Differential diagnosis of a calcified cyst found in an 18th century female burial site at St. Nicholas Church cemetery (Libkovice, Czechia)

Barbara Kwiatkowska; Agata Bisiecka; Łukasz Pawelec; Agnieszka Witek; Joanna Witan; Dariusz Nowakowski; Paweł Konczewski; Radosław Biel; Katarzyna Król; Katarzyna Martewicz; Petr Lissek; Pavel Vařeka; Anna Lipowicz

doi:10.1371/journal.pone.0254173

Peer Review History

Original SubmissionMarch 9, 2021
29 Apr 2021 Decision Letter - David Caramelli, Editor PONE-D-21-07709 Differential diagnosis of a cyst found in an 18th century female burial site at St. Nicholas Church cemetery (Libkovice, Czechia) PLOS ONE Dear Dr. Bisiecka, 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 Jun 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. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file. 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[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: N/A Reviewer #2: N/A ****** 3. Have the authors made all data underlying the findings in their manuscript fully available? 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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: Dear Authors, I really enjoyed to read this paper. I think it's very well written, original and with all the data provided. I recommend some minor revisions: - it is possible to establish the age range or the mean age and the stature for the female individual? - I will also add the brand of the machine utilised for X-Rays analyses - A 3D virtual reconstruction from the Ct-Scan of the cyst would be very interesting to see (for example a picture with the whole cyst and another one cut in norma sagittalis, so one can appreciate the section) Reviewer #2: The paper reads very well. I would recommend: 1) adding "calcified" to cyst in the title 2) pointing more on the diagnosis of lithopedion rather than the other one proposed 3) adding one more general, theoretical reference in the introduction for the workflow of palaeopathological diagnosis and its limitations: Rühli FJ, Galassi FM, Haeusler M. Palaeopathology: Current challenges and medical impact. Clin Anat. 2016 Oct;29(7):816-22. doi: 10.1002/ca.22709. ****** 6. 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Revision 1
18 May 2021 Author Response According to Reviewer #1 comments: 1.‘It is possible to establish the age range or the mean age and the stature for the female individual?’ Yes, it was possible to establish more precisely the age of the female individual. Based on the morphological details of the skull (cranial suture obliteration) and pelvis (pubic symphysis shape), as well as degenerative changes observed in the bone material, associated with the aging process (presence of articular cartilage degenerative lesions and degeneration of the vertebrae surface), considering that this individual was from a rural population, we estimated age as 45-55 years old. The individual was 158 cm tall, established by Trotter and Gleser method as well as Nainys method. We added this information in the ‘Anthropological analyses of the skeleton’ section (page 8, line 188-192) of the article. 2.‘I will also add the brand of the machine utilised for X-Rays analyses’ Of course you are right – thank you very much for pointing out this oversight. The machines used for X-Ray analyses were: a) for SEM: SEM, EVO LS15, Zeiss with energy dispersive X-ray analysis (SEM/EDX) applying quantax detector (Brüker) with 10 kV of filament tension; b) for X-Ray imaging: Konica Minolta Regius Model 110s with Siemens Polydoros LX 30 generator; c) for CT: Siemens Somatom Emotion 16 (3rd generation) apparatus. All the information was added to the ‘Methods’ part (page 7, line 173-174 & page 8, line 181-184) of the article. 3.‘A 3D virtual reconstruction from the Ct-Scan of the cyst would be very interesting to see (for example a picture with the whole cyst and another one cut in norma sagittalis, so one can appreciate the section)’ Regarding the inclusion of the three-dimensional reconstruction obtained from CT slices with the article: we agree that it would be an interesting form of object imaging, however, after detailed discussion, we decided that in our case it would not enrich the content of the article. 