Peer Review History

Original SubmissionMarch 22, 2024
Decision Letter - Tamara Sljivancanin Jakovljevic, Editor

PONE-D-24-05781Twin home birth: Outcomes of 100 sets of twins in the care of a single practitionerPLOS ONE

Dear Dr. Freeze,

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 Sep 19 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,

Tamara Sljivancanin Jakovljevic

Academic Editor

PLOS ONE

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Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. Is the manuscript technically sound, and do the data support the conclusions?

Reviewer #1: Partly

Reviewer #2: Yes

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2. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #2: No

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3. Have the authors made all data underlying the findings in their manuscript fully available?

Reviewer #1: No

Reviewer #2: Yes

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4. Is the manuscript presented in an intelligible fashion and written in standard English?

Reviewer #1: Yes

Reviewer #2: Yes

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5. Review Comments to the Author

Reviewer #1: Dear authors,

I have reviewed your manuscript reporting outcome of 100 twin homebirths. You have made a great effort presenting a significant number of twin home deliveries during 12 -year period. Your described management of home twin births in detail and presented results in seven figures and four tables with four supplement supporting information that included one video, two word documents and an excel table.

However, as you mentioned in your work, this is rather controversial topic, as it opposes standard protocols, especially ACOG. Therefore, many questions arise concerning mother and children safety during home birth, especially in women with previous Cesarean section (CS) and first baby breach. You must agree that those situations present risk for both mother and the baby even in hospital settings and home birth in those cases may be considered hazardous. I presume that you have informed your patients of all possible risks they are exposing and that you have an excellent insurance in the cases of potential adverse event and low suit.

You delivered both dichorionic and monochorionic-diamniotic twins. There is an ongoing debate about timing of delivery in monochorionic-diamniotic twins as potential stillbirth may occur in those cases. Monitoring those pregnancies demands a perinatology specialist and we may say that monochorionic-diamniotic pregnancy was uneventful only after delivery. How did you monitor monochorionic-diamniotic twins concerning TAPS (Twin Anemia Polycythemia Sequence) and did any of monochorionic-diamniotic twins have TAPS diagnosed during pregnancy or after delivery? How did you monitor those pregnancies after 38 weeks, as 37/38 weeks is the choice for delivery by ACOG protocols?

VBAC is an excellent choice for all uncomplicated pregnancies and deliveries, and should be conducted under intensive monitoring in hospital setting. Concerning patients with previous CS, what was their parity, i.e. did they have previous vaginal delivery as well, and was vaginal delivery prior or after CS? What were indications for previous CS?

All the patients were divided to primiparas and multiparas. It is questionable whether women with previous SC can be considered in the same way as women having their first delivery. I suggest you to make three groups, extracting women with VBAC in the separate group.

You mentioned that pregnancies were uncomplicated with eutrophic growth of the twins, but neonatal birth weight was from 1814g for the twin A and 1644g for the twin B, which is a low birth weight for term pregnancy even for twins.

As for the Apgar score, the lowest 1-minute Apgar was 3 for both twins, and 5-minute 7 for twin A, and 6 for twin B. How many neonates had low Apgar score, what was the reason and what resuscitation measures you performed in the home setting?

It is obvious that your patients opted not to have epidural anesthesia, but how did you managed the cases of retained placenta or excessive hemorrhage and the need of manual revision of uterine cavity? Did you perform it without anesthesia?

How did you monitor uterine integrity after VBAC?

What is average time needed for hospital transfer? Was there any problems concerning it?

Have you performed GBS screening?

In introduction, line 87, and in discussion you pointed that most transfers to the hospital ended in CS. In my opinion there is no need for to emphasize it in an almost negative way, since transfer had been made in risky situations in the cases when vaginal delivery was questionable.

There are many questions concerning home birth of a risk pregnancy and this topic is rather controversial. I encourage authors to answer the raised questions.

Reviewer #2: I would like to thank the authors for addressing this topic in obstetrics on a community-based level which is rarly done before. The results of the study will give a guide for management of such cases.

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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: Mohsen M A Abdelhafez

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Revision 1

Responses to Reviewer #1:

"However, as you mentioned in your work, this is rather controversial topic, as it opposes standard protocols, especially ACOG. Therefore, many questions arise concerning mother and children safety during home birth, especially in women with previous Cesarean section (CS) and first baby breach. You must agree that those situations present risk for both mother and the baby even in hospital settings and home birth in those cases may be considered hazardous. I presume that you have informed your patients of all possible risks they are exposing and that you have an excellent insurance in the cases of potential adverse event and low suit."

