative section on self-management interventions vs. active comparisons. However, on review as part of the revision process, we added the quantitative data from this study on physical function to the relevant meta-analysis. McBeth et al.’s measure of chronic pain grading has also been added to the relevant section. We have tracked all amendments for ease of review. The above additions do not change the original interpretation of the review. Attachments Attachment Submitted filename: Response to reviewers R2.docx https://doi.org/10.1371/journal.pone.0254642.r004
1 Jul 2021 Decision Letter - Tim Mathes, Editor Self-management for chronic widespread pain including fibromyalgia: A systematic review and meta-analysis PONE-D-20-35048R2 Dear Dr. Geraghty, 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, Tim Mathes Academic Editor PLOS ONE Additional Editor Comments (optional): Reviewers' comments: https://doi.org/10.1371/journal.pone.0254642.r005
Formally Accepted
7 Jul 2021 Acceptance Letter - Tim Mathes, Editor PONE-D-20-35048R2 Self-management for chronic widespread pain including fibromyalgia: A systematic review and meta-analysis Dear Dr. Geraghty: 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. Tim Mathes Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0254642.r006

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