Quantitative assessment of interstitial lung disease in Sjögren’s syndrome

Pablo Guisado-Vasco; Mario Silva; Miguel Angel Duarte-Millán; Gianluca Sambataro; Chiara Bertolazzi; Mauro Pavone; Isabel Martín-Garrido; Oriol Martín-Segarra; José Manuel Luque-Pinilla; Daniele Santilli; Domenico Sambataro; Sebastiano E. Torrisi; Ada Vancheri; Marwin Gutiérrez; Mayra Mejia; Stefano Palmucci; Flavio Mozzani; Jorge Rojas-Serrano; Carlo Vanchieri; Nicola Sverzellati; Alarico Ariani

doi:10.1371/journal.pone.0224772

Peer Review History

Original SubmissionJuly 25, 2019
21 Aug 2019 Decision Letter - Masataka Kuwana, Editor Transfer Alert This paper was transferred from another journal. As a result, its full editorial history (including decision letters, peer reviews and author responses) may not be present. PONE-D-19-20968 Quantitative assessment of interstitial lung disease in Sjögren’s syndrome. PLOS ONE Dear Dr. Guisado-Vasco, 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. Specifically, both reviewers found out some interests in this study, but also pointed out a number of criticisms that require improvement or even amendment. I ask the authors to fully respond to all points made by reviewers. We would appreciate receiving your revised manuscript by Oct 05 2019 11:59PM. 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The PLOS ONE style templates can be found at http://www.journals.plos.org/plosone/s/file?id=wjVg/PLOSOne_formatting_sample_main_body.pdf and http://www.journals.plos.org/plosone/s/file?id=ba62/PLOSOne_formatting_sample_title_authors_affiliations.pdf 2. Thank you for stating the following in the Competing Interests section: Prof. Carlo Vancheri is part of F. Hoffmann-La Roche Ltd. Scientific Board. He has received consulting fees and/or speaker fees from Astrazeneca, Boehringer Ingelheim, Chiesi, F. Hoffmann-La Roche Ltd and Menarini. Prof. Stefano Palmucci has reveived personal fees and honoraria for lectures from Boehringer Ingelheim, Delphi International srl and F. Hoffmann-La Roche Ltd. He has been included in the scientific board for Boehringer Ingelheim. None of the other authors have any potential conflicts of interest to disclose in relation to this work. Please confirm that this does not alter your adherence to all PLOS ONE policies on sharing data and materials, by including the following statement: "This does not alter our adherence to PLOS ONE policies on sharing data and materials.” (as detailed online in our guide for authors http://journals.plos.org/plosone/s/competing-interests). If there are restrictions on sharing of data and/or materials, please state these. Please note that we cannot proceed with consideration of your article until this information has been declared. Please include your updated Competing Interests statement in your cover letter; we will change the online submission form on your behalf. Please know it is PLOS ONE policy for corresponding authors to declare, on behalf of all authors, all potential competing interests for the purposes of transparency. PLOS defines a competing interest as anything that interferes with, or could reasonably be perceived as interfering with, the full and objective presentation, peer review, editorial decision-making, or publication of research or non-research articles submitted to one of the journals. Competing interests can be financial or non-financial, professional, or personal. Competing interests can arise in relationship to an organization or another person. Please follow this link to our website for more details on competing interests: http://journals.plos.org/plosone/s/competing-interests [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: Partly Reviewer #2: Yes ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: No Reviewer #2: Yes ****** 3. Have the authors made all data underlying the findings in their manuscript fully available? The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified. Reviewer #1: Yes Reviewer #2: Yes ****** 4. Is the manuscript presented in an intelligible fashion and written in standard English? PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here. Reviewer #1: Yes Reviewer #2: Yes ****** 5. Review Comments to the Author Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters) Reviewer #1: The authors reported quantitative assessment of interstitial lung disease in Sjögren’s syndrome. It is interesting to focus the usefulness of quantitative CT in Sjögren’s syndrome-ILD, but I have some major concerns about the data Major; 1. CTD-ILD and quantitative CT scores have already been studied, but most patients were scleroderma. Therefore, it was very interesting to examine the quantitative CT score for Sjogren's syndrome in a large number of people. However, only association between CT analysis by histogram and ILD type are not appropriate as a paper. Regarding scleroderma, CT scores and prognosis have already been examined. Did you classify ILD as limited and extensive, but was it related to prognosis as well as scleroderma? You need to consider if CT score affects prognosis in Sjogren's syndrome. 