PONE-D-20-29192

Correlation between Hemophilia Early Arthropathy Detection with Ultrasound (HEAD-US) Score and Hemophilia Joint Health Score (HJHS) in Patients with Hemophilic Arthropathy

PLOS ONE

Dear Dr. Prasetyo,

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.

Both reviewers felt that this paper showed some merit although it would benefit from minor corrections. A concern was that the population sampled (adolescents) may not be the highest risk for hemarthropathy and the care described (episodic rather than prophylactic factor replacement) no longer represent standard of care. In addition, the minor points raised regarding the use of portable ultrasonography should be considered.

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Academic Editor

PLOS ONE

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Additional Editor Comments:

Both reviewers felt that this paper showed some merit although it would benefit from minor corrections. A concern was that the population sampled (adolescents) and the care described (episodic rather than prophylactic factor replacement) no longer represent standard of care. In addition, the minor points raised regarding the use of portable ultrasonography should be considered.

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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: Yes

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

Reviewer #1: No

Reviewer #2: 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

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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

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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: Correlation between Hemophilia Early Arthroplasty Detection with Ultrasound (HEADUS) Score and Hemophilia Joint Health Score (HJHS) in Patients with Hemophilic Arthropathy

In this single-centre, cross-sectional study, the authors evaluated the correlation between the HJHS and HEAD-US in twenty pediatric and adolescent patients with severe hemophilia using already established protocols. A trained physiotherapist evaluated only elbows, knees and ankles. The ultrasound and HJHS were done the same day of the visit and showed a correlation of 0.65.

Although this work has already been performed elsewhere, validation of this correlation in the setting of the authors geographic location may add value to the existing body of knowledge. This report could be improved by addressing the following queries.

1. The introduction should include the rationale for doing this correlation assessment in patients receiving episodic treatment when it is not the standard of care in hemophilia in 2020 anymore. How is this knowledge going to be translated into clinical practice?

2. The study population is adolescent and pediatric patients who often have minimum arthropathy. Is there a rationale for doing the study in this group only and not in adults?

3. The researchers should include the standard shortcomings of ultrasound examination in the introduction or discussion as the study shortcomings.

4. Given that the ultrasound was done in a hospital setting and the expanding role of portable ultrasound, the authors should point out the role of portable ultrasound in their setting.

5. In the methods, it is unclear how the physiotherapists/ rehabilitation specialists were trained and what their intra-individual and inter-individual coefficient of variation was.

6. The discussion should include the strengths and weaknesses of the study.

7. In the abstract, reference is made to joint bleed and hemarthrosis, which mean the same thing.

8. The reference page numbers should be reviewed for consistency, some have single digit and others have two digit last page numbers.

9. Can the authors comment on their statement that a correlation of 0.65 is regarded as a strong correlation in hemophilia?

Reviewer #2: It is my opinion that the authors have demonstared a relatively easy reproducible way of assesng joint involvement in Hemophilia and the paper should be published

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Reviewer #1: Yes: Johnny Mahlangu

Reviewer #2: Yes: BF Jacobson

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Correlation between Hemophilia Early Arthropathy Detection with Ultrasound (HEAD-US) Score and Hemophilia Joint Health Score (HJHS) in Patients with Hemophilic Arthropathy

PONE-D-20-29192R1

Dear Dr. Prasetyo,

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,

Elizabeth S. Mayne, M.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

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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

**********

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

Reviewer #1: 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

**********

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

**********

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)

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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: Yes: Johnny Mahlangu