Authors’ response: Our paper demonstrates that VBAC with twins and vaginal birth of breech-first twins is a reasonable option, both in hospital settings (as per our reference list mentioned in the paper) as well as in a home setting. SJF’s patient counseling is extensive, as his model of care allows him the time to review all risks, benefits, and alternatives to all possible options. His clients are well-informed and choose to reject the obstetrical model and ACOG guidelines because those do not align with their values. The discussion of liability insurance is irrelevant to this presentation of clinical data outcomes and shows that this reviewer is not familiar with common home birth practices; many home birth attendants in the US forego liability insurance. (This comment above does not seem to ask for revision in the paper itself, so we have addressed the comments in this author response.)

"You delivered both dichorionic and monochorionic-diamniotic twins. There is an ongoing debate about timing of delivery in monochorionic-diamniotic twins as potential stillbirth may occur in those cases. Monitoring those pregnancies demands a perinatology specialist and we may say that monochorionic-diamniotic pregnancy was uneventful only after delivery. How did you monitor monochorionic-diamniotic twins concerning TAPS (Twin Anemia Polycythemia Sequence) and did any of monochorionic-diamniotic twins have TAPS diagnosed during pregnancy or after delivery? How did you monitor those pregnancies after 38 weeks, as 37/38 weeks is the choice for delivery by ACOG protocols?"

Authors’ response: Our paper is part of this ongoing debate, showing that routine induction at a set time point may not be necessary, especially for those who are motivated to avoid induction (as is the case with our population). We disagree that mono-di pregnancies “demand” a perinatology specialist. SJF consulted with a perinatologist when appropriate but not in every case of mono-di twins. In lines 144- 152, we explain the monitoring protocols for mono-di pregnancies. SJF did his own ultrasound scans, allowing him to screen and monitor his twin cases directly for possible anomalies and complications. None of the mono-di twins developed TAPS or signs associated with TAPS. SJF did not screen specifically for TAPS, as TAPS alone would not have been an indication to change his management. As mentioned in the paper, SJF offered twice-weekly fetal surveillance after 38 weeks, which clients were free to accept or decline. Again, the model of care in a home birth setting allows for individualized care and patient autonomy; most patient in his model are looking for something outside of ACOG protocols. Please refer to lines 282-287 and 451-482 for our discussion of these issues.

"VBAC is an excellent choice for all uncomplicated pregnancies and deliveries, and should be conducted under intensive monitoring in hospital setting. Concerning patients with previous CS, what was their parity, i.e. did they have previous vaginal delivery as well, and was vaginal delivery prior or after CS? What were indications for previous CS?"

Authors’ response: The statement that VBAC “should be conducted under intensive monitoring in hospital setting” is the reviewer’s belief and an ACOG recommendation, but again, we are examining a different style of care and a different approach to twins. Both SJF and the clients he serves desire an alternative approach to ACOG management; the paper demonstrates excellent outcomes without having to adhere to every ACOG guideline for twins. SJF did not take into account indications for previous C-sections as a limiting factor for who would plan a VBAC with twins, because there is no accurate way to predict the success of a VBAC based on indications for the previous surgery. Thus we have not included indications for prior C-sections in our data, as it was not clinically relevant to SJF’s care. In the Excel spreadsheet provided with the paper, you can see how many VBACs entering labor still under SJF’s care had prior vaginal births and (1) and how many were functional primips (no previous vaginal births, n=5). We have also added an additional supplemental file (S2) with more information about the VBAC twin births.

"All the patients were divided to primiparas and multiparas. It is questionable whether women with previous SC can be considered in the same way as women having their first delivery. I suggest you to make three groups, extracting women with VBAC in the separate group."

Authors’ response: To remain consistent with our prior publication of SJF’s breech & cephalic singleton data (Fischbein & Freeze 2018, cited in the paper), we would prefer to keep the two original groups. However, to address this reviewer’s concerns, we have included an additional addendum with more information about each VBAC (which is also available in the data spreadsheet attached with the paper). We feel that it is most appropriate to include functional primips (i.e., VBACs with no previous vaginal births) in the group with primips, while keeping VBACS with previous vaginal births in the multip group, as those two groups most closely align in terms of their risk factors and labor patterns.

