PONE-D-23-29643A COST ANALYSIS OF MACHADO-JOSEPH'S DISEASE (MJD)PLOS ONE

Dear Dr. Silva,

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.

==============================Thank you for your article submission to PlosONE. We apologize for the delay in our response. Due to a reviewer becoming unresponsive, we needed to reassess the manuscript. Your article has been reviewed by three individuals; however, except for reviewer 1, the others have opted to remain anonymous for the study.

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Asif Ali

Academic Editor

PLOS ONE

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Additional Editor Comments:

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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?

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: Partly

Reviewer #3: Yes

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

Reviewer #1: I Don't Know

Reviewer #2: No

Reviewer #3: Yes

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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

Reviewer #2: Yes

Reviewer #3: Yes

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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

Reviewer #3: 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: I don't believe this to be a rigorous research endeavor. No valuable insights are provided. I am not sure if this rudimentary economic analysis of MACHADO-JOSEPH'S DISEASE (MJD) makes it fit for the publication.

Reviewer #2: The authors intent to calculate the direct and indirect health-related expenses linked to lost productivity caused by Machado-Joseph’s Disease (MJD). This study may serve to amplify voices urging authorities to provide increased supportive care for families affected by rare diseases such as MJD. The authors must revise the manuscript in accordance with the identified revision points below:

1. Please avoid any citation in the abstract section, therefore, please move citation 1 to the introduction, if necessary

2. "Please incorporate more formal language into your phrasing, for example, avoid phrases like 'one can think that' or 'which is good news for the hospital'.

3. Please indicate how many caregivers and in- or out-patients are involved in the statistics.

4. In your statistical analysis, you should add the exact p value you found.

5. The first paragraph of the results section should move to the material and methods, and avoid to use future tense in the materials and methods.

6. Indicate your standard deviation or standard error as well as p value in the Graph 1 and explain it properly in the figure legend.

7. All figure legends should be moved beneath their respective tables or graphs. Each legend must include a concise explanation of the figure, including details such as statistical analysis and sample size.

Reviewer #3: The manuscript " A COST ANALYSIS OF MACHADO-JOSEPH'S DISEASE" by Silva et al. is a good analysis about MACHADO-JOSEPH'S DISEASE from cost perspective. This manuscript is of interest to the clinical studies, human biology researchers, as well as different biology researchers and I expect that the article will be well-cited. I have only one comment to consider. Authors add method and statistical details in their figures and tables legend and statistical section in materials and methods.

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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.

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Reviewer #1: No

Reviewer #2: Yes: Sera Averbek

Reviewer #3: Yes: Pawan Kumar

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A cost analysis of Machado-Joseph's disease (MJD)

PONE-D-23-29643R1

Dear Dr. Silva,

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,

Asif Ali

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

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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 #2: Yes

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

Reviewer #2: Yes

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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 #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 #2: 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 #2: (No Response)

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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 #2: Yes: Sera Averbek

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