PONE-D-23-37777Hidden stories of caregivers with children living with Sickle Cell Disease: experiences, coping strategies and outcomesPLOS ONE

Dear Dr. ALINDA,

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.

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We look forward to receiving your revised manuscript.

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Deogratias Munube

Academic Editor

PLOS ONE

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Additional Editor Comments:

Dear Author,

A decision of a major revision has been made of your manuscript.

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Reviewers' comments:

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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

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

Reviewer #1: N/A

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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

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

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Reviewer #1: Yes

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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: While I believe the manuscript is technically sound, some of the methods could benefit from more informed rationales.

Although the manuscript has been presented in an intelligible fashion and written in standard English, it will benefit from proofreading, which is highly recommended.

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Reviewer #1: No

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Attachments

Attachment

Submitted filename: EAB Comments PONE-D-23-37777.docx

PONE-D-23-37777R1Hidden stories of caregivers with children living with Sickle Cell Disease in Uganda: experiences, coping strategies and outcomesPLOS ONE

Dear Dr. ALINDA,

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 12 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,

Aloysius Gonzaga Mubuuke

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.

Additional Editor Comments:

None

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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: (No Response)

Reviewer #2: (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 #1: Yes

Reviewer #2: Yes

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

Reviewer #1: Yes

Reviewer #2: N/A

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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 #1: Yes

Reviewer #2: Yes

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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 #1: Yes

Reviewer #2: No

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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 #1: I am pleased that the authors have made some changes and/or provided further explanation to some sections of the manuscript. These have made the manuscript more insightful. There are a few more issues that need further attention, though.

1. While the authors have provided some background information on Uganda, they should give some specific details. For instance, how much does the treatment cost? What is the average income of families in Uganda?

Providing this information would aid readers in appreciating the financial challenges families face.

2. Member checking is not a required step for reflexive thematic analysis, and certainly, it is not done to ensure data saturation. Please, revise that statement in the methods section.

3. What is the rationale for summarizing the table showing the characteristics of the participants. There was no comment against the previous version. A reversion to the earlier format is recommended.

4. The authors maintain that reviewing extant literature is helpful to the interpretation of their data. I would be happy to have some citations that support this exercise.

5. I do not see ‘gender imbalance’ as a limitation – many of the caregivers are generally females so what you have is a true reflection of the population. In other words, you don’t have to state fewer male participation as a limitation.

6. On page 29, the main author has been described as “DM”. What does that mean?

Reviewer #2: Thanks to authors, they have attended to most of comments previously raised.

However, I feel the responses to the following could be improved.

Introduction

3. Taking care of a child with sickle cell disease can be expensive, and many families in Uganda may find it difficult to pay for the required therapies and drugs.” It will be more appropriate to provide contextual information on SCD and its management in Uganda in the ‘study setting’ section. The authors need to describe for instance what costs are incurred given that Mulago national referral hospital is a public facility.

Methods

7. How was the accuracy of data translation from Luganda to English ensured? The authors response “Consultation with Participants: Where necessary, the translated transcripts were reviewed with participants to ensure that the translated content accurately represented their original statements and meanings” is not clear to me. How this was practical given that participants were interviewed at the hospital while they had come to attend the clinic. Provide more details.

Results

1. I do not see the relevance of (n=12), (n=2) in the manuscript. Delete this style of reporting. Although, the authors had accepted the suggestion and deleted some, it was not implemented through the whole section. Please delete this style of reporting throughout the manuscript

3. I do not understand the sub-theme ‘coping strategies at school’. Such a phrase will be more suitable for the children’s experiences, rather than the experiences of caregivers. The authors’ responses are not conceiving. The issues covered here must directly be related to the study topic. I had for instance expected to find out how caregivers ensure that their children continue to take their treatment while at school, how do they ensure that medications are kept safely, get special permission to avoid stressful conditions, how they ensure that their children are able to attend clinic appointment dates during school term, any special diet arrangements at school etc Which is in line with quote from participant 4. The school fees challenges are cross cutting to most parents and is not specific to SCD caregivers!

Discussion

2. ‘Hidden’ or ‘concealed’ are not appropriate terms to use to describe the experiences of caregivers – consider using ‘untold’. The authors partially accepted the feedback. I re-echo the same. The authors should use consider using ‘untold’ instead of ‘hidden’ throughout the manuscript including the title.

Authors’ contributions

Reading authors’ contributions and acknowledgement section together leaves out confused. Both LK and HS are acknowledged for their input in this manuscript. HS was a research assistant and LK guided during the data collection and analysis process. How do they then turn around to be authors? Who si the hidden main author DM? Surprisingly, the contribution of IA is not shown.

Proof reading

The work will benefit from major proofreading as the errors are quite too many.

There are still many errors especially in the participants’ quotes. Actually, most of them are difficult to understand. E.g

".. I want him to go back [to school], because he is too worried, he says that the whole year, and yet in the future I will have like 2 years off…"

Participant 4, “…. things that helped me a lot after they had told me and I got to know that he is having sickle cell disease, the first thing I got to do was, I asked the Almighty to forgive me, since I decided it for myself, that I will never produce again, and by then I was pregnant preparing for the second baby, and then I said to myself that I would not produce a 2nd child because for the 1st child made me spend most of the times in the hospital while bed ridden

The participant was pregnant and is being quoted saying she will never ‘produce’ again. What happened or what plan did she have for the pregnancy?

This long sentence is very hard to comprehend: “Similarly in another study Social support played a crucial role in helping caregivers cope, but negative reactions from certain individuals highlighted the need for increased awareness and destigmatization efforts in the community in this study, In addition to describing the networking and support that caregivers sought to engage in for the purpose of support and information, the Lived Experience of Caregivers of Children with Sickle Cell Disease study also highlighted numerous parenting challenges that caregivers face while caring for their sickle cell disease children (20)”

Finally, under conclusion, authors refer to Mulago hospital’s role as a primary health care facility for SCD. However, in the previous sections, they had referred to Mulago hospital as a national referral hospital. The authors need to categorically state whether patients were attending Mulago SCD clinic as their primary care clinic or as a referral point.

Thank you

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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 #1: No

Reviewer #2: Yes:  Vincent Mubangizi

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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.

Attachments

Attachment

Submitted filename: EAB Comments PONE-D-23-37777 R1.docx

Hidden stories of caregivers with children living with Sickle Cell Disease in Uganda: experiences, coping strategies and outcomes

PONE-D-23-37777R2

Dear Dr. ALINDA,

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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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,

Aloysius Gonzaga Mubuuke

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

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