Alarm-assisted urotherapy for daytime urinary incontinence in children: A meta-analysis

Liesbeth L. de Wall; Antje J. Nieuwhof-Leppink; Renske Schappin

doi:10.1371/journal.pone.0275958

Peer Review History

Original SubmissionNovember 12, 2021
12 Jul 2022 Decision Letter - Emily Chenette, Editor PONE-D-21-35570Alarm-assisted urotherapy for daytime urinary incontinence in children. A meta-analysis.PLOS ONE Dear Dr. Wall, Thank you for submitting your manuscript to PLOS ONE; I sincerely apologise for the unusually delayed review timeframe. Your manuscript has been assessed by two reviewers, whose comments are appended below. 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. In particular, please ensure that the statistical analysis and data presentation is revised per the recommendations of reviewer 2. Please submit your revised manuscript by Aug 25 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. 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Note that it is not acceptable for the authors to be the sole named individuals responsible for ensuring data access. We will update your Data Availability statement to reflect the information you provide in your cover letter. 4. 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 If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references.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 If you need to cite a retracted article, indicate the article’s retracted status in the References list and also include a citation and full reference for the retraction notice.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. 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: Yes ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: Yes 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: 1. Please make sure that "data" are used as plural throughout 2. On line 113, “trials” is misspelled twice 3. Please comment further based on your thorough review on what is needed from the literature on the topic in future studies Reviewer #2: The study aims to systematically evaluate, summarize, review, and analyze existing evidence about the effect of wearable alarm systems in urotherapy for children with functional DUI (daytime urinary incontinence). This is an interesting study, well-analyzed and presented. However, the manuscript can be further improved based on the following comments. Data synthesis and statistical analysis Line 179-185, the use of fixed effect and random effect is not clear and the sentence requires revision. The software that was used in performing the analysis is to be stated. Results Table 1, the title is too short. In the treatment duration days column, some indicated protocol and few without protocol. For Intervention and control condition column, for those that indicated a single group apart from those studies which had intervention and control, more information is to be provided. Dash(-) to be denoted in the table footnote. The subtitles in Table 1 could be reworded. For example, state authors (year), country, study design, mean age (sd, range). For Median 114 [CI 64-146], the level of CI is to be stated. For the nonadherence 10%, how many were from intervention and control to be stated. More information could be provided in the table to provide more information to readers. Table 2, CI 95 to be rewritten as 95%CI. Grade to GRADE. Study outcomes Line 256, the use of OR for RCTs is inappropriate, Line 266, what are outliners? Secondary outcomes Line 279, the representation of variance component to be clearly stated. Treatment duration Line 299, the R^2 value to be stated. At least one decimal point for the percentage figure to be provided. P or italicize P to be standardized throughout. Figure 1, the exclusion of n=19 to be separated and denoted with an asterisk for the individual items that sum up to n=19 Figure 3B, event rate to be denoted in the table footnote. Figure 3A, 3B, Figure 5, the decimal points for the data were converted to commas in the PDF format. The citation of references in the text and list of references to follow journal format. The manuscript requires some English editing. ****** 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 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. 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.0275958.r001
Revision 1