2. The percentage of patients with pSS-ILD is 72% (Table 1). The secondary SS needs to be described in detail. 3. It seems necessary to create a table of correlation between quantitative CT analysis and clinical findings, respiratory function tests, and semi-quantitative CT analysis. Minor 1.The figure is difficult to see and should be considered. 2 .In figure1. 2, the statistical differences between the three groups need to be clearly stated. Reviewer #2: The authors reported the usefulness of quantitative chest computed tomography (QCT) assessment of interstitial lung disease (ILD) in patients with Sjögren’s syndrome (SS). In this multi-center and retrospective study, QCT indices identified patients with SS and ILD (SS-ILD), and discriminated those with lesser or greater lung disease. This is an important study that demonstrates that QCT indices can characterize subjects with SS-ILD in comparison to the standard visual, semi-quantitative methods such as Goh and Taouli scoring. Major 1. This study showed that QCT indices discriminated the severity of ILD in patients with SS. However, optimal cut-off points for each indicator were not determined. How QCT indices can be utilized in future research. 2. With respect to Taouli scores, in their original paper (Eur Radiol 2002; 12:1504-1511), the authors calculated scores including ground-glass attenuation, honeycombing, centrilobular nodules, reticular pattern, mosaic perfusion, and air trapping. In the present study, how were the Taouli scores determined ? Minor 1. There were no descriptions of the correlation between QCT indices and the Goh and Taouli scores or pulmonary function test findings in Table 1. Please modify the descriptions. ****** 6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files. If you choose “no”, your identity will remain anonymous but your review may still be made public. Do you want your identity to be public for this peer review?** For information about this choice, including consent withdrawal, please see our Privacy Policy. Reviewer #1: No Reviewer #2: No [NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. Please log into your account, locate the manuscript record, and check for the action link "View Attachments". If this link does not appear, there are no attachment files to be viewed.] 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. 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Revision 1
26 Sep 2019 Author Response Dear Editor, Thank you for considering our manuscript, ‘Quantitative assessment of interstitial lung disease in Sjögren’s syndrome’ (PONE-D-19-20968), sent to PLOS One. We really appreciate the opportunity to review our manuscript in order to fully address your concerns and comments of the referee. Then, we are going to address point by point the reviewer’s recommendations. 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 http://www.journals.plos.org/plosone/s/file?id=wjVg/PLOSOne_formatting_sample_main_body.pdf and http://www.journals.plos.org/plosone/s/file?id=ba62/PLOSOne_formatting_sample_title_authors_affiliations.pdf I have reviewed the POLS ONE’s style guidelines, as you pointed out. First, I have added a title page to the manuscript body, according to the format recommended by your editorial office. Second, I have done some changes in the body of the template/manuscript according to them -mainly headings, as required. 2. Thank you for stating the following in the Competing Interests section: Prof. Carlo Vancheri is part of F. Hoffmann-La Roche Ltd. Scientific Board. He has received consulting fees and/or speaker fees from Astrazeneca, Boehringer Ingelheim, Chiesi, F. Hoffmann-La Roche Ltd and Menarini. Prof. Stefano Palmucci has reveived personal fees and honoraria for lectures from Boehringer Ingelheim, Delphi International srl and F. Hoffmann-La Roche Ltd. He has been included in the scientific board for Boehringer Ingelheim. None of the other authors have any potential conflicts of interest to disclose in relation to this work. Please confirm that this does not alter your adherence to all PLOS ONE policies on sharing data and materials, by including the following statement: "This does not alter our adherence to PLOS ONE policies on sharing data and materials.” (as detailed online in our guide for authors http://journals.plos.org/plosone/s/competing-interests). If there are restrictions on sharing of data and/or materials, please state these. Please note that we cannot proceed with consideration of your article until this information has been declared. Please