A prospective model of the potential clinical and economic impact of cervical cancer screening supported by a mobile phone app

Fredrick Chite Asirwa; Brian W. Bresnahan; Faith Yego; Dana Duncan; James K. Karichu; Louis P. Garrison Jr.

doi:10.1371/journal.pone.0316001

Peer Review History

Original SubmissionAugust 13, 2024
1 Oct 2024 Decision Letter - Edward Adekola Oladele, Editor PONE-D-24-32009A Prospective Model of the Potential Clinical and Economic Impact of Cervical Cancer Screening Supported by a Mobile Phone AppPLOS ONE Dear Dr. Garrison, 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 15 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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Reviewer #1: Yes Reviewer #2: Partly ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: N/A Reviewer #2: Yes ****** 3. Have the authors made all data underlying the findings in their manuscript fully available? The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified. Reviewer #1: Yes Reviewer #2: Yes ****** 4. Is the manuscript presented in an intelligible fashion and written in standard English? PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here. Reviewer #1: Yes Reviewer #2: Yes ****** 5. Review Comments to the Author Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters) Reviewer #1: The paper does a good job explaining the limitations of this type of modeling tool while also highlighting the important role played by preliminary models like this one. You also highlighted some additional data that would be useful for future studies. Reviewer #2: The authors have picked an interesting topic - the use of mobile technology in the followup of women being screened for Cervical Cancer, an important health problem. The study has been generally well conducted but I would like to address the following. 1. A clear definition of the time horizon over which costs and consequences were accrued is lacking, which is different from the study period. 2. A clear definition of the consequences being considered is not outlined. It is summarized as clinical, workflow and guideline pathways which is inadequate. 3. Also the phrase " standardized to 2022 cost" is not appropriate. This is because are cost are adjusted to a particular year to account for inflation and there are different approaches which can be used and the choice of approach should be specified. 4. Discounting is not mentioned and the rationale for not doing so should be provided. ****** 6. 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Revision 1
3 Dec 2024 Author Response Response to Reviewers Reviewer's Responses to Questions and Our Responses to Reviewers. Comments to the Author 1. Is the manuscript technically sound, and do the data support the conclusions? The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented. Reviewer #1: Yes Reviewer #2: Partly Authors' response: Thanks! We address the four specific questions raised by Reviewer #2 below. 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: N/A Reviewer #2: Yes Authors' response: Thanks! 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 Authors' response: Thanks! The data are provided in the manuscript. 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 Authors' response: Thanks! We appreciate this. We have corrected some minor errors that are visible in the marked-up version. 5. Review Comments to the Author Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters) Reviewer #1: The paper does a good job explaining the limitations of this type of modeling tool while also highlighting the important role played by preliminary models like this one. You also highlighted some additional data that would be useful for future studies. Authors' response: Thanks! We appreciate your understanding that this is a prospective clinico-economic evaluation using modeling. Reviewer #2: The authors have picked an interesting topic - the use of mobile technology in the follow up of women being screened for Cervical Cancer, an important health problem. The study has been generally well conducted but I would like to address the following. Authors' response: Thanks! We appreciate these questions: 1. A clear definition of the time horizon over which costs and consequences were accrued is lacking, which is different from the study period. Authors' response: Thanks! The analytic time horizon is the period (less than one year) for a typical patient to complete the three visits. It is shorter than the study period. From a practical perspective, our modeling approach assessed “events” occurring or not occurring (such as follow-up visits), rather than specific number of days or weeks/months. Timing of second or third visits, for example, varied. This is a realistic scenario, particularly in a low-to-middle-income country setting. We have added this language on page 5, line 98 to make it clear: "(usually completed in less that 6 months)" 2. A clear definition of the consequences being considered is not outlined. It is summarized as clinical, workflow and guideline pathways which is inadequate. Authors' response: Thanks for this comment. We have revised the text on page 6 line 104-5 to address this, adding: " "Key consequences include intermediate endpoints (such as test results) and final endpoints (viz., pre-cancerous and cancer cases detected and treated)." And on page 7, line 128-9, adding: " such as cost-per-case detected and cost-per-cancer-case avoided)'. 3. Also the phrase " standardized to 2022 cost" is not appropriate. This is because are cost are adjusted to a particular year to account for inflation and there are different approaches which can be used and the choice of approach should be specified. Authors' response: Thanks for this comment. We agree that the word "standardized' is unclear. We have changed it to "estimated costs in 2022," which more accurately reflects our approach. Medical cost finding for economic evaluation from a (limited) societal perspective is difficult in both developed and developing counties if the aim is to use opportunity costs for clinical and economic policy making. Prices in medical "markets" are affected by government price setting and insurance--both public or private. For this prospective modelling exercise, we needed to have plausible estimates or assumptions for the model parameters. By necessity, we used cost information from multiple sources to generate plausible base-case assumptions and ranges. Thus, our cost estimates were "triangulated" from multiple input sources, including: prospective data collection at sites, literature-based estimates, and expert opinion at the primary clinical site of the principal investigator in Kenya. The prospective cost-related data was collected in 2022 in multiple Kenyan sites, including with hospital and clinic administrators (as IRB approved). Thus, we report our costs as 2022 U.S. dollars, converted from Kenyan shillings. We also used a time-and-motion estimation approach in 2022 to collect supporting data related to resources used during visits, time, and costs. We did not apply a specific formulaic inflation adjustment to the literature-based costs estimates for visits, tests, or therapies, which were published in various years. We applied "face-validity testing" with experts (clinical and economic) to our 2022 base-case cost estimates and to our ranges of costs. We assessed how our estimates compared with recent published ranges for variables similar to our model inputs for amounts and ranges of cost. A challenge is that literature-based costs varied in the inputs included in various studies, depending on the research question being asked and addressed, and other regional or national differences. Also, unlike the availability in the United States for having annual medical care price change percentages, there is not a similar precise "medical care inflation-adjustor." In Kenya, general inflation data is available through the Kenya National Bureau of Statistics (KNBS), and healthcare-related components might be included in the broader Consumer Price Index (CPI). However, these components typically lack the granularity or specificity of a dedicated "medical care inflation" index, making it more challenging to precisely adjust healthcare costs annually. 4. Discounting is not mentioned and the rationale for not doing so should be provided. Authors' response: Thanks for spotting this omission. We have added this sentence on page 9, lines 180-1, to make address this: "Since the time horizon for the primary analysis is short-term (i.e., less than one year to complete the 3 visits), there is no strong need to perform discounting of costs or outcomes." Inquiry under Journal Requirements 3. In the ethics statement in the Methods, you have specified that verbal consent was obtained. Please provide additional details regarding how this consent was documented and witnessed, and state whether this was approved by the IRB. Authors' Response: We have added on page 9, lines 174-7: "For the verbal consent process, the study was explained to the participants who were selected based on their expertise in the study topic, and once they agreed to participate, the consent was documented and the interview conducted. All informed consenting processes were approved by the IRB." Attachments Attachment Submitted filename: Response to Reviewers.docx https://doi.org/10.1371/journal.pone.0316001.r002
5 Dec 2024 Decision Letter - Edward Adekola Oladele, Editor A Prospective Model of the Potential Clinical and Economic Impact of Cervical Cancer Screening Supported by a Mobile Phone App PONE-D-24-32009R1 Dear Dr. Garrison, 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, Edward Adekola Oladele, MD, MPH, PhD Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0316001.r003
Formally Accepted
Acceptance Letter - Edward Adekola Oladele, Editor PONE-D-24-32009R1 PLOS ONE Dear Dr. Garrison Jr., 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. Edward Adekola Oladele Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0316001.r004

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