Peer Review History
| Original SubmissionMay 25, 2023 |
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PONE-D-23-15590The impact of estimation methods for alcohol-attributable mortality on long-term trends for the general population and by educational level in Finland and Italy (Turin)PLOS ONE Dear Dr. Van Hemelrijck, 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. ============================== The paper is of great value, but recommendations for improvements have been made by reviewers. After consideration of their comments, we find it necessary to request major revisions to ensure the manuscript meets the required standards for publication. ============================== Please submit your revised manuscript by Sep 16 2023 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. Please include the following items when submitting your revised manuscript:
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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: Partly ********** 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: No ********** 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: This paper sets out to compare levels and trends over time in alcohol-attributable mortality (AAM) in Finland and Italy using 3 alternative approaches. The authors demonstrate that the choice of approach has a substantial impact on both levels and trends in AAM and propose a new approach which they argue is an improvement on the widely-used Underlying Cause Of Death (UCOD) approach. Overall this is an interesting and worthwhile thing to do, and the paper is generally clear and well-written, however I have some significant concerns about the conceptual basis of the paper. Fundamentally the three approaches examined in this paper: UCOD, MCOD and PAF-based are estimating different underlying concepts. UCOD is the minimum set of deaths which we can be all but certain are caused by alcohol as the underlying cause is one which could only be caused by alcohol. MCOD captures all UCOD deaths, plus some deaths which will be certainly attributable to alcohol, but which were not coded as such due to stigma, or clinical error, plus some further deaths where causal attribution to alcohol is more debatable. Finally, the PAF-based approach uses epidemiological evidence combined with data on alcohol consumption to estimate the overall number of deaths from all causes, including those where alcohol is but one of many risk factors. Thus UCOD is a subset of deaths from wholly alcohol-attributable conditions, MCOD is closer to all deaths from wholly alcohol-attributable conditions, but also some deaths that are not directly caused by alcohol and PAF-based is an estimate of all deaths from both wholly and partially alcohol-attributable conditions. The relationship between these three measures will depend on various factors: • The quality of mortality records, including the prevalence of autopsies • The level of stigma associated with AAM and the extent to which this might influence clinical coding • Guidelines and standard practice for recording contributory causes on a death certificate • Patterns of alcohol consumption (as wholly alcohol-attributable deaths are very strongly associated with heavy drinking, whereas deaths from partially alcohol-attributable causes can occur, albeit at low rates, in people drinking at lower levels – so a population of entirely moderate drinkers will have a different UCOD:PAF ratio than a population with a large number of dependent drinkers) • The PAFs used, the assumptions inherent in their calculation and whether they vary over time or between population subgroups The authors touch on some of these issues to some extent, but the conceptual differences between the three measures aren’t well set out in the paper and so it is hard in places to follow the theoretical logic behind the proposed new method. If this logic is not well set out then there is a risk that readers are left with the impression that there is no solid theoretical basis for the new approach, it is simply a mathematical combination of available data that gives a plausible result. Further, the necessary assumptions that must hold in order for the approach, which is based only on Finnish data, to be meaningful for other countries, are not well elaborated. One other, more minor, point is that some aspects of the analysis of inequalities are underdeveloped. It is almost impossible to discern the trends in inequalities from the graphs and the data is not presented elsewhere. The discussion brings in mention of SII and RII which are not elsewhere defined or presented. If this is an important component of the paper then please provide more detail in the main results section of the text. Finally, I would suggest that the graphs could be made clearer and more appealing with the use of a little colour. Reviewer #2: Dear Authors, Estimation methods in alcohol-attributable mortality are important to the field. I think it’s a valuable contribution to explore other methodologies above the standard PAF (and, sometimes UCOD and MCOD) methods in Finland and Turin. I don’t think enough has been done to “choose” the MCOD analysis as the gold standard for which to shoot for in the development of your enhanced method. Both the UCOD and MCOD methods entirely ignore (weight to 0.00) all partially-attributable health conditions, like cancer, IHD, stroke and injury, which together make up the majority of AA deaths. Any similarity the MCOD rates have to the PAF rates seem to simply be a coincidence and not anything that would suggest that the MCOD method is the most desirable. This is my view, in any regard. Towards the article, I think the discussion about which method is best needs to be built out and expanded. It may be OK to suggest the UCOD Enhanced method as another/competing method, but I’m not sure you go as far as saying it is the “preferred” method, given the enormous limitation of ignoring completely most of the health conditions that are causally related to alcohol. There are other theoretical limitations (described in more detail below) with the MCOD. In effect, it gives a selective and differential weighting of 1.00 to the alcohol-specific diagnosis regardless of where it appears on the death record. It is different for each record and this is not particularly rigorous or logical, in terms of a weighting strategy. Abstract Results 1. I don’t believe the article has done enough to theoretically describe why the MCOD method 2 would provide the most “realistic” rates towards which to shoot. Conclusions 2. I don’t think this has been developed enough, throughout the article, to state that the new method is preferable. As in my last three comments below, using either UCOD, MCOD, or UCOD Enhanced entirely ignores all the partially-attributable health conditions that together make up the majority of AA death. It doesn’t seem reasonable to put forward any of these methods as the gold standard towards which we should shoot. (Although I recognize the adjustment factor used for the Enhanced method is based on the PAF method). Introduction 3. First statement. Prefer just “Alcohol consumption” instead of “excessive.” All use *may* carry some risk and it isn’t defined what you mean by “excessive” at this stage. 