include your updated Competing Interests statement in your cover letter; we will change the online submission form on your behalf. Please know it is PLOS ONE policy for corresponding authors to declare, on behalf of all authors, all potential competing interests for the purposes of transparency. PLOS defines a competing interest as anything that interferes with, or could reasonably be perceived as interfering with, the full and objective presentation, peer review, editorial decision-making, or publication of research or non-research articles submitted to one of the journals. Competing interests can be financial or non-financial, professional, or personal. Competing interests can arise in relationship to an organization or another person. Please follow this link to our website for more details on competing interests: http://journals.plos.org/plosone/s/competing-interests I confirm, it does not alter the conflict of interest policy of your journal. I have added the following sentence, as recommended: ‘This does not alter our adherence to PLOS ONE policies on sharing data and materials’. Reviewer #1: The authors reported quantitative assessment of interstitial lung disease in Sjögren’s syndrome. It is interesting to focus the usefulness of quantitative CT in Sjögren’s syndrome-ILD, but I have some major concerns about the data Major; 1. CTD-ILD and quantitative CT scores have already been studied, but most patients were scleroderma. Therefore, it was very interesting to examine the quantitative CT score for Sjogren's syndrome in a large number of people. However, only association between CT analysis by histogram and ILD type are not appropriate as a paper. Regarding scleroderma, CT scores and prognosis have already been examined. Did you classify ILD as limited and extensive, but was it related to prognosis as well as scleroderma? You need to consider if CT score affects prognosis in Sjogren's syndrome. Before any new diagnostic technique can be used as an outcome measurement instrument, it should be tested its reliability comparing to actual standards. It is the aim of the present research. Later, it could be used as a prognostic tool – it is our next step in the research. In fact, the development of the quantitative indices in systemic sclerosis followed up the same idea of our working group – the count with some members whose developed this scoring system in scleroderma. First, it was release a publication assessing the reliability of these indices (2015), and them, a model of prediction of mortality was developed (2017). References Jousse-Joulin S, et al. Video clip assessment of a salivary gland ultrasound scoring system in Sjögren’s syndrome using consensual definitions: an OMERACT ultrasound working group reliability exercise. Ann Rheum Dis 2019;78:967–973 Ariani A et al. Operator-independent quantitative chest computed tomography versus standard assessment of interstitial lung disease related to systemic sclerosis: a multi-center study. Mod Rheumatol 2015; 25(5):724-30. Ariani A et al. Quantitative chest computed tomography is associated to two prediction models of mortality in interstitial lung disease related to systemic sclerosis. Rheumatology (Oxford), 2017; 56(6): 922-927. 2. The percentage of patients with pSS-ILD is 72% (Table 1). The secondary SS needs to be described in detail. A paragraph was added at the beginning of the results section, detailing the diseases associated to SS. The diseases associated to the development of SS, secondary SS, were: rheumatoid arthritis (5,9%), systemic lupus erythematosus (2%), systemic sclerosis (12,7%), and undifferentiated connective tissue disease (9,8%). Not all cases were affected of ILD, of course: just 10 have secondary SS. 3. It seems necessary to create a table of correlation between quantitative CT analysis and clinical findings, respiratory function tests, and semi-quantitative CT analysis. This table has been inserted in the text – previously it was designed as supplementary material. It has been named as table 2. Hence, the former table 2 as labeled now as table 3. Minor 1.The figure is difficult to see and should be considered. The figures have been edited and introduced some changes to maximize its resolution. 2 .In figure1. 