Willingness to participate in, support or carry out scientific studies for benefit assessment of available medical interventions: A stakeholder survey

Julia Stadelmaier; Joerg J. Meerpohl; Ingrid Toews

doi:10.1371/journal.pone.0271791

Peer Review History

Original SubmissionJuly 20, 2021
7 Dec 2021 Decision Letter - Paavani Atluri, Editor PONE-D-21-23551Willingness to participate in, support or carry out scientific studies for benefit assessment of available medical interventions: a stakeholder surveyPLOS ONE Dear Dr. Ingrid Toews 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. Kindly go through the reviewer comments carefully and address them as requested and required. Once the revised manuscript is submitted , we might send the article for re-review before finalizing our decision to publish. Thank you for your time and patience. Please submit your revised manuscript by Jan 21 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, Paavani Atluri 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. During the internal evaluation of your manuscript we noted that informed consent was not necessary for this study. Please could you clarify whether the IRB specifically waived the need for informed consent for the study. 3. Thank you for stating the following in your Competing Interests section: None Please complete your Competing Interests on the online submission form to state any Competing Interests. If you have no competing interests, please state "The authors have declared that no competing interests exist.", as detailed online in our guide for authors at http://journals.plos.org/plosone/s/submit-now This information should be included in your cover letter; we will change the online submission form on your behalf. [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: Partly Reviewer #2: Yes ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: No 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: I have read this manuscript with much interest. This is mostly a descriptive study of participants’ willingness to participate, support or carry out scientific studies for benefit assessment of medical interventions. My comments and concerns are listed below. 1- Where exactly was the study conducted? Was there any geographical limitation? Where participants from a specific city, region, country, etc. 2- The introduction is quite short and choppy. It does not lead well to the objectives of the study. 3- The authors devised a 51-item questionnaire for data collection. There is no information on the validity and reliability of the questionnaire. I believe that no psychometric testing been done on the questionnaire. 4- Participants: There is not much information on who the participants are (except a general statement that they are from patients, patient representatives, scientists, healthcare professionals and representatives, and private sector groups). The authors used snow-ball sampling techniques to recruit further participants, but how the primary participants were identified (e.g., from which hospital, university, etc.)? 5- Please revise Table 1 as: a. Please add row and column totals. b. I would suggest adding row percentage instead of column percentage. c. The last block of the table (i.e., Experience) is quite confusing. I would suggest a separate table for this section with proper title. Reviewer #2: The authors conducted an observational study to investigate the willingness of relevant stakeholder groups to participate or engage in post-entry studies. There are, at any rate, several issues to be addressed as follows. 1) “New, innovative interventions…”, there is an extra comma. 2) Sampling was not described in sufficient details. have the authors applied inclusion and exclusion criteria? How did the author select the target organisations? 3) “The completion rate was 161 calculated using the ratio of people who had submitted the final questionnaire page and those”, have the authors compared the two groups? 4) “For comparison between two groups an independent t-test was 167 used for a response frequency n > 30, and the Mann-Whitney-U-Test for n < 30 162”, this reviewer understands that Excel was used for descriptive analysis but what statistical tool was used for statistical tests? 5) Have the authors checked the effect of age and gender on responses? 6) For reasons that might or might not be under the authors' control, the figures have a low-resolution appearance ******** 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. [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.0271791.r001
Revision 1
21 Jan 2022 Author Response When submitting your revision, we need you to address these additional requirements. We thank you for your advice. We revised our manuscript accordingly. 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 We rechecked our manuscript against the requirements of the journal and revised it where needed. 2. During the internal evaluation of your manuscript we noted that informed consent was not necessary for this study. Please could you clarify whether the IRB specifically waived the need for informed consent for the study. Response: Thanks for highlighting this issue. In its written statement of reasons for the vote, the ethics committee of the Albert-Ludwigs-University pointed out that it was assumed that no personal data but only anonymised data would be collected and evaluated. Accordingly, the need for informed consent was waived. We address this issue by complementing our methods section with the following information: “No personal but only anonymised data were collected and analysed for the purpose of this study. Therefore, no informed consent for the study was necessary according to the standards of the Ethics Committee of the Albert-Ludwigs-University Freiburg and the data protection officer.” (ll.136-139) 3. Thank you for stating the following in your Competing