Development of the simulation-based German albuminuria screening model (S-GASM) for estimating the cost-effectiveness of albuminuria screening in Germany

Paul Kairys; Thomas Frese; Paul Voigt; Johannes Horn; Matthias Girndt; Rafael Mikolajczyk

doi:10.1371/journal.pone.0262227

Peer Review History

Original SubmissionAugust 9, 2021
9 Aug 2021 Author Response Attachments Attachment Submitted filename: response_to_reviewers.docx https://doi.org/10.1371/journal.pone.0262227.r001
3 Nov 2021 Decision Letter - Tatsuo Shimosawa, Editor PONE-D-21-25470Development of the Simulation-based German albuminuria screening model (S-GASM) for estimating the cost-effectiveness of albuminuria screening in GermanyPLOS ONE Dear Dr. Kairys, 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 Dec 18 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. 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The PLOS ONE style templates can be found at https://journals.plos.org/plosone/s/file?id=wjVg/PLOSOne_formatting_sample_main_body.pdf and https://journals.plos.org/plosone/s/file?id=ba62/PLOSOne_formatting_sample_title_authors_affiliations.pdf 2. We note that you have stated that you will provide repository information for your data at acceptance. Should your manuscript be accepted for publication, we will hold it until you provide the relevant accession numbers or DOIs necessary to access your data. If you wish to make changes to your Data Availability statement, please describe these changes in your cover letter and we will update your Data Availability statement to reflect the information you provide. 3. 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 If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references.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. Additional Editor Comments: I, as an academic editor, am so sorry not to reply you earlier because the original reviewers of your submission declined to re-evaluate your article and took a longer time to find suitable reviewers. A specialist commented minor comments. In addition to the reviewer's comments, I have some suggestions for you. This article aimed to establish S-GASM method, then it is required to validate it in different data set. There would be no data set in Germany other than you used for this assumption and authors should acknowledge this limitation. [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: I Don't Know ****** 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: This cost-effective analysis of the estimated effects of albuminuria screening seems to be rigorously scrutinized by the authors. The responses to the previous reviewers seem to be addressed appropriately. Although there are certainly several limitations to this estimation, those limitations were addressed in the literature. I have just a few more questions. 1) I am concerned about the large gap in the presence of albuminuria and assumption in the analysis, as shown in the validation analysis, in the younger population. (To begin with, could such prevalence of albuminuria be really true in the general young population?) And as the authors mentioned, although the testing costs may not be affected, several burdens such as psychological and cost will greatly affect this population. I feel it is important to be aware of any evidence on the prevalence of albuminuria in the young general population and its pathological significance. The relevance of this estimation analysis would differ according to the pathological and treatment related significance (with ACEI/ARBs) in this population. How do the authors relate to this? Would the evidence really suggest a conservative assumption for this model (eg if treating young people with ACEI/ARBs have no influence on future outcomes) ? Maybe to be more conservative, it could be suggested that this model be better use for those above 40 years old? 2) Line 163-166 the authors describe the factors integrated in the model but should not BMI be in there to? ****** 6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files. If you choose “no”, your identity will remain anonymous but your review may still be made public. Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy. Reviewer #1: Yes:** Hiroo Kawarazaki [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. 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Revision 1
18 Dec 2021 Author Response We uploaded a separate rebuttal letter, a marked-up copy of our manuscript and an unmarked version. We edited our file naming to meet the style requirements. We added a DOI for our data, including all files to simulate cohorts with different parameters, and the data we analyzed in our examplary simulations. We reviewed our reference list and have not found a retracted paper. We uploaded our figure to PACE to ensure it meets PLOS requirements. 