"You mentioned that pregnancies were uncomplicated with eutrophic growth of the twins, but neonatal birth weight was from 1814g for the twin A and 1644g for the twin B, which is a low birth weight for term pregnancy even for twins."

Authors’ response: While small, these twins were both growing well on their respective growth curves. As mentioned in the paper, SJF’s selection criteria included having both twins growing consistently on their own ultrasound growth curves, which they were even for these two smaller babies.

"As for the Apgar score, the lowest 1-minute Apgar was 3 for both twins, and 5-minute 7 for twin A, and 6 for twin B. How many neonates had low Apgar score, what was the reason and what resuscitation measures you performed in the home setting?"

Authors’ response: 1-minute Apgar scores are generally not considered clinically relevant; most papers only publish 5-minute scores. We chose to mention both, but to respond to this reviewer’s question, there were two 5-minute Apgars of 6 for twin B and none for twin A. One twin B with a low Apgar score had Goldenhar’s syndrome. The other low Apgar for a Twin B was after fetal bradycardia, leading to an IPV & breech extraction. SJF’s team followed NRP resuscitation guidelines, leaving the cord intact during resuscitation. We added a description of the equipment carried as well as these details about the two cases of low Apgar scores.

"It is obvious that your patients opted not to have epidural anesthesia, but how did you managed the cases of retained placenta or excessive hemorrhage and the need of manual revision of uterine cavity? Did you perform it without anesthesia?"

Authors’ response: Yes, all of these situations were managed at home without anesthesia.

"How did you monitor uterine integrity after VBAC?"

Authors’ response: If the mother was stable, SJF managed VBACs like any other postpartum patient. No additional uterine monitoring or exam was considered necessary.

"What is average time needed for hospital transfer? Was there any problems concerning it?"

Authors’ response: We had no problems arise due to time of hospital transport among the two cases that transported via ambulance (maternal seizure postpartum and a laboring VBAC mother at 10 cm). The other case that transported (neonatal transport for persistent low 02 sats) was not urgent and transported via private vehicle. Los Angeles is a well-served urban area with ample access to EMS services and hospitals. On average, SJF’s clients lived under 30 minutes away from the nearest hospital and often much closer.

"Have you performed GBS screening?"

Authors’ response: GBS screening was offered but not required, as with all potential screenings and interventions in SJF’s model of care.

"In introduction, line 87, and in discussion you pointed that most transfers to the hospital ended in CS. In my opinion there is no need for to emphasize it in an almost negative way, since transfer had been made in risky situations in the cases when vaginal delivery was questionable."

Authors’ response: Since the reviewer was clear that this was a matter of personal opinion (“in my opinion”) and not an issue with the presentation of the clinical data, we are choosing to leave these parts as-is. We do not feel that including C-section rates after transport is biased or negative; rather, we are simply stating the outcomes of transports. We disagree that this data is presented in an “almost negative” way; it is simply presented as a matter of fact and percentage. Given that 2 of the 8 transports ended vaginally, and that more likely could have ended vaginally had vaginal birth been an option at the receiving hospital, whether the transports ended vaginally is relevant and important information. (In line 87, we were citing another article, not our own data.)

"There are many questions concerning home birth of a risk pregnancy and this topic is rather controversial. I encourage authors to answer the raised questions."

Responses to Reviewer #2:

"I would like to thank the authors for addressing this topic in obstetrics on a community-based level which is rarely done before. The results of the study will give a guide for management of such cases."

Authors’ response: Thank you

General comments from the authors:

Responding to one of the reviewer’s responses to the question: “Has the statistical analysis been performed appropriately and rigorously?”

• We added a note in the paper that the sample sizes were too small for most statistical analysis.

Responding to one of the reviewer’s responses to the question: “Have the authors made all data underlying the findings in their manuscript fully available?”

• One of the reviewers responded “no.” We have made all data fully available via the included Excel spreadsheet. We cannot, of course, include the original medical charts with the patient’s name and identifying information, but we excerpted all relevant anonymized data onto the spreadsheet.

