Care and support when a baby is stillborn: A systematic review and an interpretive meta-synthesis of qualitative studies in high-income countries

Margareta Persson; Ingegerd Hildingsson; Monica Hultcrantz; Maja Kärrman Fredriksson; Nathalie Peira; Rebecca A. Silverstein; Josefin Sveen; Carina Berterö

doi:10.1371/journal.pone.0289617

Peer Review History

Original SubmissionFebruary 8, 2023
18 Apr 2023 Decision Letter - Pracheth Raghuveer, Editor PONE-D-23-03683Care and support when a baby is stillborn: A systematic review and an interpretive meta-synthesis of qualitative studiesPLOS ONE Dear Dr. Bertero, 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 02 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: 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, Pracheth Raghuveer, MD, DNB Academic Editor PLOS ONE Journal Requirements: When submitting your revision, we need you to address these additional requirements. 1. 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 [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 ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: N/A ****** 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 ****** 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 ****** 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: Thank you for submitting this interesting qualitative review. It was an interesting read, however, I think there are some points that require exploration. I will note these in the order in which they appeared on the submitted text. Title and Abstract: • The title of the review does not indicate that the context refers to high income countries only nor is this apparent in the Abstract. Abstract: • The introduction section is a little confusing and could be more focused. The last sentence in this section, in particular, is long and confusing. Methods: • the opening line of the methods is not clear. What do you mean by “This article is an update of the qualitative part of a larger health technologies assessment covering both qualitative and quantitative aspects of care and support after stillbirth …”, please review. • Line 125 on page 6, what do you mean by adjustments to the PRISMA Guidance in this context, did the review team consider also using a reporting guideline developed specifically for qualitative systematic reviews? • Line 129 page 6 – what do you mean by a generic term in this sentence? • While the methods advanced by Barbara Paterson are mentioned in this closing paragraph, the narrative is not explicit and I suggest that this could be reviewed and made more accessible for a reader. Study selection: • Why were studies in relation to miscarriage, stillbirth and neonatal death set for inclusion if 50% of the participants experienced a stillbirth? What informed this %? Critical Appraisal: • The manner in which the critical appraisal informed the final number of studies included in this review is not clearly articulated in this section. I suggest that you make this more explicit. • It is also worth considering how the study selection, that occurred at this stage, impacted the characteristic of studies included in this review. Data extraction and Analysis: • Is the concept of synthesis missing from this heading? • The review team refer to synthesised findings as fusions in this sections and others. Perhaps a concise explanation as to why they do this, aligned to the methodology, would be helpful for the notice researcher when reading this study. Confidence in synthesis findings: • The placing of this section in the manuscript could be considered so that the operationalisation of this method is included in the Methods section of the paper. And the findings or the assessment made when applying the GRADECERQual framework is noted in the findings section. Validation of the findings using a patient-led co-designed focus group study: • This is an interesting approach but the process is not entirely clear. Why was this study selected to inform the triangulation, how did this study relate to the eligibility criteria and screening of the review? More information in relation to the rational here would be welcome. Discussion: • Some repetition of narrative noted in this section, for reader comfort please review. Strengths and Limitations: • The opening line of this section needs to be reviewed. • This section may need to be reviewed in the context of other feedback offered to ensure that all limitations are noted. Summary of confidence: • The summary of findings statements are long and contain different elements of the reported finding. It is not clear if all studies aligned in Table 2 contribute to all the elements of the finding. This needs to be reconsidered and perhaps the GRADECERQual assessment may need to be applied to a summary of findings. ****** 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 ******** [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.0289617.r001
Revision 1
26 Apr 2023 Author Response Care and support when a baby is stillborn: A systematic review and an interpretive meta-synthesis of qualitative studies Response to reviewer’s comments We thank the reviewer for their comments about the manuscript and recognition that this is “an interesting paper”. Accordingly, we have revised the manuscript (shown in red colour) as requested. Point-by-point responses to the reviewer’s comments are given below. Reviewer 1: The title of the review does not indicate that the context refers to high income countries only nor is this apparent in the Abstract. Authors: The title and abstract have been reviewed to clarify the high-income context Reviewer 1: The introduction section (in the abstract) is a little confusing and could be more focused. The last sentence in this section, in particular, is long and confusing. Authors: The introduction paragraph has been revised and, hopefully, more distinct now. Reviewer 1; Method. the opening line of the methods is not clear. What do you mean by “This article is an update of the qualitative part of a larger health technologies assessment covering both qualitative and quantitative aspects of care and support after stillbirth …”, please review. Authors: We used the wording update to elaborate on the qualitative part of the text in this paper. So, to not confuse the readers, we have deleted “this is an update…” Reviewer 1: Line 125 on page 6, what do you mean by adjustments to the PRISMA Guidance in this context, did the review team consider also using a reporting guideline developed specifically for qualitative systematic reviews? Authors: Thank you for this comment. The only adjustment done is that some items in the checklist are reported in other places than suggested in this PRISMA checklist. So, we are pleased to delete this statement. Reviewer 1: Line 129 page 6 – what do you mean by a generic term in this sentence? Authors: Generic means that it is shared by or relating to a group of similar things, in