2, the statistical differences between the three groups need to be clearly stated. Thank you very much for this comment. In order to a better understanding and simplified of the images, it has been added a footnote in both set of figures, which explains the differences between every group (0,1,2), and corresponding p-values. Reviewer #2: The authors reported the usefulness of quantitative chest computed tomography (QCT) assessment of interstitial lung disease (ILD) in patients with Sjögren’s syndrome (SS). In this multi-center and retrospective study, QCT indices identified patients with SS and ILD (SS-ILD), and discriminated those with lesser or greater lung disease. This is an important study that demonstrates that QCT indices can characterize subjects with SS-ILD in comparison to the standard visual, semi-quantitative methods such as Goh and Taouli scoring. Major 1. This study showed that QCT indices discriminated the severity of ILD in patients with SS. However, optimal cut-off points for each indicator were not determined. How QCT indices can be utilized in future research. It was inserted/added a new table (number 4) to sum up these cut-off points, according to the Youden index applying using the model of area under the curve. A brief comment has been inserted in the results section. I am agreeing with the point, that these cut-off points could be use in the next step of the research as predictive models of mortality in ILD related to SS. 2. With respect to Taouli scores, in their original paper (Eur Radiol 2002; 12:1504-1511), the authors calculated scores including ground-glass attenuation, honeycombing, centrilobular nodules, reticular pattern, mosaic perfusion, and air trapping. In the present study, how were the Taouli scores determined ? These scores were calculating by two expert radiologists, properly trainee in semiquantitative scores. Hence, the score was calculated by the sum of all the categories that represent lung fibrosis, namely: honeycombing, reticular pattern, and ground-glass attenuation. Minor 1. There were no descriptions of the correlation between QCT indices and the Goh and Taouli scores or pulmonary function test findings in Table 1. Please modify the descriptions. A new table (Table 2) has been inserted where the correlations are described as recommended. It is mentioned in the results paragraph. We hope that these comments and improvements have fulfilled the referees’ recommendations. Such way, your editorial office could consider the current version of the manuscript for its final acceptance. If there are any further questions or comments after this revision, please do not hesitate to contact us. Sincerely, Pablo Guisado Vasco Attachments Attachment Submitted filename: Response to reviewers v.1.0.docx https://doi.org/10.1371/journal.pone.0224772.r002
22 Oct 2019 Decision Letter - Masataka Kuwana, Editor Quantitative assessment of interstitial lung disease in Sjögren’s syndrome. PONE-D-19-20968R1 Dear Dr. Guisado-Vasco, We are pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it complies with all outstanding technical requirements. Within one week, you will receive an e-mail containing information on the amendments required prior to publication. When all required modifications have been addressed, you will receive a formal acceptance letter and your manuscript will proceed to our production department and be scheduled for publication. Shortly after the formal acceptance letter is sent, an invoice for payment will follow. To ensure an efficient production and billing process, please log into Editorial Manager at https://www.editorialmanager.com/pone/, click the "Update My Information" link at the top of the page, and update your user information. 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 enable them to help maximize its impact. If they will be preparing press materials for this manuscript, you must inform our press team as soon as possible and 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. With kind regards, Masataka Kuwana, MD, PhD 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: Thank you for addressing my comments from initial review.The data are potentially interesting and worthy of evaluation for SS-ILD. ****** 7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files. If you choose “no”, your identity will remain anonymous but your review may still be made public. Do you want your identity to be public for this peer review?** For information about this choice, including consent withdrawal, please see our Privacy Policy. Reviewer #2: No https://doi.org/10.1371/journal.pone.0224772.r003
Formally Accepted
1 Nov 2019 Acceptance Letter - Masataka Kuwana, Editor PONE-D-19-20968R1 Quantitative assessment of interstitial lung disease in Sjögren’s syndrome Dear Dr. Guisado-Vasco: I am 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 notify them about your upcoming paper at this point, to enable them to help maximize its impact. If they will be preparing press materials for this manuscript, 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. For any other questions or concerns, please email plosone@plos.org. Thank you for submitting your work to PLOS ONE. With kind regards, PLOS ONE Editorial Office Staff on behalf of Prof. Masataka Kuwana Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0224772.r004

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