8 Sep 2022 Author Response Reviewer 1 1. Please make sure that “data” are used as plural throughout. Thank you for the remark, we adjusted it throughout the manuscript. 2. On line 113, “trials” is misspelled twice. Thank you for the comment. A native speaker edited the whole manuscript and the manuscript was adjusted accordingly. This included the misspelled “trials”. 3. Please comment further based on your thorough review on what is needed from the literature on the topic in future studies. To clarify future needs on this topic the paragraph strength and limitations was extended, see also line 460-463; “Future research should focus on high methodological quality research including a control group and sham device with use of clear defined inclusion criteria (therapy naïve versus therapy refractory children, exact duration of treatment/use of device, definition outcome according to ICCS criteria and definitions for adherence).” Reviewer 2 The study aims to systematically evaluate, summarize, review and analyze existing evidence about the effect of wearable alarm systems in urotherapy for children with functional DUI (daytime urinary incontinence). This is an interesting study, well-analyzed and presented. However, the manuscript can be further improved based on the following comments: 1. Data synthesis and statistical analysis Line 179-185, the use of fixed and random effects Is not clear and the sentence requires revision. Thank you for the remark. In a fixed-effect model it is assumed that all studies in the meta-analysis share a common (true) effect size. Or in others words, all factors that could influence the effect size is the same for all the individual studies. This is often implausible as the studies are not completely identical in all aspects. Therefore a random effect model is preferable. Initially with only two RCT’s the estimate of the between studies variance will have poor precision and cannot be applied appropriately. A fixed effect model seemed the second best option to at least get an idea of the summarize effect size of both RTC’s. However, due to the long time between initial submission and review (November 2011-July 2022) the literature was not up to date anymore. To overcome this, we decided to repeat our complete search. This led to an extra published RCT in the meantime and gave us the opportunity to do a preferred random-effect model including three instead of two RCT’s. We additionally adjusted the section: “Data synthesis and statistical analysis” accordingly by mentioning the random effect model to estimate overall effect size of alarm intervention added to urotherapy. 2. The software that was used in performing the analysis is to be stated. Thank you for the comment. In the revised manuscript the software used for data analysis is mentioned in line 190 (including its reference).We tried to rephrase the sentence as we understand CMA might not be a familiar software program. “ The software used to analyze the data was Comprehensive Meta-Analysis V3 . [30]” 3. Table 1, the title is too short We adjusted the title to: “Study characteristics of all studies included in this systematic review.” 4. In the “treatment duration, days” column, some indicated protocol and few without protocol. Dash(-) to be noted in the footline Thank you for the comment. We understand this might be confusing. If a protocol was used in the included study, the actual days of alarm use was mentioned. In some studies no specific protocol was used and the alarm was worn for a different period of time in each individual child. In that case either the median of mean (upon available in the article) was mentioned. A footnote is added to clarify this. The meaning of (-) is also included in the footnote. 5. For the “Intervention and control condition column”, for those that indicated a single group apart from the studies which had intervention and control more information is to be provided. Thank you for the comment. We adjusted it in the Table and renamed it into “ Type of alarm” for better clarification. This was further subclassified in case of more than one group was present. 6. The subtitles in Table 1 could be reworded. For example, state authors (year), country, study design, mean age (sd, range). For Median 114 [CI 64-146], the level of CI is to be stated. Thank you for the comment. We reworded the content- including subtitles and footnote- to clarify its content. The level of CI is stated. 7. For the nonadherence 10%, how many were from intervention and control to be stated. Unfortunately the authors of this specific study did not further subclassified the 10% non-adherence into those who had used the contingent or non-contingent alarm. Due to its publication date in 1987, this information could not be retrieved in another way. This is added to the table/ footnote. The table provides all available information retrieved from the included study. However, due to the low methodologic quality of some of the studies some information is simply not available. 8. Table 2, CI 95 to be rewritten as 95%CI. Grade to GRADE. Thank you for the comment. This was adjusted throughout the manuscript. 9. Line 256, the use of OR for RCTs is inappropriate. Thank you for the comment. We adjusted it to risk ratio (RR). 10. Line 266, what are outliners? Thank you for the comment, this should be outliers. Outliers are studies with potential impact on the overall summary effect like the study of Boelens et al. The continence rate after treatment in this study is relatively low compared to the other studies included in this Meta-Analysis. By performing a sensitivity analyses excluding this study in particular we could determine how robust our findings were and how much the summary effect would change. We rephrased (line 279-281) the sentence into : “Subsequent sensitivity analyses, which removed the study by Boelens et al. because the relatively low continence rate in this study might potentially influence summary effect, revealed a summary effect comparable to the main analyses, namely a 48% continence rate (95% CI: 35–64%).” 