Interests section: None Please complete your Competing Interests on the online submission form to state any Competing Interests. If you have no competing interests, please state "The authors have declared that no competing interests exist.", as detailed online in our guide for authors at http://journals.plos.org/plosone/s/submit-now This information should be included in your cover letter; we will change the online submission form on your behalf. Response: We thank you for the changes you make on our behalf with regard to the statement about competing interests. We state that the authors have declared that no competing interests exist and will make sure that this information is also included where needed during the resubmission Reviewers comments Reviewer 1: I have read this manuscript with much interest. This is mostly a descriptive study of participants’ willingness to participate, support or carry out scientific studies for benefit assessment of medical interventions. My comments and concerns are listed below. Response: Thank you for your feedback and your support in improving this manuscript. 1- Where exactly was the study conducted? Was there any geographical limitation? Where participants from a specific city, region, country, etc. Response: Thank you for pointing out these questions. We did no limit the survey to a specific geographical area. However, since the survey was in German language and recruiting was conducted mainly through German associations and societies, we assume that respondents are German-speaking people in Germany. We added this information in the method section (subheading ‘sampling’) as follows: “Members of the following stakeholder groups were the target population of the survey: (i) patients or patient representatives, (ii) healthcare providers (physicians and health professionals), (iii) scientists and trialists in medical and health science, and (iv) representatives of the medical or pharmaceutical private sector (e.g. medical technology or the pharmaceutical industry). No geographical limitation was applied. However, as the questionnaire was written in German, knowledge of the German language was necessary to participate in the survey.” (ll.148-154) 2- The introduction is quite short and choppy. It does not lead well to the objectives of the study. Response: To improve this section, we have added some informative sentences and moved the second paragraph down. This resulted in a number of edits that can be seen in the accompanying revised manuscript. If you still have concerns with regard to specific aspects within the introduction, we would appreciate more details about suggested changes. 3- The authors devised a 51-item questionnaire for data collection. There is no information on the validity and reliability of the questionnaire. I believe that no psychometric testing been done on the questionnaire. Response: Thank you for your comment. In order to provide more information on the questionnaire we added the following sentences in the method section (subheading ‘questionnaire’): “In the course of the systematic literature searches we did not identify any suitable, validated questionnaires for the specific purpose of our study. Therefore, we designed a de-novo self-administrated questionnaire in German language.” (ll.104-106) “We did not perform psychometric testing to validate our questionnaire.” (l.122) 4- Participants: There is not much information on who the participants are (except a general statement that they are from patients, patient representatives, scientists, healthcare professionals and representatives, and private sector groups). The authors used snow-ball sampling techniques to recruit further participants, but how the primary participants were identified (e.g., from which hospital, university, etc.)? Response: We agree with you and we added more information about the sampling and associations we contacted. Moreover, we attached a list with details of the contacted associations and societies to the S2 Appendix. “A google search was performed to identify relevant associations and societies representing the target stakeholder groups in Germany. In order to recruit patients and patient representatives, the office of patient representatives in the German Federal Joint Committee (Gemeinsamer Bundesausschuss, G-BA), as well as the four organisations that are currently entitled to appoint patient representatives to the G-BA were contacted. Healthcare providers (physicians, dentists, psychologists, nurses, allied health professionals) were contacted via associations representing the interests of their respective profession. Scientists and trialists were recruited via representatives of the German network for evidence-based medicine, the German network for healthcare research and the Clinical Trials Unit of the University of Freiburg. Trade organisation for the medical technology and pharmaceutical industries were approached in order to invite representatives of the private sector. The contacted associations and societies are listed in detail in the S2 Appendix.” (ll.157-168) 5- Please revise Table 1 as: a. Please add row and column totals. b. I would suggest adding row percentage instead of column percentage. c. The last block of the table (i.e., Experience) is quite confusing. I would suggest a separate table for this section with proper title. Response: Thank you for this thoughtful comment. To complement this table, we have added row and column totals and moved information about experience to new created tables (Table 2A and 2B). We did not include row percentages since several respondents identified to belong to more than one stakeholder group, the sum of row percentage does no attain 100 per cent. We added an explanation of this issue as footnote to our table. Reviewer 2: The authors conducted an observational study to investigate the willingness of relevant stakeholder groups to participate or engage in post-entry studies. There are, at any rate, several issues to be addressed as follows. Response: Thank you for your feedback and your support in improving this manuscript. 