The impact of changing cigarette smoking habits and smoke-free legislation on orofacial cleft incidence in the United Kingdom: Evidence from two time-series studies

Matthew Fell; Craig Russell; Jibby Medina; Toby Gillgrass; Shaheel Chummun; Alistair R. M. Cobb; Jonathan Sandy; Yvonne Wren; Andrew Wills; Sarah J. Lewis

doi:10.1371/journal.pone.0259820

Peer Review History

Original SubmissionJuly 20, 2021
2 Sep 2021 Decision Letter - Sinan Kardeş, Editor PONE-D-21-23595 The impact of changing cigarette smoking habits and smoke-free legislation on orofacial cleft incidence in the United Kingdom: evidence from two time-series studies PLOS ONE Dear Dr. Fell, 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 Oct 11 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. 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 If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references.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 If you need to cite a retracted article, indicate the article’s retracted status in the References list and also include a citation and full reference for the retraction notice.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 ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: 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 ****** 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: General comments Dear authors, thank you for the opportunity to read your work. This paper aims to investigate the effect of active and passive smoking on the orofacial clefts incidence in the UK. To do this, the authors adopted the state-of-the-art of regression and time-series analysis. In this regard, I congratulate the authors for the extensive comparison with the existing literature, the transparency, completeness, and clarity of the methodology, and the detailed analysis of possible biases. Finally, the authors conclude that a significant 8% reduction occurred in England, Scotland, and Northern Ireland, while this did not occur in Scotland. At present, I believe there are only a few aspects to be clarified before proceeding with the publication. ============= Major comments 1) Section: Methods, Statistical methods. 1.1. A brief explanation of the use of the p-value should be provided. In particular, has a significance threshold been adopted? If so, specify which one. Or (recommended, https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4877414/) were p-values used as graded measures of the strength of the evidence against the null hypothesis? 1.2. Similarly, a brief explanation should be provided as to the strength of the correlations (e.g., RR). In particular, were “intensity” thresholds used or, in this case too, were the results evaluated using a continuous scale? 1.3. In some cases, in the “Results” section, the authors showed IQRs and, in others, the mean values. Was this done in consideration of the distributive nature of the data (i.e., non-Gaussians and Gaussians, respectively)? I suggest specifying this detail in the “Methods” section. 2) Section: Conclusions. For completeness and transparency, I suggest also reporting the negative result obtained for Scotland (i.e., the absence of evident correlations). ============= Minor comments m1) Section: Abstract, Lines 42-46. Please also provide values for negative results. m2) Section: Methods, Lines 184-187. It is not clear why - although a decreasing trend was already present - we should not have expected a change in slope after the smoking ban implementation. In particular, why is this not supposed to have increased the reduction “speed”? m3) Section: Figures 1, 4. I suggest specifying the quantity reported on the y-axis (e.g., Figure 1, "OFC incidence [...]"). In this way, the graphs will be clear regardless of the caption. m4) Section: Results, Line 276. Is a "mean of 0.8%" meant? ============= Other comments o1) Section: Methods, Lines 130-132. I kindly ask the authors if they can better explain what they mean by this sentence. Thank you. o2) Section: Methods, Lines 224-226. I kindly ask why the chosen lag was exactly one year (that is, why three months more than the gestation duration? Is this simply an upward approximation since the model works with annual rates?) Thank you. ****** 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:** Alessandro Rovetta [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.0259820.r001
Revision 1
25 Sep 2021 Author Response Many thanks to Reviewer 1 for their kind comments and helpful suggestions. Major comments 1) Section: Methods, Statistical methods. 1.1. A brief explanation of the use of the p-value should be provided. In particular, has a significance threshold been adopted? If so, specify which one. Or (recommended, https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4877414/) were p-values used as graded measures of the strength of the evidence against the null hypothesis? RESPONSE: Thank you for raising this point and for the helpful reference to Greenland et al., 2016. We resisted the use of the term ‘statistical significance’ and arbitrary dichotomous significance thresholds in the manuscript (in agreement with Greenland et al 2016 and Stern and Davey Smith 2001 https://www.ncbi.nlm.nih.gov/pmc/articles/PMC1119478). We have added an explanation of this in the statistical methods section (Page 9, lines 209-212) and hope this adds clarity. “Effect estimates were reported as incidence rate ratios (RR) with 95% confidence intervals (CI) and p-values. P values were interpreted as continuous measures of the strength of evidence against the null hypothesis.” 