Cost-effectiveness of HPV vaccination in 195 countries: A meta-regression analysis

Katherine L. Rosettie; Jonah N. Joffe; Gianna W. Sparks; Aleksandr Aravkin; Shirley Chen; Kelly Compton; Samuel B. Ewald; Edwin B. Mathew; Danielle Michael; Paola Pedroza Velandia; Molly B. Miller-Petrie; Lauryn Stafford; Peng Zheng; Marcia R. Weaver; Christopher J. L. Murray

doi:10.1371/journal.pone.0260808

Peer Review History

Original SubmissionMarch 2, 2021
10 May 2021 Decision Letter - Carlos Alberto Zúniga-González, Editor Transfer Alert This paper was transferred from another journal. As a result, its full editorial history (including decision letters, peer reviews and author responses) may not be present. PONE-D-20-32128 Cost-effectiveness of HPV vaccination in 195 countries: A meta-regression analysis PLOS ONE Dear Dr. Christopher JL Murray, 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. 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Integral assessment (financial, economic, social, environmental and productivity) of using bagasse and fossil fuels in power generation in Nicaragua. Revista Tecnología en Marcha, 28(4), 94-107. c) Zuniga González, C. (2020). Total factor productivity growth in agriculture: Malmquist index analysis of 14 countries, 1979-2008. Revista Electrónica De Investigación En Ciencias Económicas, 8(16), 68-97. https://doi.org/10.5377/reice.v8i16.10661 Journal Requirements: When submitting your revision, we need you to address these additional requirements. 1. Please ensure that your manuscript meets PLOS ONE's style requirements, including those for file naming. The PLOS ONE style templates can be found at https://journals.plos.org/plosone/s/file?id=wjVg/PLOSOne_formatting_sample_main_body.pdf and https://journals.plos.org/plosone/s/file?id=ba62/PLOSOne_formatting_sample_title_authors_affiliations.pdf 2. Please review your reference list to ensure that it is complete and correct. 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However, the manuscript needs revisions. 1.Different formats have been adopted to quote references, the style should be according to the PLOS requirements and uniform. 2.The policy implications need attention. There is an ample room to suggest more policy implications in the conclusion section. ****** 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:** DURSUN BALKAN Reviewer #2: No [NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. 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Revision 1
1 Nov 2021 Author Response 3.1. The author must add a sub section in the methods parts which is about Meta Regression analysis. Otherwise, the metholodogical part of your paper become weak. Response: In the revised subsection on Modelling approaches, we expanded the description of the five stages of the mixed-effects meta-regression framework from one paragraph to six, with a paragraph devoted to each of the five stages, as reproduced below: “The statistical model and fitting procedures for the analysis of ICERs was conducted in five stages, and used a mixed-effects meta-regression framework (MR-BRT).20 This model included priors on all covariates and a study-specific random intercept. Each stage is described briefly below; for further information, see S3 Appendix. “In the first stage, we estimated priors for selected covariates by leveraging the fact that one-way sensitivity analyses differ in no unmeasured covariates from their reference analyses. Four covariates had a sufficient number of sensitivity analyses reported published CEA to estimate priors using crosswalk models: vaccine cost, vaccine coverage, cost discount rate, and discount rate for health outcomes. We matched each sensitivity analysis with its corresponding reference analysis, and the crosswalk model estimated the difference in log-ICERs between sensitivity and reference analyses as a function of the difference between values of that covariate. We then constructed Gaussian priors for these covariates to use in all subsequent stages of the analysis with means and standard deviations equal to the crosswalk parameter estimates and standard errors from these crosswalk models. “In the second stage, we estimated a nonlinear response curve for log-GDP per capita by modeling the log-ICERs as a nonlinear function of log-GDP per capita. Log cervical cancer DALYs per capita was entered linearly into this model, in addition to the four covariates addressed in the first stage, and the priors calculated in the first stage were placed on the corresponding covariates. To make this stage more robust to model misspecification, we placed a spline ensemble on log GDP per capita. This model also used a robust statistical approach for outlier detection, and outliers trimmed at this stage were discarded from subsequent steps of the analysis. The nonlinear response curve estimated by this model was used to transform log-GDP per capita for use in subsequent stages of the analysis. “In the third stage, we selected additional covariates to include in the final meta-regression using a generalized Lasso approach for linear mixed effects models. The four crosswalk covariates, log cervical cancer DALYs per capita, and spline-transformed log-GDP per capita were