Outcomes of children with clear cell sarcoma of kidney following NWTS strategies in Shanghai China (2003–2021)

Anan Zhang; Xiaojun Yuan; Shayi Jiang; Dongqing Xu; Can Huang; Jing yan Tang; Yijin Gao

doi:10.1371/journal.pone.0306863

Peer Review History

Original SubmissionDecember 17, 2023
20 Feb 2024 Decision Letter - Gregory Tiao, Editor PONE-D-23-37507Outcomes of children with clear cell sarcoma of kidney following NWTS strategies in Shanghai China (2003–2021)PLOS ONE Dear Dr. Gao, 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 Apr 04 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. 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: https://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, Gregory Tiao, M.D. Academic Editor PLOS ONE Journal requirements: 1. When submitting your revision, we need you to address these additional requirements. Please ensure that your manuscript meets PLOS ONE's style requirements, including those for file naming. 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. PLOS requires an ORCID iD for the corresponding author in Editorial Manager on papers submitted after December 6th, 2016. Please ensure that you have an ORCID iD and that it is validated in Editorial Manager. To do this, go to ‘Update my Information’ (in the upper left-hand corner of the main menu), and click on the Fetch/Validate link next to the ORCID field. This will take you to the ORCID site and allow you to create a new iD or authenticate a pre-existing iD in Editorial Manager. Please see the following video for instructions on linking an ORCID iD to your Editorial Manager account: " ext-link-type="uri" xlink:type="simple">https://www.youtube.com/watch?v=_xcclfuvtxQ". 3. We note that your Data Availability Statement is currently as follows: [All relevant data are within the manuscript and its Supporting Information files.] Please confirm at this time whether or not your submission contains all raw data required to replicate the results of your study. Authors must share the “minimal data set” for their submission. PLOS defines the minimal data set to consist of the data required to replicate all study findings reported in the article, as well as related metadata and methods (https://journals.plos.org/plosone/s/data-availability#loc-minimal-data-set-definition). For example, authors should submit the following data: - The values behind the means, standard deviations and other measures reported; - The values used to build graphs; - The points extracted from images for analysis. Authors do not need to submit their entire data set if only a portion of the data was used in the reported study. If your submission does not contain these data, please either upload them as Supporting Information files or deposit them to a stable, public repository and provide us with the relevant URLs, DOIs, or accession numbers. For a list of recommended repositories, please see https://journals.plos.org/plosone/s/recommended-repositories. If there are ethical or legal restrictions on sharing a de-identified data set, please explain them in detail (e.g., data contain potentially sensitive information, data are owned by a third-party organization, etc.) and who has imposed them (e.g., an ethics committee). Please also provide contact information for a data access committee, ethics committee, or other institutional body to which data requests may be sent. If data are owned by a third party, please indicate how others may request data access. Additional Editor Comments: Please address the questions raised in the review [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: No 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 describe the sites of metastasis in the 8 patients with Stage IV disease 2. Did LN status impact outcomes in the Stage III patients? 3. There is a mention of only using lung RT in cases where there was residual pulmonary disease after 6 weeks of systemic therapy. Does that mean that those patients with complete radiographic response did not get lung RT? It is important to highlight this point as this is a start contrast to the recommended therapy for lung metastasis in CCSK on NWTS/COG studies 4. Would recommend saying “margin status” rather than “envelope status” 5. Please provide data specifically about intraoperative tumor spill (as opposed to preoperative tumor rupture) as logically these are the cases that may have been possibly avoided with preoperative therapy. 6. Was tumor size correlated with intraoperative tumor rupture? 7. Please discuss how this information may be informative for future clinical practice or clinical trial conduct. It appears that the area for potential improvement may be around those with Stage III disease who may warrant more intensive therapy such as modified UH-1 as is used for Stage IV disease? 8. Please provide numbers at risk for your Kaplan Meier Curves Reviewer #2: This is a single institutional report on a tumor that has been largely described by cooperative group studies. While the series is large, this does not add anything additional from prior multi-institutional studies, and therapies employed are somewhat outdated. Well-written, but offers no new or insightful information. ****** 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: Nicholas Cost 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.0306863.r001
