Development and validation of the Iranian Minimum Data Set for Epidermolysis Bullosa: A mixed method approach

Somayeh Paydar; Shahrbanoo Pahlevanynejad; Farkhondeh Asadi; Hamideh Ehtesham; Azam Sabahi

doi:10.1371/journal.pone.0316791

Peer Review History

Original SubmissionNovember 2, 2023
11 Oct 2024 Decision Letter - Bibi Razieh Hosseini Farash, Editor PONE-D-23-32911Development and validation of the Iranian Minimum Data Set for Epidermolysis Bullosa: A mixed method approachPLOS ONE Dear Dr. Sabahi, 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 25 2024 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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Please see our Supporting Information guidelines for more information: http://journals.plos.org/plosone/s/supporting-information. Additional Editor Comments: Major Issues: Lack of Clarity in Methods: The manuscript states the use of the Delphi technique, but the detailed methodology lacks clarity. There is no comprehensive explanation of the number of participants in each Delphi round, nor a clear rationale for why two rounds were sufficient. This leaves questions about the robustness of the Delphi process used. Limited Generalizability: While the study is focused on developing a Minimum Data Set (MDS) for Epidermolysis Bullosa in Iran, the manuscript does not discuss how the findings can be generalized or applied beyond Iran. Considering PLOS ONE's international audience, it is critical to emphasize how the developed MDS may have broader applications. Inconsistent Structure: There are several instances of structural inconsistency in the presentation of sections. For instance, the transitions between the "Methods" and "Results" sections are not smooth. The "Discussion" could have linked the results better with the broader global context and existing literature. Insufficient Statistical Analysis: The manuscript does not provide sufficient detail on the statistical analysis used to validate the data set. While descriptive statistics are mentioned, there is no mention of more robust statistical methods to ensure the reliability and validity of the identified data elements. Ethical Considerations: The ethical section mentions consent and ethical approval but lacks detail on how data confidentiality and privacy were maintained during the focus group discussions and Delphi surveys. Minor Issues: Language and Grammar: The manuscript contains several minor grammatical errors and awkward sentence structures that can hinder comprehension. For example, the abstract and introduction sections have some overly long sentences that can be difficult to follow. Figures and Tables: The presentation of figures and tables is somewhat unclear. For example, Figure 2's flowchart is not described in detail, and Table 1 lacks clear labels and explanations. Improving their clarity would enhance the readability of the manuscript. References and Citations: While the references seem to be comprehensive, there is an over-reliance on older studies in the discussion section. Including more recent studies would strengthen the manuscript and make it more relevant to current research. Recommendations for Improvement: Clarify the methodology, particularly the Delphi technique, and provide detailed participant numbers and the rationale for the rounds. Discuss the potential international relevance of the Iranian MDS and how it can inform global standards. Improve the flow between sections, particularly by connecting the findings to broader literature in the discussion section. Ensure that the ethical considerations are more thoroughly explained, especially in regard to data privacy and confidentiality. Revise the manuscript for language clarity and structure, and improve the presentation of figures and tables. [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 ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: 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 ****** 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 ****** 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: Comments Let me congratulate the authors for taking the time to research such an important phenomenon. I find the following comments useful for enriching the content of the manuscript. Abstract • Under the abstract on line 33 page 1, the first statement is not clear. Introduction • The authors need to provide some background information on Epidermolysis bullosa in Iran • The authors should also indicate the specific objectives of the study. • An outline of the study should also be provided at the end of the introduction to give readers the direction of the study. Methods • Line 169 page 8, in case of disagreement, how many authors review the papers? The authors need to state the number of authors who review the papers in case the two researchers disagree. • The sentence on lines 186 to 191 on page 9 is too long. Results • The authors should adopt the format (n=..., %) for their results presentation for lines 224 to 226 on page 10 as this (n=..., %) was done on page 16. Discussion • I suggest the authors give a title such as “Strengths and limitations of the study”. • Apart from the validation of data elements using the Delphi method, has the minimum data set tool been tested? If not, can that be captured under the limitation? ****** 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. https://doi.org/10.1371/journal.pone.0316791.r001