this case, interpretive integration, rather than any particular method or synthesis. Synonyms are common, standard, general, etc. We have changed the word generic to common. Reviewer 1: While the methods advanced by Barbara Paterson are mentioned in this closing paragraph, the narrative is not explicit, and I suggest that this could be reviewed and made more accessible for a reader. Authors: Our apologies; we cannot demand that all readers should know about Paterson. We have added some text about Paterson’s three processes: meta-data analysis, meta-method, and meta-theory. Hopefully, this paragraph is now more accessible to a reader. Reviewer 1; Study selection. Why were studies in relation to miscarriage, stillbirth and neonatal death set for inclusion if 50% of the participants experienced a stillbirth? What informed this %? Authors: We aimed to focus on the experiences of stillbirth and no other pregnancy or infant losses for our review. Thus, we wanted to include papers where most study participants had that experience or studies that explicitly separated the findings related to stillbirth in their result presentations. We have added some additional information to this paragraph to illustrate this inclusion. Reviewer 1; Critical appraisal. The manner in which the critical appraisal informed the final number of studies included in this review is not clearly articulated in this section. I suggest that you make this more explicit. Authors: We have elaborated on the paragraph of the critical appraisal procedure further to show the systematical and independent manner this evaluation was performed. Hopefully, this procedure is more clearly articulated now. Reviewer 1: It is also worth considering how the study selection, that occurred at this stage, impacted the characteristic of studies included in this review. Authors: Please, see the comment above. We hope that the elaborated and more detailed description has clarified any concerns. Reviewer 1: Data extraction and Analysis. Is the concept of synthesis missing from this heading? Authors: Thank you for pointing this out. We changed analysis to synthesis. Reviewer 1: The review team refer to synthesised findings as fusions in this sections and others. Perhaps a concise explanation as to why they do this, aligned to the methodology, would be helpful for the notice researcher when reading this study. Authors: We are using Gadamer’s hermeneutics in our interpretation and synthesis. In his “method,” he discusses different horizons that merge into fusions. A theme comprises participants’ accounts characterising perceptions and/or experiences the researcher sees as relevant to the research question. A fusion is when both horizons from the text and the interpreter are expanding and giving a broader and deeper understanding than just answering a question. Additional text is added to the manuscript to explain further. Reviewer 1: Confidence in synthesis findings: The placing of this section in the manuscript could be considered so that the operationalisation of this method is included in the Methods section of the paper. And the findings or the assessment made when applying the GRADECERQual framework is noted in the findings section. Authors: This section is moved to the end of the methods section to show the logical flow of the analysis better. Reviewer 1: Validation of the findings using a patient-led co-designed focus group study: This is an interesting approach but the process is not entirely clear. Why was this study selected to inform the triangulation, how did this study relate to the eligibility criteria and screening of the review? More information in relation to the rational here would be welcome. Authors: We aimed to identify important aspects of care and support when babies are stillborn in high-income countries. The paper by Gillis and co-authors is performed in Canada (a high-income country with a low prevalence of stillbirth). It presents 15 recommendations to enhance bereavement care for parents based on parental experiences of stillbirth. We excluded this paper at the critical appraisal stage as it did not meet our inclusion criteria. Later we found that these recommendations mirrored our findings to a large extent. Thus, this paper which had another focus and was performed in a similar high-income country, could help discuss and validate our findings. Some short explanations are added to the manuscript. Reviewer 1: Discussion: • Some repetition of narrative noted in this section, for reader comfort please review. Authors: Thank you for pointing this out. We have revised the discussion section and hope all repetition is removed. Reviewer 1: Strengths and Limitations: The opening line of this section needs to be reviewed. Authors: The initial sentence in this paragraph is revised. Reviewer 1: This section may need to be reviewed in the context of other feedback offered to ensure that all limitations are noted. Authors: Some revisions of the strength and limitations text sections have been made. Hopefully, the limitations of the paper are addressed now. Reviewer 1: Summary of confidence. The summary of findings statements are long and contain different elements of the reported finding. It is not clear if all studies aligned in Table 2 contribute to all the elements of the finding. This needs to be reconsidered and perhaps the GRADECERQual assessment may need to be applied to a summary of findings. Authors: Thank you for your suggestion. In table 2 it is stated how many studies that contributed to the findings and those who did not are mentioned in the text Attachments Attachment Submitted filename: Response to reviewer.docx https://doi.org/10.1371/journal.pone.0289617.r002
24 Jul 2023 Decision Letter - Pracheth Raghuveer, Editor Care and support when a baby is stillborn: A systematic review and an interpretive meta-synthesis of qualitative studies in high-income countries PONE-D-23-03683R1 Dear author, 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, Pracheth Raghuveer, MD, DNB 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: 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 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: (No Response) Reviewer #2: An extremely fascinating and well-organized topic for research. I want to thank you for responding to the reviewers' comments. ****** 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: Yes: Mena Abdalla ******** https://doi.org/10.1371/journal.pone.0289617.r003
Formally Accepted
2 Aug 2023 Acceptance Letter - Pracheth Raghuveer, Editor PONE-D-23-03683R1 Care and support when a baby is stillborn: A systematic review and an interpretive meta-synthesis of qualitative studies in high-income countries Dear Dr. Berterö: 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. Pracheth Raghuveer Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0289617.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 .