Revision 1
5 Mar 2024 Author Response Response to Reviewers Thank you for the constructive advice by editors and reviewers. We agree with the suggestion and have revised according to the comments. We will be happy to edit the paper further based on your helpful comments. We have highlight the changes of the manuscript by red font. To Editor: Thank you for all your help, support and good suggestions! 1)About figure files: We have resubmitted the figure files according to the guidelines of the journal， the PACE tool, thank you for your help and kindly reminder. 2)About the PLOS ONE’s style requirement: Thank you for providing us the PLOS ONE style templates of articles and authors and affiliations, we have revised according to the requirements. 3)About the ORCID ID for the corresponding author： Thank you for reminder and help, we have authenticated the pre-existing ORCID ID in the Editorial Manager. 4)About the Data Availability: As the journal requested the raw data to replicate all study findings reported in the article, we are willing to upload our excel file that contain the raw data as Supporting Information files. Thank you for your help. To Reviewer #1: � The reviewer thinks the authors have not made all the data full available. Response to reviewer: Thank you for the reminder. As the journal requested the raw data to replicate all study findings reported in the article, we are willing to upload our excel file that contain the raw data as Supporting Information files. Thank you for your help. 1)Please describe the sites of metastasis in the 8 patients with Stage IV disease Response to reviewer: Thank you for the reminder, we have explained it in the Result Part (Patient characteristics) and also added it in the abstract to facilitate reading. There were 8 patients with stage IV disease. 6 patients were lung metastasis, one patient was bone metastasis and one was presented with intra-orbital and cervical lymph node metastasis. 2) Did LN status impact outcomes in the Stage III patients? Response to the reviewer: The EFS of LN involvement is 61.5%±13.5%, while EFS of LN negative is 87.5 ±11.7%. X2=1.561, P=0.212,the fig is in the "response to reviewer" There was a trend that in stage III patients, LN involvement was related to inferior results, but the P value did not have significance. So we did not put it in the paper. We just mentioned the positive prognostic factor, what is your advice? 3)There is a mention of only using lung RT in cases where there was residual pulmonary disease after 6 weeks of systemic therapy. Does that mean that those patients with complete radiographic response did not get lung RT? It is important to highlight this point as this is a start contrast to the recommended therapy for lung metastasis in CCSK on NWTS/COG studies. Response to the reviewer: Yes, it means that if the patients with lung metastasis have a compete radiographic response after 6 weeks of systemic therapy, they did not receive lung RT anymore. We are so thankful for you to highlight this point, it is true that the practice of lung RT is different from COG/SIOP practice, and it brings hope that the prognosis is good without RT. “None of the six patients with lung metastasis received lung radiotherapy. In one case, the lung metastasis was surgically removed after chemotherapy, and in the other five cases, the lung metastases disappeared within six weeks after chemotherapy.” “And among lung metastasis, only one patient developed a relapse (CASE 3 in table) and are salvaged by RT after relapse and are now in second complete remission.” We have highlighted this point in the paper (treatment, event) and in the abstract and also added one paragraph to discuss about it, really thank you for pointing out it!! 4)Would recommend saying “margin status” rather than “envelope status” Response to the reviewer: Thank you for your advice, we have revised it. 5）Please provide data specifically about intraoperative tumor spill (as opposed to preoperative tumor rupture) as logically these are the cases that may have been possibly avoided with preoperative therapy. Response to the reviewer: Thank you for the advice, we have added it in the paper. Peri-operative tumor rupture occurred in 5 cases, and four cases were intraoperative tumor spill. One patient was seriously ill with preoperative tumor rupture. And we reviewed the operation note that we think some tumor spill was related to the inappropriate surgical practices by surgeons (some were receiving surgery in a not so experienced hospital in China and then transferred to our centers). 6)Was tumor size correlated with intraoperative tumor rupture? Response to the reviewer: This is a great point, thank you. I am sorry not all the operative note provided the exact tumor volume, some just provided the largest diameter of the tumor. From the available data, we can see: the median tumor size of upfront resection was 10cm (range 9.4cm-14.5cm), The median tumor size of tumor spill was 13.1cm(11-18cm) It seems larger tumor may have higher risk of tumor spill. Therefore, if the surgeons think the tumor has high risk of tumor spill (for example very large tumor volume?), preoperative chemotherapy or needle biopsy maybe more preferred. In the future, we should detect the risk factors of tumor spill, and investigate the selective use of preoperative chemotherapy or needle biopsy first in such high-risk patients. What is your suggestion about this point? Thank you for this question, we have added a little discussion in the paper. 