1) I am concerned about the large gap in the presence of albuminuria and assumption in the analysis, as shown in the validation analysis, in the younger population. (To begin with, could such prevalence of albuminuria be really true in the general young population?) And as the authors mentioned, although the testing costs may not be affected, several burdens such as psychological and cost will greatly affect this population. I feel it is important to be aware of any evidence on the prevalence of albuminuria in the young general population and its pathological significance. The relevance of this estimation analysis would differ according to the pathological and treatment related significance (with ACEI/ARBs) in this population. How do the authors relate to this? Would the evidence really suggest a conservative assumption for this model (eg if treating young people with ACEI/ARBs have no influence on future outcomes) ? Maybe to be more conservative, it could be suggested that this model be better use for those above 40 years old? Our model utilizes data based on NHANES III, and if comparing the simulated with the published values, the difference is not big. The values published by Girndt et al. differ significantly from this, and we are unsure which values to rely on. Especially since a probably comparable population from the Netherlands in the PREVEND study provides data that match the values from NHANES III, but also Zhou et al., a more recent study from the US, provides even higher values than Girndt et al. 18-19 20-29 30-39 Garg [1] NHANES III 1988-1994 USA 5.7% (n=3071) 5.5% (n=2928) Gracchi [2] PREVEND 1997-1998 Groningen, NL ≈ 5.8% ≈ 5.7% (N=40000) Girndt [3] 2008-2011 Germany 12.7% (n=205) 11.9% (n=781) 8.8% (n=830) Zhou [4] 2011-2018 USA + Canada 24.7% (n =506) We are not really able to solve this problem but decided to emphasize the importance of new research in this direction, as there is a considerable influence on our topic. If we assume that these cases of “young albuminuria” have no impact on the incidence of renal replacement therapy in the later course, from a strict economic cost-effectiveness point of view, the influence from the younger age groups on the overall results is relatively small because the costs of an annual test (36,18€) are very similar to the costs of therapy for one year (54,74€) and due to the cohort-setting, 11% of all simulated person-years fall within the ages under 40. However, changing the period between the tests from one or two years increases this influence. Until we are able to engage this topic with further insights, it is possible to limit the testing to individuals older than 40 to reach maximum conservativeness. We decided not to limit the testing for our exemplary results because we assess the S-GASM to be working as intended with the data it is based on, there is no final evaluation on the topic of albuminuria in younger individuals possible, and the changes in the results are to be estimated as minor. We decided to add a section about this problem to the manuscript and also added this discussion to the supplementary material. 2) Line 163-166 the authors describe the factors integrated in the model but should not BMI be in there to? This section (Line 159-163 in the revised manuscript) was meant to describe the parameters directly influencing the prevalence, incidence, and progression of albuminuria, the initial GFR, and annual GFR loss. BMI is influencing indirectly through higher risks for hypertension and diabetes but not directly. We added the word „directly“ to make this clearer. Line 117-121 of the revised manuscript describe all factors integrated into the model. 1. Garg, A.X., et al., Albuminuria and renal insufficiency prevalence guides population screening: results from the NHANES III. Kidney Int, 2002. 61(6): p. 2165-75. 2. Gracchi, V., et al., Prevalence and distribution of (micro)albuminuria in toddlers. Nephrol Dial Transplant, 2016. 31(10): p. 1686-92. 3. Girndt, M., et al., The Prevalence of Renal Failure. Results from the German Health Interview and Examination Survey for Adults, 2008-2011 (DEGS1). Dtsch Arztebl Int, 2016. 113(6): p. 85-91. 4. Zhuo, M., et al., High Prevalence and Low Awareness of Albuminuria in the Community Setting in the KDSAP. Kidney Int Rep, 2020. 5(4): p. 475-484. Attachments Attachment Submitted filename: Response_to_Reviewers.docx https://doi.org/10.1371/journal.pone.0262227.r003
20 Dec 2021 Decision Letter - Tatsuo Shimosawa, Editor Development of the Simulation-based German albuminuria screening model (S-GASM) for estimating the cost-effectiveness of albuminuria screening in Germany PONE-D-21-25470R1 Dear Dr. Kairys, 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, Tatsuo Shimosawa, M.D., Ph.D. Academic Editor PLOS ONE Additional Editor Comments (optional): Reviewers' comments: Reviewer's Responses to Questions Comments to the Author 1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation. Reviewer #1: All comments have been addressed ******** 2. Is the manuscript technically sound, and do the data support the conclusions? The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented. Reviewer #1: Yes ****** 3. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: Yes ****** 4. Have the authors made all data underlying the findings in their manuscript fully available? The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified. Reviewer #1: Yes ****** 5. Is the manuscript presented in an intelligible fashion and written in standard English? PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here. Reviewer #1: Yes ****** 6. Review Comments to the Author Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters) Reviewer #1: All previous comments have been adequately addressed and I have no further comments to make. ****** 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:** Hiroo Kawarazaki https://doi.org/10.1371/journal.pone.0262227.r004
Formally Accepted
27 Dec 2021 Acceptance Letter - Tatsuo Shimosawa, Editor PONE-D-21-25470R1 Development of the Simulation-based German albuminuria screening model (S-GASM) for estimating the cost-effectiveness of albuminuria screening in Germany Dear Dr. Kairys: 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 Prof. Tatsuo Shimosawa Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0262227.r005

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