Revision 1
5 Dec 2024 Author Response Dear Editor; Thank you very much for considering our manuscript and the opportunity for revision. We would like to express our appreciation for the constructive and valuable comments and suggestions offered by the reviewers. We considered all issues mentioned in the reviewers' comments carefully to improve the quality of our manuscript. All modifications are highlighted in yellow in the revised manuscript. Thank you for updating your data availability statement. You note that your data are available within the Supporting Information files, but no such files have been included with your submission. At this time we ask that you please upload your minimal data set as a Supporting Information file, or to a public repository such as Figshare or Dryad. Authors’ Answer: Minimal data set added as Supporting Information files and cited in manuscript. Major Issues: 1. Lack of Clarity in Methods: The manuscript states the use of the Delphi technique, but the detailed methodology lacks clarity. There is no comprehensive explanation of the number of participants in each Delphi round, nor a clear rationale for why two rounds were sufficient. This leaves questions about the robustness of the Delphi process used. Authors’ Answer: To enhance the clarity of the methodology, we will provide a more detailed explanation of the Delphi technique, the number of participants involved in each round of the Delphi technique and the reason for the two rounds in Step III (Validation of data elements using the Delphi technique) of section method. 2. Limited Generalizability: While the study is focused on developing a Minimum Data Set (MDS) for Epidermolysis Bullosa in Iran, the manuscript does not discuss how the findings can be generalized or applied beyond Iran. Considering PLOS ONE's international audience, it is critical to emphasize how the developed MDS may have broader applications. Authors’ Answer: The generalizability dimension of the study is the possibility of Iran's presence in comparing endemic disease data between different countries, which was added to the introduction according to the opinion of the respected referee. 3. Inconsistent Structure: There are several instances of structural inconsistency in the presentation of sections. For instance, the transitions between the "Methods" and "Results" sections are not smooth. The "Discussion" could have linked the results better with the broader global context and existing literature. Authors’ Answer: The method was edited and presented based on three phases and the results are presented in the order of these three phases. Discussion was revised based on results. 4. Insufficient Statistical Analysis: The manuscript does not provide sufficient detail on the statistical analysis used to validate the data set. While descriptive statistics are mentioned, there is no mention of more robust statistical methods to ensure the reliability and validity of the identified data elements. Authors’ Answer: We have explained both in the method section and in the findings section that the validity of the data was based on the Delphi technique. In this part, the percentage and frequency were important for us to decide whether the data elements should be approved, rejected, or asked again in the second stage. 5. Ethical Considerations: The ethical section mentions consent and ethical approval but lacks detail on how data confidentiality and privacy were maintained during the focus group discussions and Delphi surveys. Authors’ Answer: Details of this section were added. Minor Issues: 1. Language and Grammar: The manuscript contains several minor grammatical errors and awkward sentence structures that can hinder comprehension. For example, the abstract and introduction sections have some overly long sentences that can be difficult to follow. Authors’ Answer: According to the reviewer's opinion, the manuscript was grammatically edited. 2. Figures and Tables: The presentation of figures and tables is somewhat unclear. For example, Figure 2's flowchart is not described in detail, and Table 1 lacks clear labels and explanations. Improving their clarity would enhance the readability of the manuscript. Authors’ Answer: We improved the clarity of figures and tables. Details of Fig 2 explained in result section (3.1. Comprehensive literature review). Details of Table 1 explained in method section (Comprehensive literature review). 