Incidence of extraovarian clear cell cancers in women with surgically diagnosed endometriosis: A cohort study

Liisu Saavalainen; Heini Lassus; Anna But; Mika Gissler; Oskari Heikinheimo; Eero Pukkala

doi:10.1371/journal.pone.0253270

Peer Review History

Original SubmissionFebruary 2, 2021
31 Mar 2021 Decision Letter - Paola Viganò, Editor PONE-D-21-03577 Incidence of extraovarian clear cell cancers in women with surgically diagnosed endometriosis: a cohort study PLOS ONE Dear Dr. Heikinheimo, 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. Three Reviewers have assessed this manuscript. Their observations on the scientific reliability of the study are very different. The Authors should carefully consider the issues raised and revise the manuscript accordingly. Please submit your revised manuscript by May 13 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. 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Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: Yes Reviewer #2: I Don't Know Reviewer #3: 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 Reviewer #3: 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: No Reviewer #3: 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 research article is very interesting and answers one important question in endometriosis and oncology. The document is well written with a clear aim and methodology. The research includes a large group population and this obviously contributes on the strength of the research All the current researches on this topic have been correctly cited in the article and discussed on the discussion session. Reviewer #2: The present study aims at evaluating the association between extraovarian clear cell carcinomas and the diagnosis of endometriosis. I have several concerns regarding the consistency of this study and in my opinion the results and conclusions do not reliably represent an original finding on this topic. - Some of the limitations of the present study are typical of all studies derived from population based national registries o the diagnosis/coding might not be punctual and patient follow up might not be precise o most commonly only the main diagnosis is indicated (as stated by the Authors themselves) o Histopathological review in all these cases is not available. This procedure in case of clear cell histotype, as for other rare tumors, should be mandatory in order to provide a reliable analysis. - Another concern is that the reported cases of clear cell tumors might not be really associated with endometriosis (the definition of endometriosis associated tumors in not simply the coexistence of these two conditions but has to fulfill precise histopathological criteria). - I do not understand the relevance and usefulness of Table 1 for data and results interpretation.To me data analysis , result interpretation , as present in this paper, is not very eady for the reader. Reviewer #3: I read manuscript entilted "Incidence of extraovarian clear cell cancers in women with surgically diagnosed endometriosis: a cohort study" with great interest. Endometriosis is known risk factor for ovarian clear cell cancer but there is no easy pathological explanation for possible influence of endometriosis on extraovarian clear cell cancer and why it even should be. And it was also proven by the Authors. But this main conculusion should be also supported by data from "general population". Please describe general population - how it was choosen? what was age structure of general population? what was the incidence of extraovarian clear cell cancer in general population? I found only statement that "The risk of extraovarian clear cell cancers in women with surgically diagnosed endometriosis is similar to that in the general population." but I did not find any data from general population or clarifications in disussion which can prove this thesis. The other main point to correct is that this study refers only to finnish population. Please state this clearly in abstract and in the main text. The results of similar study conducted in Asiatic populations may have different results, because of the known higher incidence of clear cell cancer among Asians. The explanation in discussion section about Finnish Hospital Discharge Register and Finnish Cancer Registry I found very important and supportive. ****** 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 Reviewer #3: Yes:** Maria Szubert, MD [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.0253270.r001
Revision 1
9 May 2021 Author Response Thank you for the opprotunity to revise our manuscript. -We have made some stylistic changes in the manuscript according to the journal requirements. -The ethics committee of Hospital District of Helsinki and Uusimaa (238/13/03/03/2013) approved the study. The data were fully anonymized. As this is a registry based study, no informed consent was required. -The Hospital District of Helsinki and Uusimaa has funded this research project. The first author (LS) has received salary for a week to be able to write this article. The funder had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. -The data of the study are available upon request. There are legal restrictions on sharing this de-identified data set, imposed by Finnish Institute for Health and Welfare. The possible request can be made in www.findata.fi. - And thank you for your comments on our manuscript. Please find enclosed our revised manuscript entitled “The incidence of non-ovarian clear cell cancer in women with surgically diagnosed endometriosis: a cohort study”, a manuscript with tract changes, and our point-by-point responses to reviewers’ comments. We tried to do our best to answer and renew the manuscript. Attachments Attachment Submitted filename: RESPONSE TO REVIEWERS290421LS.docx https://doi.org/10.1371/journal.pone.0253270.r002