4. First paragraph. I would request more information about the “different estimation methods” at this stage, since it is motivating the study. 5. Second paragraph. The first sentence here, where two methods are described – there’s not enough information provided to differentiate between the two methods. This paragraph may benefit from first building out the concepts of wholly-attributable and partially-attributable health conditions, and how they relate to your coming analyses. 6. Line 49 – using CCOD variables will definition result in “higher” estimates. But I am not certain they would be “more realistic”. Since the CCOD (what you later call MCOD) analysis weights all diagnosis codes as 1.00 and includes all records as being completely caused by alcohol, it’s hard to know if this is more reliable, or not. 7. End of paragraph two: it would be better if reference 11 applied to more than just the single (small in magnitude) condition of alcoholic cardiomyopathy. But #4 is a great reference for this. Also, here it may be good to state that sometimes the data needed to estimate your own country-specific AAFs (which isn’t particularly difficult if the data is present) is not always available in the country/region in question. Methods 8. It is a bit odd to have this statement about the Supplementary data and methods at the beginning of the methods section. I would put it where you feel you need to provide more information. 9. Not sure Finland and Italy qualify, in global context, as “vastly different drinking cultures.” Taken in context, they are really fairly similar. Possibly within Europe, they could be considered somewhat different. But Europe is not near representative of global alcohol use. So, some context is needed for this statement. 10. Suggest reorganinzing the subheadings into the following order: - Data sources, Study population, Approach and existing estimation methods, Novel estimation method. - It would be better, I believe, to merge the Approach and existing methods description into one. 11. For the smoothing approach. It isn’t clear to me if this is just to re-apportion the 1% of deaths with an unspecific cause or if it’s more general? 12. Can you better describe what the educational levels relate to? 13. Important. For the SES education information, it isn’t clear (in Finland) if this is individually collected (for the census) and then linked to death data. Or if it is area-based (from the census) and attached to individuals via postcode/other method. This is quite important for the study design. 14. Last paragraph of “Data”: these aren’t age groups. It would be helpful to write (30-34, 35—39, and so on) 15. I’m not certain about backwards linear extrapolation of the AAFs. Since the alcohol supply (alcohol per capita) is not included in the estimation, this seems like a bit of a dart throw. Is there a reference that has used this technique before. 1972-1989 (18 years) is a long period to extrapolate! 16. For the UCOD and MCOD methods, it would help to define a term that relates to all the wholly-attributable health condition codes. For examples, sometimes these are called “alcohol-specific” health conditions. 17. In the UCOD paragraph, I really don’t know what this statement could mean “However, we excluded causes of death that were not specific to the age group we studied.” What could you mean here – maybe infant codes, I am not sure. 18. MCOD approach. Should write (100%) alcohol-attributable – i.e. add the 100% or wholly word. 19. Does Finland only have three CCOD variables? If not, why do you only study the first three CCOD variables and not the entire record? 20. For this MCOD approach, it would help to have a statement similar to this “regardless of the position of the alcohol-specific (or whatever term you choose) cause of death ICD10 code, this entire death is categorized as alcohol-attributable. This is analogous to weighting each COD position as 1.00, regardless of where the alcohol-specific code appears. 21. PAF method. Importantly, it isn’t the death that is partially or wholly-attributable, it’s the entire health condition (disease). For example, a breast cancer death is either caused by alcohol (or not), there is no way to know of course it was or not. But we cannot say “8% of this breast cancer death was caused by alcohol.” It is either entirely caused by alcohol or not. We can say that, of 1000 breast cancer deaths, we estimate that about 80 of them occurred because of alcohol use. 22. An example of this difference is in the first sentece of PAF method. We should write “….combine deaths accruing to health conditions which are wholly- and partially-attributable to alcohol use”, instead of what is written about a single death. 23. Also for the PAF method, it isn’t clear for the partially-attributable conditions if these are enumerated only if the code is the UCOD, or if it is also enumerated if the code is anywhere on the abstract (i.e. including MCOD). 24. For negative AAFs – what did you replace them with? Was it simply that you set to 0.00? I would say this is debatable, but it might be OK to truncate them. Results 25. Figure 1 – typically the y-axes would have the same range in a figure like this. The way you have it may seem to overstate the ASMR in females (since range is 200, instead of 800). Probably, this was to allow to see better the three methods. You could try and come up with a way to let readers know you’ve chosen a different range for females, as compared to males. 26. Figure 2a and 2b. I would not use a line graph for these age group comparisons, I would reserve the line graphs for temporal analyses. It’s quite confusing this way. I would probably makes these two figures into bar graphs for each of the age groups. Also, it cannot be said which is Finland and which is Turin from looking at the figure. 27. Paragraph one – statement “to levels that seem unrealistic.” I would save this interpretation for the discussion. 