3. References and Citations: While the references seem to be comprehensive, there is an over-reliance on older studies in the discussion section. Including more recent studies would strengthen the manuscript and make it more relevant to current research. Authors’ Answer: According to the reviewer's opinion, references reviewed. Reviewer 1 Abstract • Under the abstract on line 33 page 1, the first statement is not clear. Authors’ Answer: According to the reviewer's opinion, the desired corrections were made. Introduction • The authors need to provide some background information on Epidermolysis bullosa in Iran Authors’ Answer: According to the reviewer's opinion, background information on Epidermolysis bullosa in Iran was added to the introduction. • The authors should also indicate the specific objectives of the study. Authors’ Answer: According to the reviewer's opinion, the specific objectives of the study was added to the introduction • An outline of the study should also be provided at the end of the introduction to give readers the direction of the study. Authors’ Answer: According to the reviewer's opinion, an outline of the study was added to the end of the introduction Methods • Line 169 page 8, in case of disagreement, how many authors review the papers? The authors need to state the number of authors who review the papers in case the two researchers disagree. Authors’ Answer: The items mentioned in the method section were added. (line 163-167) • The sentence on lines 186 to 191 on page 9 is too long. Authors’ Answer: The sentence was revised. Results • The authors should adopt the format (n=..., %) for their results presentation for lines 224 to 226 on page 10 as this (n=..., %) was done on page 16. Authors’ Answer: Mentioned format applied for all results. Discussion • I suggest the authors give a title such as “Strengths and limitations of the study”. Authors’ Answer: This section is in the manuscript and was highlighted. • Apart from the validation of data elements using the Delphi method, has the minimum data set tool been tested? If not, can that be captured under the limitation? Authors’ Answer: The data set was extracted through a systematic review of scientific literature and validated by subject matter experts using the Delphi decision-making technique. This is a standard method for determining MDS and is not a limitation. Several studies have been published using this method, for example: 1. Development and validation of the Neonatal Abstinence Syndrome Minimum Data Set (NAS-MDS): a systematic review, focus group discussion, and Delphi technique. 2. Defining the content of a minimal dataset for acquired brain injury using a Delphi procedure. Attachments Attachment Submitted filename: Response to Reviewers.docx https://doi.org/10.1371/journal.pone.0316791.r002
17 Dec 2024 Decision Letter - Bibi Razieh Hosseini Farash, Editor Development and validation of the Iranian Minimum Data Set for Epidermolysis Bullosa: A mixed method approach PONE-D-23-32911R1 Dear Dr. Azam Sabahi, 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 will be generated when your article is formally accepted. Please note, if your institution has a publishing partnership with PLOS and your article meets the relevant criteria, all or part of your publication costs will be covered. Please make sure your user information is up-to-date by logging into Editorial Manager at Editorial Manager® and clicking the ‘Update My Information' link at the top of the page. If you have any questions relating to publication charges, 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, Bibi Razieh Hosseini Farash Academic Editor PLOS ONE Additional Editor Comments (optional): Abstract and Introduction:<o:p></o:p> The abstract is well-structured, covering all key components of the study.<o:p></o:p> The introduction clearly outlines the research problem and the significance of the study.<o:p></o:p> Methodology:<o:p></o:p> The methodology section provides a detailed description of the processes, including the use of the Delphi technique and systematic review.<o:p></o:p> Data selection criteria and inclusion/exclusion parameters are well-defined.<o:p></o:p> Results:<o:p></o:p> Tables and figures are clearly organized, and changes based on reviewer comments have been incorporated.<o:p></o:p> Data categorization into administrative and clinical data aligns well with the study's objectives.<o:p></o:p> Discussion and Conclusion:<o:p></o:p> The discussion effectively analyzes the findings and compares them with previous studies.<o:p></o:p> The conclusion is concise, clear, and aligned with the research objectives.<o:p></o:p> References:<o:p></o:p> The references are up-to-date, relevant, and properly formatted.<o:p></o:p> Reviewers' comments: https://doi.org/10.1371/journal.pone.0316791.r003
Formally Accepted
26 Dec 2024 Acceptance Letter - Bibi Razieh Hosseini Farash, Editor PONE-D-23-32911R1 PLOS ONE Dear Dr. Sabahi, I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now being handed over to our production team. At this stage, our production department will prepare your paper for publication. This includes ensuring the following: * All references, tables, and figures are properly cited * All relevant supporting information is included in the manuscript submission, * There are no issues that prevent the paper from being properly typeset If revisions are needed, the production department will contact you directly to resolve them. If no revisions are needed, you will receive an email when the publication date has been set. At this time, we do not offer pre-publication proofs to authors during production of the accepted work. Please keep in mind that we are working through a large volume of accepted articles, so please give us a few weeks to review your paper and let you know the next and final steps. Lastly, 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 customercare@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. Bibi Razieh Hosseini Farash Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0316791.r004

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