1) “New, innovative interventions…”, there is an extra comma. Response: Thanks. We made this edit. 2) Sampling was not described in sufficient details. have the authors applied inclusion and exclusion criteria? How did the author select the target organisations? Response: We agree with you. As described above, we added more information about the sampling and the associations we contacted (ll.148-154; ll.157-168). 3) “The completion rate was 161 calculated using the ratio of people who had submitted the final questionnaire page and those”, have the authors compared the two groups? Response: We did not compare the responses of those who submitted the final questionnaire page and the non-completers. First, the completers include seven respondents that were screened out. Second, among the 46 respondents who did not submit the final page, 24 were excluded from our analysis since they did not fulfil the inclusion criteria, that is, they responded to one question only. With 22 respondents remaining (distributed among four stakeholder groups), we observe that this sample size of this subgroup is too small to conduct further post-hoc analyses to obtain meaningful results. 4) “For comparison between two groups an independent t-test was 167 used for a response frequency n > 30, and the Mann-Whitney-U-Test for n < 30 162”, this reviewer understands that Excel was used for descriptive analysis but what statistical tool was used for statistical tests? Response: Thank you for this comment. We used Microsoft Excel for both, descriptive and analytical statistics. We rephrased the sentence in our method section as follows: “All valid responses were tabulated, and statistical analyses were conducted using Microsoft Excel 2010.” (ll.185-186) 5) Have the authors checked the effect of age and gender on responses? Response: Thank you for raising this question. We did not carry out subgroup analyses due to the small size of the sample. We had data from a total of 154 respondents (distributed among different stakeholder groups). From a methodological point of view, we think that this sample size is not large enough for further analyses. We addressed this issue in our result section as follows: “We did not carry out further subgroup analyses on the effect of sociodemographic variables since the number of respondents in each stakeholder group is too small to create further representative subgroups and perform reasonable analyses.” (ll.272-274) 6) For reasons that might or might not be under the authors' control, the figures have a low-resolution appearance Response: Thank you for highlighting this issue. The figure was edited according to the journal’s requirements. In case there are inadequacies, we are happy to make changes to our figure. Attachments Attachment Submitted filename: Response to Reviewers.docx https://doi.org/10.1371/journal.pone.0271791.r002
15 Jun 2022 Decision Letter - Paavani Atluri, Editor PONE-D-21-23551R1Willingness to participate in, support or carry out scientific studies for benefit assessment of available medical interventions: A stakeholder surveyPLOS ONE Dear Dr. Toews, 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 Jul 30 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, Paavani Atluri 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 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 #3: Partly ****** 3. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: 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 #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 #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: I have reviewed the revision. The authors have addressed all of my comments. I have no further comment. Reviewer #3: I wasn't one of the original reviewers, but as far I as understand, the authors have addressed the points raised by the reviewers. I have some minor observations having read the revised paper. 1. Abstract and keywords: from the keywords and abstract, I'm not sure I would pick up that this paper has its roots in HTA, decision-making and MEA's, which is explained clearly in the Introduction. I might miss it in a title and abstract screen for a systematic review of barriers to MEAs, for example. The authors might consider some adjustments to broaden their impact; I leave that to their judgement. 2. Table 2: I found this a bit confusing - were the stakeholders asked about their experience in different units? It might help to explain in the legend. 3. I wasn't sure how the evidence supported that patients preferred observational studies or that healthcare providers were inexperienced - might help to point it out earlier than the Discussion or Conclusion. 4. I think there's an interesting paradox in the discussion, between willingness to participate in a study on an available treatment (because they assume it's therefore OK) and preference to avoid getting placebo when there's a known treatment available (the equipoise issue). The authors could make a stronger case for (their) future work on this if they chose to; again I leave that to their judgement. ****** 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 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.0271791.r003
Revision 2