Attachments
Attachment
Submitted filename: Response to Reviewers.docx
Decision Letter - Tamara Sljivancanin Jakovljevic, Editor

PONE-D-24-05781R1Twin home birth: Outcomes of 100 sets of twins in the care of a single practitionerPLOS ONE

Dear Dr. Freeze,

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 Dec 08 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,

Tamara Sljivancanin Jakovljevic

Academic Editor

PLOS ONE

Journal Requirements:

Please review your reference list to ensure that it is complete and correct. If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references. Any changes to the reference list should be mentioned in the rebuttal letter that accompanies your revised manuscript. If you need to cite a retracted article, indicate the article’s retracted status in the References list and also include a citation and full reference for the retraction notice.

[Note: HTML markup is below. Please do not edit.]

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 #3: (No Response)

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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 #3: (No Response)

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3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #3: (No Response)

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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 #3: (No Response)

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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 #3: Yes

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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 #3: ABSTRACT: The statement in lines 23-25 should be recasted and grammar corrected.

METHODS: How was the data recorded?

RESULTS: Under induction vs spontaneous labour, age 283-- how were the women whose pregnancies had passed their expected date of delivery manged?

Lines 301-302- for a statement of this nature, figures showing how significance was determined should be included.

DISCUSSION: In lines 316-317, was the 3rd degree laceration which was repaired at home repaired under anaesthesia in the house if so what type? In lies 368-369, the statement is not very clear, based on the results of their study which of the birth or indications of the instrumental vaginal deliveries are the authors referring to? Similarly, in lines 370-371, the authors should state for clarity which of the situations they would have managed otherwise and why they did not do so. The statement in lines 555-556 with quoted figures ideally should be referenced,

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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 #3: No

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[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.

Revision 2

Reviewer #3:

ABSTRACT: The statement in lines 23-25 should be recasted and grammar corrected.

• We corrected the grammar in these lines.

METHODS: How was the data recorded?

• We added further clarification on page 8, lines 217-218 to note that SJF excerpted the relevant information from the women’s medical records into an Excel spreadsheet.

RESULTS: Under induction vs spontaneous labour, age 283-- how were the women whose pregnancies had passed their expected date of delivery manged?

• As noted in the paragraph under “induction vs. spontaneous labor”, SJF offered a different standard of care and did not mandate delivery by 37 or 38 weeks. Instead, after counseling and informed consent discussions, the normal practice was to await labor. We added another sentence in this section clarifying SJF’s normal practices for clients at or past 40 weeks.

Lines 301-302- for a statement of this nature, figures showing how significance was determined should be included.

• The numbers were too small to calculate p-values; we have decided to omit this sentence to avoid confusion.

DISCUSSION: In lines 316-317, was the 3rd degree laceration which was repaired at home repaired under anaesthesia in the house? If so what type?

• It was repaired with local anesthesia (lidocaine)

In lines 368-369, the statement is not very clear, based on the results of their study which of the birth or indications of the instrumental vaginal deliveries are the authors referring to? Similarly, in lines 370-371, the authors should state for clarity which of the situations they would have managed otherwise and why they did not do so.

• We added some clarification in the paper, noting that SJF’s use of instrumental delivery is related to his experience and skillset. Each case requires an individualized judgment call and is situational (mother’s exhaustion levels, anxiety in the room, etc.). It’s impossible to predict the outcomes of each specific birth with a different care provider who could not use instrumental delivery. We wish to stress that we cannot point to each individual birth and predict the specific outcomes had a midwife been present versus an obstetrician. However, SJF feels that, in general, a midwife may been able to resolve some situations in other ways and may have transported other situations earlier.

The statement in lines 555-556 with quoted figures ideally should be referenced,

• If you are referring to Cynthia Caillagh’s data, it is currently unpublished and RF had personal correspondence with her prior to her death. There is not a formal reference available as her data is not yet published. We added a parenthetical citation to clarify this.

Attachments
Attachment
Submitted filename: Point by Point response Oct 29.docx
Decision Letter - Tamara Sljivancanin Jakovljevic, Editor

Twin home birth: Outcomes of 100 sets of twins in the care of a single practitioner

PONE-D-24-05781R2

Dear Dr. Rixa Freeze,

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.

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Kind regards,

Tamara Sljivancanin Jakovljevic

Academic Editor

PLOS ONE

Formally Accepted
Acceptance Letter - Tamara Sljivancanin Jakovljevic, Editor

PONE-D-24-05781R2

PLOS ONE

Dear Dr. Freeze,

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.

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on behalf of

Dr. Tamara Sljivancanin Jakovljevic

Academic Editor

PLOS ONE

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