19 May 2021 Decision Letter - Paola Viganò, Editor PONE-D-21-03577R1 Incidence of extraovarian clear cell cancers in women with surgically diagnosed endometriosis: a cohort study PLOS ONE Dear Dr. Heikinheimo, 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. ============================== ACADEMIC EDITOR: The manuscript has been improved. There is a minor issue that should be addressed. The Authors extensively discussed the types of cancer detected in endometriosis patients in the Discussion but, apart from the table, the Results section does not mention this topic at all. A discussion should be referred to the findings observed. A brief description of the cancer types observed would be useful also in the Results section. Abstract: there is a repetition in the Results ============================== Please submit your revised manuscript by Jul 03 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. Please include the following items when submitting your revised manuscript: A rebuttal letter that responds to each point raised by the academic editor and reviewer(s). You should upload this letter as a separate file labeled 'Response to Reviewers'. A marked-up copy of your manuscript that highlights changes made to the original version. You should upload this as a separate file labeled 'Revised Manuscript with Track Changes'. An unmarked version of your revised paper without tracked changes. You should upload this as a separate file labeled 'Manuscript'. If you would like to make changes to your financial disclosure, please include your updated statement in your cover letter. Guidelines for resubmitting your figure files are available below the reviewer comments at the end of this letter. If applicable, we recommend that you deposit your laboratory protocols in protocols.io to enhance the reproducibility of your results. Protocols.io assigns your protocol its own identifier (DOI) so that it can be cited independently in the future. For instructions see: http://journals.plos.org/plosone/s/submission-guidelines#loc-laboratory-protocols. Additionally, PLOS ONE offers an option for publishing peer-reviewed Lab Protocol articles, which describe protocols hosted on protocols.io. Read more information on sharing protocols at https://plos.org/protocols?utm_medium=editorial-email&utm_source=authorletters&utm_campaign=protocols. We look forward to receiving your revised manuscript. Kind regards, Paola Viganò Academic Editor PLOS ONE Journal Requirements: 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.] [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.0253270.r003
Revision 2
25 May 2021 Author Response Thank you for your comments concerning our manuscript. Please find enclosed our replies and revisions: _______________________________ ACADEMIC EDITOR: 1) The manuscript has been improved. There is a minor issue that should be addressed. The Authors extensively discussed the types of cancer detected in endometriosis patients in the Discussion but, apart from the table, the Results section does not mention this topic at all. A discussion should be referred to the findings observed. A brief description of the cancer types observed would be useful also in the Results section. _______________________________ 1) Thank you. Results section very briefly summarizes all our results shown in Table 2. To highlight that we also studied the specific sites of clear cell cancer added some words in the text. (page 8, lines 156-157) "Altogether 23 extraovarian clear cancers were observed in women with history of surgically diagnosed endometriosis; the expected number of cases based on the incidence rate in comparable women of the Finnish female population was 26. The incidence of extraovarian clear cell cancer in women with endometriosis was not increased in any age category, specific type of endometriosis, or specific site of cancer (intestine, cervix, uterus, kidney, urinary organs). All cancer diagnoses had been made more than 5 years after the date of endometriosis surgery. (Table 2)" In Discussion the first section presented other similar-like studies, the second association with clear cell cancers in kidney, uterine and cervix cancer, and the third bladder, urethra and intestinal cancers. We would like to keep that in our manuscript. ______________________________ ACADEMIC EDITOR: 2) Abstract: there is a repetition in the Results ______________________________ 2) We have corrected this repetition. Attachments Attachment Submitted filename: RESPONSE TO REVIEWERS250521.docx https://doi.org/10.1371/journal.pone.0253270.r004
2 Jun 2021 Decision Letter - Paola Viganò, Editor Incidence of extraovarian clear cell cancers in women with surgically diagnosed endometriosis: a cohort study PONE-D-21-03577R2 Dear Dr. Heikinheimo, 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, Paola Viganò Academic Editor PLOS ONE Additional Editor Comments (optional): The manuscript is now suitable for publication Reviewers' comments: https://doi.org/10.1371/journal.pone.0253270.r005
Formally Accepted
18 Jun 2021 Acceptance Letter - Paola Viganò, Editor PONE-D-21-03577R2 Incidence of extraovarian clear cell cancers in women with surgically diagnosed endometriosis: A cohort study Dear Dr. Heikinheimo: 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. Paola Viganò Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0253270.r006

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