7）Please discuss how this information may be informative for future clinical practice or clinical trial conduct. It appears that the area for potential improvement maybe around those with stage III disease who may warrant more intensive therapy such as modified UH-1 as is used for stage IV disease? Response to the reviewer: A. We think one reason of poor prognosis of stage III in our study is the high proportion of tumor rupture. And tumor spill seems to be a problem in developing country where surgeon is not experienced enough and there are often delays of diagnosis in developing countries. So we think if the surgeons think the tumor has high risk of tumor spill, preoperative chemotherapy or needle biopsy maybe more preferred. In the future, we should detect the risk factors of tumor spill, and investigate the selective use of preoperative chemotherapy or needle biopsy first in such high-risk patients. And the RT doses maybe adjusted for tumor rupture patients. B. For patients without tumor rupture, the EFS was about 86% in our study. But the patients with inferior vena cava tumor thrombus also had poor prognosis (41.7%), for such patient, a more intense chemotherapy regimen including carboplatin maybe indicated in the future. Thank you for your good suggestion, we have added a little discussion in the paper. Looking forward to your further advice. 8）Please provide numbers at risk for your Kaplan Meier Curves Response to the reviewer: Thank you for your suggestion, we have revised it in all figures. To Reviewer #2: This is a single institutional report on a tumor that has been largely described by cooperative group studies. While the series is large, this does not add anything additional from prior multi-institutional studies, and therapies employed are somewhat outdated. Well-written, but offers no new or insightful information. Response to the reviewer: Thank you for your suggestions, and let us think about the new insights of our study. 1)First, CCSK is a rare disease, so there are not so many papers involving large series. Our study is not a single institutional report, but a multicenter study with large series in developing country, I think this is one new sight. 2)And secondly, thank you for the reviewers’ wonderful suggestions. In our study, for lung metastasis patient, we are a contrast to the recommendation of NWTS and SIOP practice, we did not give lung RT if the patients achieve CR after 6 weeks of systemic treatment. And the long results are good, for only one of them developed a relapse (1/6). And he was also salvaged by RT after relapse and achieve a second complete remission. So lung RT for lung metastasis maybe omitted in some highly selected patients. 3)And thirdly, our results of stage 4 were better than the NWTS-5. Because of the rarity, the NWTS has not published their newest results of CCSK. 4)Lastly we find that tumor rupture is a main reason of poor prognosis of stage III patients which should be further pay attention on especially in developing countries. And the RT doses maybe adjusted for tumor rupture patients. We should detect the risk factors of tumor spill in the future, and investigate the selective use of neoadjuvant chemotherapy or needle biopsy first in such high-risk patients. We think our paper is worth publication, and we are happy if you can give us further suggestions to revise our paper. Thank you very much indeed!! Attachments Attachment Submitted filename: Response to Reviewers.docx https://doi.org/10.1371/journal.pone.0306863.r002
25 Jun 2024 Decision Letter - Gregory Tiao, Editor Outcomes of children with clear cell sarcoma of kidney following NWTS strategies in Shanghai China (2003–2021) PONE-D-23-37507R1 Dear Dr. Gao, 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, Gregory Tiao, M.D. Academic Editor PLOS ONE Additional Editor Comments (optional): The authors have addressed the concerns raised in previous review 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: Thank you for addressing the prior comments. Thank you for providing your experience for the wider medical community. ****** 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: Nicholas G. Cost ******** https://doi.org/10.1371/journal.pone.0306863.r003
Formally Accepted
28 Jun 2024 Acceptance Letter - Gregory Tiao, Editor PONE-D-23-37507R1 PLOS ONE Dear Dr. Gao, 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. Gregory Tiao Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0306863.r004

Open letter on the publication of peer review reports

PLOS recognizes the benefits of transparency in the peer review process. Therefore, we enable the publication of all of the content of peer review and author responses alongside final, published articles. Reviewers remain anonymous, unless they choose to reveal their names.

We encourage other journals to join us in this initiative. We hope that our action inspires the community, including researchers, research funders, and research institutions, to recognize the benefits of published peer review reports for all parts of the research system.

Learn more at ASAPbio .