1.2. Similarly, a brief explanation should be provided as to the strength of the correlations (e.g., RR). In particular, were “intensity” thresholds used or, in this case too, were the results evaluated using a continuous scale? RESPONSE: Thank you too for this extension to the previous point. We did not use any intensity thresholds for the incidence rate ratios for the same reason of not wanting to arbitrarily dichotomise the results. Results were evaluated using a continuous scale. We hope that the added text (as per the point above) in the statistical methods (Page 9, lines 209-212) section will sufficiently address this point. 1.3. In some cases, in the “Results” section, the authors showed IQRs and, in others, the mean values. Was this done in consideration of the distributive nature of the data (i.e., non-Gaussians and Gaussians, respectively)? I suggest specifying this detail in the “Methods” section. RESPONSE: Thank you for highlighting this confusing mix of reporting. To improve consistency, we have discarded the means and have instead reported all descriptive statistics with an interquartile range. This has been specified in the statistical methods section (page 9, lines 202-203). “All variables were described initially through visualisation and summary statistics with interquartile range (IQR).” 2) Section: Conclusions. For completeness and transparency, I suggest also reporting the negative result obtained for Scotland (i.e., the absence of evident correlations). RESPONSE: We have added a comment to this effect in the conclusion (page 22, lines 510-511). “but this was not replicated in data from Scotland”. ============= Minor comments m1) Section: Abstract, Lines 42-46. Please also provide values for negative results. RESPONSE: this has been added (page 3, line 50) “(RR 1.16, 95%CI 0.94 to 1.44; P=0.173)”. m2) Section: Methods, Lines 184-187. It is not clear why - although a decreasing trend was already present - we should not have expected a change in slope after the smoking ban implementation. In particular, why is this not supposed to have increased the reduction “speed”? RESPONSE: We did not anticipate a change in slope (or ‘speed) because the smoking ban represents a singular event leading to a one-off change in passive smoking exposure. We know from data reported in this paper (i.e. active smoking prevalence and maternal age) that other factors were changing consistently during the study period, in comparison with the sudden change in passive smoking exposure following the ban. Therefore, we anticipated a change in level after the ban but not a change in slope. The anticipated model is of course, a hypothesis, but the current literature on interrupted time series specifies this impact model as an important step that should be reported. We hope that the current succinct description of this in the text is adequate. m3) Section: Figures 1, 4. I suggest specifying the quantity reported on the y-axis (e.g., Figure 1, "OFC incidence [...]"). In this way, the graphs will be clear regardless of the caption. RESPONSE: The y-axis on the graphs have been amended as per suggestion (Figure 1, 2 and 4). m4) Section: Results, Line 276. Is a "mean of 0.8%" meant? RESPONSE: Thank you. The means have now been discarded as per major comment and response above ============= Other comments o1) Section: Methods, Lines 130-132. I kindly ask the authors if they can better explain what they mean by this sentence. Thank you. RESPONSE: The sentence has been amended. Addition of ‘due to the change in annual reporting intervals’ to improve clarity (page 6, lines 131-132). “Children born in Scotland between January-April 2010 were counted twice due to the change in annual reporting intervals, but exclusion of 2010 data in the analysis did not impact the results.” o2) Section: Methods, Lines 224-226. I kindly ask why the chosen lag was exactly one year (that is, why three months more than the gestation duration? Is this simply an upward approximation since the model works with annual rates?) Thank you. RESPONSE: Yes this is correct and has been explained in the text to improve clarity (page 10, lines 230-231) “The final model was adjusted for the yearly long-term incidence trend and maternal age with a 1-year lag, as an upward approximation to account for the 9-month human gestation period since the model works with annual rates.” Attachments Attachment Submitted filename: Response to Reviewers.docx https://doi.org/10.1371/journal.pone.0259820.r002
27 Oct 2021 Decision Letter - Sinan Kardeş, Editor The impact of changing cigarette smoking habits and smoke-free legislation on orofacial cleft incidence in the United Kingdom: evidence from two time-series studies PONE-D-21-23595R1 Dear Dr. Fell, 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, Sinan Kardeş, M.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: Dear Authors, Thank you for your professional revisions. All the points I raised have been adequately addressed. Therefore, I'm glad to vote for the publication of the manuscript. I wish you the best for this and your future research. ****** 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:** Alessandro Rovetta https://doi.org/10.1371/journal.pone.0259820.r003
Formally Accepted
29 Oct 2021 Acceptance Letter - Sinan Kardeş, Editor PONE-D-21-23595R1 The impact of changing cigarette smoking habits and smoke-free legislation on orofacial cleft incidence in the United Kingdom: evidence from two time-series studies Dear Dr. Fell: 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. Sinan Kardeş Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0259820.r004

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