3D reconstructions are generally performed to reveal a closed object, the structure of which we do not want to break. In this case, we did a CT to view the internal structure of an object that we did not know what it was. After confirming that there were structures resembling human (fetal) bones inside, we extracted them from the inside of the shell to perform the necessary EDS and histological analyzes (which would not have been possible without physical interference with the object). Based on the scans (both in 512 dpi and 1080 dpi resolution), we made a 3D reconstruction, but its quality is not satisfactory (due to the delicate structure of the cyst, the edges of the three-dimensional object were blurred). We believe that this reconstruction does not bring significant informational or aesthetic value to the article, so we preferred to include in the article other images showing the way of our thinking from the general (outer view of burial no. 1095) to the detail (histological stainings and SEM). We hope that you will accept our explanation of this issue and lack of the 3D model does not decrease the value of this article. According to Reviewer #2 comments: 1.‘I would recommend adding "calcified" to cyst in the title’ We added the word ‘calcified’ to the article's title as suggested. 2.‘I would recommend pointing more on the diagnosis of lithopedion rather than the other one proposed’ Regarding the suggestion to emphasize the greater possibility that the discovered object is an example of a lithopedion: we cannot fully agree with this proposal as we believe that the information we have does not allow us to fully draw such a conclusion. Although many features of the object indicate its similarity to the diagnostic criteria of lithopedion, the EDS analysis of the shell indicated that it is not composed of calcium, which is known right now as the basic mechanism of lithopedion formation. We agree that lithopedion is more often described in the literature and has already been identified in the archaeological context, however – referring to the review article suggested by the Reviewer – the frequency of publications on a given phenomenon does not necessarily have to be related to the frequency (and therefore higher probability) of its occurrence. We decided to write this article in such a form that it does not resolve this issue unequivocally, in the hope that it will cause a discussion in the literature on the possibility of fetus-in-fetu occurrence in archaeological remains or on the possibility of lithopedion’s shell formation by a mechanism other than calcium deposition. We trust that such an approach will contribute to the deepening of the scientific potential. We have provided a brief overview of this explanation in the 'Conclusions' section (page 14, line 327-329) and we hope that the current form of the article will be acceptable. 3.‘I would recommend adding one more general, theoretical reference in the introduction for the workflow of palaeopathological diagnosis and its limitations: Rühli FJ, Galassi FM, Haeusler M. Palaeopathology: Current challenges and medical impact. Clin Anat. 2016 Oct;29(7):816-22. doi: 10.1002/ca.22709.’ We read the recommended article and agree that it provides valuable support for the content of our theoretical introduction. We referred to it in the 'Introduction' section (position 6 in the Bibliography). Attachments Attachment Submitted filename: Response to Reviewers.pdf https://doi.org/10.1371/journal.pone.0254173.r002
22 Jun 2021 Decision Letter - David Caramelli, Editor Differential diagnosis of a calcified cyst found in an 18th century female burial site at St. Nicholas Church cemetery (Libkovice, Czechia) PONE-D-21-07709R1 Dear Dr. Bisiecka, 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, David Caramelli, Ph.D Academic Editor PLOS ONE Additional Editor Comments (optional): Reviewers' comments: Reviewer's Responses to Questions Comments to the Author 1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation. Reviewer #1: All comments have been addressed 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 #1: Yes Reviewer #2: Yes ****** 3. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: N/A Reviewer #2: N/A ****** 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 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 #1: Yes 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 #1: Dear author/s, your reply is fine to me. You have made all the requested changes and I agree with you regarding the 3D virtual reconstruction. Reviewer #2: Thanks for making the requested edits and for explaining your views on the correct diagnostic approach. ****** 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: No Reviewer #2: No https://doi.org/10.1371/journal.pone.0254173.r003
Formally Accepted
25 Jun 2021 Acceptance Letter - David Caramelli, Editor PONE-D-21-07709R1 Differential diagnosis of a calcified cyst found in an 18^th century female burial site at St. Nicholas Church cemetery (Libkovice, Czechia) Dear Dr. Bisiecka: 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 Professor David Caramelli Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0254173.r004

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