Refractory inflammatory arthritis definition and model generated through patient and multi-disciplinary professional modified Delphi process

Hema Chaplin; Ailsa Bosworth; Carol Simpson; Kate Wilkins; Jessica Meehan; Elena Nikiphorou; Rona Moss-Morris; Heidi Lempp; Sam Norton

doi:10.1371/journal.pone.0289760

Peer Review History

Original SubmissionMay 29, 2023
21 Jul 2023 Decision Letter - Ryu Watanabe, Editor PONE-D-23-16246Refractory Inflammatory Arthritis definition and model generated through patient and multi-disciplinary professional modified Delphi processPLOS ONE Dear Dr. Chaplin, 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 Sep 04 2023 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. 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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. Thank you for stating the following financial disclosure: This paper represents independent research funded by the National Institute for Health Research (NIHR) Maudsley Biomedical Research Centre at South London and Maudsley NHS Foundation Trust and King’s College London, in the form of a PhD Studentship (IS-BRC-1215-20018) for the first author (HC). Please state what role the funders took in the study. If the funders had no role, please state: "The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript." If this statement is not correct you must amend it as needed. Please include this amended Role of Funder statement in your cover letter; we will change the online submission form on your behalf. 3. Your ethics statement should only appear in the Methods section of your manuscript. If your ethics statement is written in any section besides the Methods, please move it to the Methods section and delete it from any other section. Please ensure that your ethics statement is included in your manuscript, as the ethics statement entered into the online submission form will not be published alongside your manuscript. 4. 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. 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: The presented manuscript describes in details the Delphi process of establishing the wide definitions concerning refractory inflammatory arthritis. As nicely explained in the introduction, there is a need of defining the refractory disease in children and adults alike, a the study addresses this need in line with the highest standards. My only concern and suggestion is regarding the distinction of polyarticular JIA and RA. While there is an increasing number of papers and opinions which aims to unify the terminology for the inflammatory arthritis in adults and children, there are still many dissimilarities which prevents RA and pJIA to be used interchangeably. Therefore, I suggest to provide a more detailed description of how pJIA patients after they turned 16 were termed by the authors of the paper - as having pJIA or RA. Moreover, there are also some concerns regarding the involvement of pediatric rheumatologists in the study, since as shown in table 1, there was only one (of five rheumatologist) in face-to-face group, with the number not significantly increased in round 1 and 2. Reviewer #2: First of all I need to congratulate the authors to this tremendous and holistic work that they have conducted over the past years. It is a topic of utmost importance, as the authors nicely outline in their introduction. The manuscript is well written in general, but still needs the attention of the reader to follow all steps that have been undertaken to retrieve the presented results. The limitations that came into my mind during reading overlap with those mentioned in the discussion section. My personal opinion does not 100% overlap with the results, but this is not of relevance for the validity and accuracy of the study conduction. The authors further present a vast compendium of supplementary material that helps the in depth reader to better understand the process and results. ****** 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? 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Please note that Supporting Information files do not need this step. https://doi.org/10.1371/journal.pone.0289760.r001
Revision 1
24 Jul 2023 Author Response Dear Dr Watanabe and Reviewers, Thank you for comments and acknowledgment that this papers addresses an important issue, and we welcome the decision of minor revisions. Please find below our comments in reply to the reviewers’ comments and how we have addressed these in the manuscript. Please find tracked changes in the manuscript, otherwise the previously uploaded documents remain unchanged: 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 I have now amended the manuscript to meet PLOS ONE’s style requirements. 2. Thank you for stating the following financial disclosure: This paper represents independent research funded by the National Institute for Health Research (NIHR) Maudsley Biomedical Research Centre at South London and Maudsley NHS Foundation Trust and King’s College London, in the form of a PhD Studentship (IS-BRC-1215-20018) for the first author (HC). Please state what role the funders took in the study. If the funders had no role, please state: "The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript." If this statement is not correct you must amend it as needed. Please include this amended Role of Funder statement in your cover letter; we will change the online submission form on your behalf. Thank you for highlighting this and providing appropriate wording which I have included in the revised cover letter. 3. Your ethics statement should only appear in the Methods section of your manuscript. If your ethics statement is written in any section besides the Methods, please move it to the Methods section and delete it from any other section. Please ensure that your ethics statement is included in your manuscript, as the ethics statement entered into the online submission form will not be published alongside your manuscript. The ethics statement is now only in the Methods Section of the manuscript (Lines 174-175). 