pre-selected covariates at this stage, and the priors estimated for the crosswalk covariates were placed on those covariates. This process selected from nine additional candidate covariates: target sex, the proportion of model population assumed to have access to cervical cancer treatment, vaccine type, perspective, time horizon, comparator, and the outcome measure, and whether or not the intervention included a booster dose. Only one of these covariates, the assumption of a booster dose, was not selected for inclusion in the final model. “In the fourth stage we selected the standard deviation of a Gaussian prior to apply to all regression parameters other than the intercept and the parameters for the four crosswalk covariates. To select a standard deviation, we fit a mixed effects meta-regression models with random intercepts by study, and priors on crosswalk covariates as calculated in the first stage. We normalized all other covariates and included Gaussian priors on those covariates, centered at zero and with a standard deviation that was constant across covariates. We varied this standard deviation using a grid-search and used 10-fold cross-validation to select the standard deviation that minimized the mean squared error for predicting values in the holdout set. We then converted the prior standard deviation back to the unstandardized scale for each covariate. “In the fifth stage, we fit a mixed effects model with a random intercept and priors on covariates determined in the first and fourth stages. This model included priors on covariates calculated in the first and fourth stages and the transformed version of log-GDP per capita, and random intercepts by study.” 3.2 Also, the conclusion part must be improved Response: The conclusion has been revised as follows: This is the first attempt to generate a complete and consistent set of ICERs for HPV vaccines with UI for 195 countries. Meta-regression analysis can be conducted on CEA, where the one-way sensitivity analyses are used to quantify the effects of factors at the intervention and method-level. There is substantial uncertainty in the predicted ICERs in some countries, due to underlying heterogeneity of published CEA. Our results however, identified countries where the HPV vaccine is a good value, despite the uncertainty, and can facilitate decision-making across a wide range of settings. Globally, introducing the HPV vaccine and achieving high HPV vaccine coverage are critical steps to eliminating cervical cancer burden. Building on all available information, our results support introducing and expanding HPV vaccination, especially in many countries that are eligible for subsidized vaccines from Gavi, the Vaccine Alliance, and the Pan American Health Organization. Vaccine cost is a key covariate, and our estimated models can be readily predictions ICERs and UI whenever vaccine subsidies are extended to additional countries or the vaccine price changes. Reviewer #2: The manuscript seems interesting . The authors have conducted a thorough literature review, and analysed information accurately and sufficiently. However, the manuscript needs revisions. 4.1. Different formats have been adopted to quote references, the style should be according to the PLOS requirements and uniform. Response: Thank you for this helpful feedback. The format for citing references complies with the PLOS One requirements in the revised manuscript. 4. 2. The policy implications need attention. There is an ample room to suggest more policy implications in the conclusion section. Response: Please see the response to comment 3.2 above. Attachments Attachment Submitted filename: Response to reviewers.docx https://doi.org/10.1371/journal.pone.0260808.r002
18 Nov 2021 Decision Letter - Carlos Alberto Zúniga-González, Editor Cost-effectiveness of HPV vaccination in 195 countries: A meta-regression analysis PONE-D-20-32128R1 Dear Dr. Christopher JL Murray, 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, Carlos Alberto Zúniga-González, Ph.D Academic Editor PLOS ONE Additional Editor Comments (optional): Congratulations dear authors for the effort to improve the quality of your manuscript. 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 #2: 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 #2: Yes ****** 3. Has the statistical analysis been performed appropriately and rigorously? Reviewer #2: 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 #2: 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 #2: 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 #2: The topic of the study appears to be interesting. The review comments have been addressed adequately. I wish the author(s) all the best. ****** 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 #2: No https://doi.org/10.1371/journal.pone.0260808.r003
Formally Accepted
10 Dec 2021 Acceptance Letter - Carlos Alberto Zúniga-González, Editor PONE-D-20-32128R1 Cost-effectiveness of HPV vaccination in 195 countries: A meta-regression analysis Dear Dr. Murray: 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. Prof. Carlos Alberto Zúniga-González Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0260808.r004

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