29 Jun 2022 Author Response We appreciate the valuable comments made by the editor and peer reviewers and their support in improving this manuscript. We have addressed their comments in full and revised our manuscript accordingly. Editors comment. 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. Response: We thank you for your advice. We rechecked our reference list and revised it where needed. We added the corrections of the articles of Sheridan et al. 2020 and Eysenbach 2012 to the reference list: Sheridan R, Martin-Kerry J, Hudson J, Parker A, Bower P, Knapp P. Why do patients take part in research? An overview of systematic reviews of psychosocial barriers and facilitators. Trials. 2020;21(1):840. doi: 10.1186/s13063-020-04793-2. Corrected and republished from: Trials. 2020;21(1):259. doi: 10.1186/s13063-020-4197-3. Eysenbach G. Improving the quality of Web surveys: the Checklist for Reporting Results of Internet E-Surveys (CHERRIES). J Med Internet Res. 2012;14(1):e8. doi: 10.2196/jmir.2042. Corrected and republished from: J Med Internet Res. 2004;6(3):e34. doi: 10.2196/jmir.6.3.e34. Reviewer #3: I have some minor observations having read the revised paper. Response: Thank you for your feedback and your support in improving this manuscript. 1. Abstract and keywords: from the keywords and abstract, I'm not sure I would pick up that this paper has its roots in HTA, decision-making and MEA's, which is explained clearly in the Introduction. I might miss it in a title and abstract screen for a systematic review of barriers to MEAs, for example. The authors might consider some adjustments to broaden their impact; I leave that to their judgement. Response: Thank you for this comment. We added the following phrases to the abstract: “Post-entry studies are a key element in managed entry agreements and aim at generating evidence about the additional benefit of new medical interventions before reimbursement decisions are made.” (ll.20-22) Moreover, we added “managed entry agreements” to the list of keywords. 2. Table 2: I found this a bit confusing - were the stakeholders asked about their experience in different units? It might help to explain in the legend. Response: Thank you for raising this question. The item ‘experience’ differed between stakeholder groups: We asked patients, healthcare providers and scientists about the number of participation/ engagements in studies of medical research, whereas for patient representatives and representatives of the private sector we asked for their experience in medical research in years. We added this information to the methods section as follows: “(iv) demographic and professional background including experience in years (for patient representatives and representatives of the private sector) or number of engagements in studies of medical research (for patients, healthcare providers and scientists)”(ll.119-122) Furthermore we redefined the title and added a legend to Table 2A and 2B: Table 2A. Engagement of the respondents in medical research (in number of studies)a Table 2B. Experience of the respondents in medical research (in years)a a Wording and scales of the item ‘experience’ differed between the respective stakeholder groups. (ll. 224-227) 3. I wasn't sure how the evidence supported that patients preferred observational studies or that healthcare providers were inexperienced - might help to point it out earlier than the Discussion or Conclusion. Response: Thank you for highlighting this issue. Information about the characteristics of the respondents is displayed in Table 1, 2A and B. We agree with you in highlighting some results regarding the characteristics in the text, and added the following phrases: “The majority of respondents were female (52.6%), aged between 35 and 54 years (46.8%), and reported to have little experience with regard to participation or engagement in medical research.” (ll. 228-230) Differences in the willingness to participate or engage in RCTs or observational studies are already described in the results section ‘Willingness with regard to study type’ (ll.251ff). Since significant differences were only observed in the group of the scientists, we toned down our wording in the conclusion section to the following: “Study type was found to be less relevant to stakeholders when considering willingness to engage in a study. However, we observed tendencies that the scientific community and healthcare providers reported a higher willingness to engage in RCTs, but patients and patient representatives reported to prefer OSs.” (ll. 413-416) 4. I think there's an interesting paradox in the discussion, between willingness to participate in a study on an available treatment (because they assume it's therefore OK) and preference to avoid getting placebo when there's a known treatment available (the equipoise issue). The authors could make a stronger case for (their) future work on this if they chose to; again I leave that to their judgement. Response: Thank you for this comment. We added the following: “Future research that specifies details of the comparator (active or inactive) might therefore be indicated. This could contribute to resolving the paradox between high willingness of study participation as reported in our survey and other research findings that demonstrate a reluctance of study participation in general.” (ll. 354-357) Attachments Attachment Submitted filename: Response to Reviewers.docx https://doi.org/10.1371/journal.pone.0271791.r004
8 Jul 2022 Decision Letter - Paavani Atluri, Editor Willingness to participate in, support or carry out scientific studies for benefit assessment of available medical interventions: A stakeholder survey PONE-D-21-23551R2 Dear Dr. Toews, 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, Paavani Atluri Academic Editor PLOS ONE Additional Editor Comments (optional): Reviewers' comments: https://doi.org/10.1371/journal.pone.0271791.r005
Formally Accepted
3 Aug 2022 Acceptance Letter - Paavani Atluri, Editor PONE-D-21-23551R2 Willingness to participate in, support or carry out scientific studies for benefit assessment of available medical interventions: A stakeholder survey Dear Dr. Toews: 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. Paavani Atluri Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0271791.r006

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