28. New method. I believe this should be in the “Methods” section, although I understand you wanted to put it after the motivating figures. 29. New method. There really needs to be a formula here, and more information about what the method is doing. For example, it would help to have a table of the “average of these yearly ratios by country and sex over the 1990-2017 period.” This table should be in the article proper (and you don’t even have it in the supplemental), since it’s so critical to the new method. And it would help us as readers understand the ratio-ing work that you’ve done. 30. I think I’m correct that the “correction factor” is (generally) PAF/UCOD for each country/sex, but I am not totally certain that is what was done. 31. In “Development of a novel….” section. Again, I would save the “unrealistic” statement about the PAF method for the discussion and interpretation. Right now, they are simply the findings. Discussion Summary of results 32. I’m not certain what “trends in AAM are the same for the two COD-based methods in Finland” is referring to? What do you mean by the trends are the same? There is no metric of this (I don’t think), so do you mean a visual analysis seems to indicate that the MCOD method 2 is similar, but proportionally higher than the UCOD method 1. Or similar? 33. What do you mean by the trends being “less favourable.” I am not sure I follow that section. 34. In this paragraph, it would help to have a clearer description of the new “UCOD enhanced” method. Here, why are the findings from your PAF method “unrealistic” – here’s where a few references are needed as to why those findings are too high for your liking. 35. Last sentence of this subsection – this should be moved to the “appraisal of the new method” section, as you’re getting a bit ahead of the conclusions. Interpretation of the findings 36. Here again suggest modering the discussion of the differences between Finland and Italy in terms of drinking cultures, since they are quite similar in global perspective. For example, what are the APC levels in each, and at what global percentile is each country? They are probably within 10% (or less) or each other, though I may be wrong. 37. “…our finding that AAM trends are generally more favourable ….” I really don’t know what this could mean. 38. The PAF method is still a (type of) COD method – it just layers another method on top of the COD method, which is still the basis. I think this is lost through the article and would confuse a reader not familiar with all three methods used. E.g. line 293. 39. Line 296, again here I would prefer a broader reference than Manthey et al (this is a good article but only about cardiomyopathy) 40. Around line 300, with the discussion of the PAF / CVD / declining in Europe. It would useful to add a discussion of the fact that, if what you write is true, then one main the reason for the PAF method failing is because the RR functions used are (1) international (and not country-specific, or Europe-specific), and (2) either not age-group specific, or only a few age groups. That would be a suggestion for future work that would help to “fix” the PAF method. 41. At line 307, I’m quite surprised to now find an entirely new analysis (RII and SII) being discussed, which was not presented in the results section, or any of the figures (or tables, if some are added)! Surely, this should be in the results, with accompanying methods in the methods section. 42. In general, there may be too many results to present them all and it could benefit the article to focus on what you deem the most important findings. 43. Limitations are a bit sprinkled throughout the three subsections, it may help to bring them all together under a “strengths and limtations” subsection. 44. Around line 390, Again, it is stated these “overestimations” in the PAF-approach” but this hasn’t been motivated to the strength needed in the rest of the article. Overall conclusion 45. Hmmm. I’m not sure that you’ve developed the argument that the MCOD method is the most accurate, to use it as the gold standard to shoot for when developing the UCOD Enhanced method for Italy. Recall what the MCOD method is going – it is entirely ignoring all causes of death that are partially-attributable to alcohol (cancer, IHD, stroke, injury, and many more). Together, these make up the vast majority of AA deaths. It seems difficult to believe that a method that weights all of these deaths as 0.00, even though alcohol is known to be causative for all those conditions, is the gold standard that we should reach for. 46. For the MCOD analysis, again what the method does is not very logical. Any death that has any contributing diagnosis from among the ICD10 codes at line 120 is weight as being 100% caused by alcohol use (equivalently, that the death would not have occurred in the absence of exposure to alcohol use). If the death was really 100% caused by alcohol (i.e. there is no doubt that the death would not have occurred in the absence of exposure) why would the alcohol-specific diagnosis not be coded as the UCOD? It doesn’t make a lot of sense to suggest this. 47. At the least, both of these ideas need to be significantly developed in the article rationale, before it would be reasonable to shoot for the MCOD method as the gold standard. At the most, ignoring health conditions that would together for about 70% of AA mortality would be difficult to overcome in providing a new preferred method. ********** 6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). 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| Revision 1 |
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The impact of estimation methods for alcohol-attributable mortality on long-term trends for the general population and by educational level in Finland and Italy (Turin) PONE-D-23-15590R1 Dear Dr. Van Hemelrijck, 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, Sina Azadnajafabad, MD, MPH 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: You have rigourously addressed my proposed comments and changes, and are commended for a strong article! ********** 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: Yes: Adam Sherk ********** |
| Formally Accepted |
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PONE-D-23-15590R1 The impact of estimation methods for alcohol-attributable mortality on long-term trends for the general population and by educational level in Finland and Italy (Turin) Dear Dr. Van Hemelrijck: 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 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. Sina Azadnajafabad Academic Editor PLOS ONE |
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