11. Line 279, the representation of variance component to be clearly stated. Thank you for the comment. We adjusted this in the sections: “ Data analysis and statistical analysis.” Line 198: “To investigate if relapse, treatment duration, or adherence were relevant predictors that could account for the variance component Tau2 in the overall event rate, a meta-regression analysis was performed assuming a random effect model, using a Z distribution.” It was further adjusted in the section “Type of Alarm”, line 289: “A subgroup analysis revealed 49% continence (95% CI:32-67%) after treatment if a pants alarm was used versus 45% continence (95% CI: 23-68%) when a timer watch was used, Qbetween=0.10, df =1, P=0.751, Tau2 =0.63.” 13. Line 299, the R^2 value to be stated. Thank you for the comment. We adjusted this in the sections: “ Data analysis and statistical analysis.” Line 199: “The regression coefficient R2 was used as a measure of the proportion of the observed variance that was not due to sampling error.” It was further adjusted in the section “Treatment duration”, line 311: The overall proportion of variance explained by treatment duration was 0% (R2 = 0.00, 95% CI: -0.01-0.02, Qmodel =0.07, df =1, P=0.790). 14. At least one decimal point for the percentage figure to be provided. Thank you for the comment. We would like to ask the reviewer for clarification which specific Figure needs to be adjusted as to our knowledge there is no figure with percentages. 15. P or italicize P to be standardized throughout. Thank you for the comment. This was adjusted throughout the manuscript. 16. Figure 1, the exclusion of n=19 to be separated and denoted with an asterisk for the individual items that sum up to n=19. We adjusted the figure to clarify the individual items of the total of 19. Figure 3B, event rate to be denoted in the table footnote. The footnote/ title was adjusted. Figure 3A, 3B, Figure 5, the decimal points for the data were converted to commas in the PDF format. Thank you for the comment, the figures are adjusted. 17. The citation of references in the text and list of references to follow journal format. Thank you for the comment. The reference list and citations are adjusted to the journal format. In addition we updated the reference list with relevant current literature. Two references were retracted: Hellstrom AL, Hanson E, Hansson S, Hjalmas K, Jodal U. Micturition habits and incontinence in 7-year-old Swedish school entrants. Eur J Pediatr. 1990;149(6):434-7. Swithinbank LV, Heron J, von Gontard A, Abrams P. The natural history of daytime urinary incontinence in children: a large British cohort. Acta Paediatr. 2010;99(7):1031-6. And replaced by: Linde JM, Nijman RJM, Trzpis M, Broens PMA. Prevalence of urinary incontinence and other lower urinary tract symptoms in children in the Netherlands. J Pediatr Urol. 2019;15(2):164 e1- e7. Epub 20181108. doi: 10.1016/j.jpurol.2018.10.027. PubMed PMID: 30583907. Xing D, Wang YH, Wen YB, Li Q, Feng JJ, Wu JW, et al. Prevalence and risk factors of overactive bladder in Chinese children: A population-based study. Neurourol Urodyn. 2020;39(2):688-94. Epub 20 In addition a new study was added to the systematic review: Caldwell PHY, Kerr M, Hamilton S, Teixeira-Pinto A, Craig JC. An Alarm Watch for Daytime Urinary Incontinence: A Randomized Controlled Trial. Pediatrics. 2022;149(1). doi: 10.1542/peds.2021-053863. PubMed PMID: 34907443. 16. The manuscript requires some English editing. Thank you for the comment. The revised manuscript has been reviewed by a native speaker and adjusted accordingly. Attachments Attachment Submitted filename: rebuttalplosone.docx https://doi.org/10.1371/journal.pone.0275958.r002
27 Sep 2022 Decision Letter - Muhammad Shahzad Aslam, Editor Alarm-assisted urotherapy for daytime urinary incontinence in children. A Meta-Analysis. PONE-D-21-35570R1 Dear, 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, Muhammad Shahzad Aslam, Ph.D.,M.Phil., Pharm-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 #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 percentage figures in Table 1 are without decimal points. Please check if the actual figures are with or without decimal points. ****** 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.0275958.r003
Formally Accepted
3 Oct 2022 Acceptance Letter - Muhammad Shahzad Aslam, Editor PONE-D-21-35570R1 Alarm-assisted urotherapy for daytime urinary incontinence in children: A meta-analysis. Dear Dr. Wall: 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. Muhammad Shahzad Aslam Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0275958.r004

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