4. 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. I have checked all the references and reference [88] has been updated to reflect the most recent erratum. Otherwise the reference list is complete and correct. Review Comments to the Author Reviewer #1: The presented manuscript describes in details the Delphi process of establishing the wide definitions concerning refractory inflammatory arthritis. As nicely explained in the introduction, there is a need of defining the refractory disease in children and adults alike, a the study addresses this need in line with the highest standards. My only concern and suggestion is regarding the distinction of polyarticular JIA and RA. While there is an increasing number of papers and opinions which aims to unify the terminology for the inflammatory arthritis in adults and children, there are still many dissimilarities which prevents RA and pJIA to be used interchangeably. Therefore, I suggest to provide a more detailed description of how pJIA patients after they turned 16 were termed by the authors of the paper - as having pJIA or RA. Moreover, there are also some concerns regarding the involvement of pediatric rheumatologists in the study, since as shown in table 1, there was only one (of five rheumatologist) in face-to-face group, with the number not significantly increased in round 1 and 2. We would like to thank Reviewer 1 for their considered review and would like to address their concerns. Regarding their first concern, I have included the following descriptions as those with PolyJIA were termed as such regardless of age (not RA) in the Introduction and Methods: Lines 139-140 “Although current RD research focuses on Rheumatoid Arthritis (RA), there is clear justification for incorporating those diagnosed with Polyarticular Juvenile Idiopathic Arthritis (PolyJIA) who are now in adulthood, who despite presenting a RD course are commonly excluded from such research therefore outcomes and impact of disease and treatment are not fully understood and under recognised [1, 2].” Line 191 “Firstly, an initial inductive thematic analysis of 25 patient (RA and Adult PolyJIA) and 32 HCP interviews conducted between August 2018 and April 2019 identified 17 components for common experiences of RD and persistent symptoms, whilst an additional seven were patient-specific (including JIA-specific) and four professional-specific [3] (see unpublished thematic map in S1).” Line 243 “Three independent inclusion criteria determined who was invited [4]: (1) Patients with RA/PolyJIA (determined by diagnosis, regardless of age) or HCPs within Rheumatology, (2) involvement with RA/JIA patient organisations or (3) recognized academic career in RD or Persistent symptoms in RA/JIA.” Regarding their second concern, about the number of paediatric rheumatologists, I have now acknowledged this limitation in the Discussion, with the following statement: “There was a predominant bias towards participants from the UK followed by Europe, in particular adult services, and caution is required for applicability of this definition in other countries and for purely paediatric application.” However I would like to also mention that some of paediatric professionals were also categorised in the “Both” group as they indicated they were trained or worked predominantly in both paediatric and adult services which particularly captures those working in adolescent rheumatology. So the total involvement of paediatric rheumatologists was increased in subsequent rounds (doubling the paediatric only numbers), if taking the “Both” category into account. Reviewer #2: First of all I need to congratulate the authors to this tremendous and holistic work that they have conducted over the past years. It is a topic of utmost importance, as the authors nicely outline in their introduction. The manuscript is well written in general, but still needs the attention of the reader to follow all steps that have been undertaken to retrieve the presented results. The limitations that came into my mind during reading overlap with those mentioned in the discussion section. My personal opinion does not 100% overlap with the results, but this is not of relevance for the validity and accuracy of the study conduction. The authors further present a vast compendium of supplementary material that helps the in depth reader to better understand the process and results. We would like to thank Reviewer 2 for their balanced review and supportive comments. We look forward to these minor revisions being accepted and publication of the manuscript in due course. Once again thank you for your comments and suggestions to improve this piece of work. Attachments Attachment Submitted filename: Reply to Reviewers Letter 21.07.2023.docx https://doi.org/10.1371/journal.pone.0289760.r002
25 Jul 2023 Decision Letter - Ryu Watanabe, Editor Refractory Inflammatory Arthritis definition and model generated through patient and multi-disciplinary professional modified Delphi process PONE-D-23-16246R1 Dear Dr. Chaplin, 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, Ryu Watanabe, M.D., Ph.D. Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0289760.r003
Formally Accepted
31 Jul 2023 Acceptance Letter - Ryu Watanabe, Editor PONE-D-23-16246R1 Refractory Inflammatory Arthritis definition and model generated through patient and multi-disciplinary professional modified Delphi process Dear Dr. Chaplin: 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. Ryu Watanabe Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0289760.r004

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