PONE-D-24-36104Integrating post-validation surveillance of lymphatic filariasis with a non-communicable disease program in Niue, a study protocolPLOS ONE

Dear Dr. Craig,

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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ACADEMIC EDITOR: I have invited four experts to review the protocol. Of them, three reviewers agreed to our request. All the reviewers considered this an important study in the context of LF post validation surveillance, but also raised a number of constructive comments. I would request the authors to concentrate and address the issues related to study methodology (hypotheses, sampling, etc) and Discussion.

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Please submit your revised manuscript by Nov 21 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,

Swaminathan Subramanian, Ph.D.

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 2. Please amend your list of authors on the manuscript to ensure that each author is linked to an affiliation. Authors’ affiliations should reflect the institution where the work was done (if authors moved subsequently, you can also list the new affiliation stating “current affiliation:….” as necessary). 3. Please include a caption for figure 3a 4. Please include a caption for table 2. 5. Please include a complete copy of PLOS’ questionnaire on inclusivity in global research in your revised manuscript. Our policy for research in this area aims to improve transparency in the reporting of research performed outside of researchers’ own country or community. The policy applies to researchers who have travelled to a different country to conduct research, research with Indigenous populations or their lands, and research on cultural artefacts. The questionnaire can also be requested at the journal’s discretion for any other submissions, even if these conditions are not met.  Please find more information on the policy and a link to download a blank copy of the questionnaire here: https://journals.plos.org/plosone/s/best-practices-in-research-reporting. Please upload a completed version of your questionnaire as Supporting Information when you resubmit your manuscript. 6. Thank you for stating the following financial disclosure:  [This work is funded by the Global Institute for Disease Elimination (FADE2024/03), the (Australian) National Centre for Immunisation Research and Surveillance (NCIRS) and Operational Research and Decision Support for Infectious Diseases Program at the Centre for Clinical Research, the University of Queensland (UQ). Staff from UQ led the study design, the decision to publish and the reparation of the manuscript.].  Please state what role the funders took in the study.  If the funders had no role, please state: ""The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript."" If this statement is not correct you must amend it as needed. Please include this amended Role of Funder statement in your cover letter; we will change the online submission form on your behalf.

7. 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.

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Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. Does the manuscript provide a valid rationale for the proposed study, with clearly identified and justified research questions?

The research question outlined is expected to address a valid academic problem or topic and contribute to the base of knowledge in the field.

Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

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2. Is the protocol technically sound and planned in a manner that will lead to a meaningful outcome and allow testing the stated hypotheses?

The manuscript should describe the methods in sufficient detail to prevent undisclosed flexibility in the experimental procedure or analysis pipeline, including sufficient outcome-neutral conditions (e.g. necessary controls, absence of floor or ceiling effects) to test the proposed hypotheses and a statistical power analysis where applicable. As there may be aspects of the methodology and analysis which can only be refined once the work is undertaken, authors should outline potential assumptions and explicitly describe what aspects of the proposed analyses, if any, are exploratory.

Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

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3. Is the methodology feasible and described in sufficient detail to allow the work to be replicable?

Descriptions of methods and materials in the protocol should be reported in sufficient detail for another researcher to reproduce all experiments and analyses. The protocol should describe the appropriate controls, sample size calculations, and replication needed to ensure that the data are robust and reproducible.

Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

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4. Have the authors described where all data underlying the findings will be made available when the study is complete?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception, at the time of publication. 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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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

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 and, if applicable, provide comments about issues authors must address before this protocol can be accepted for publication. You may also include additional comments for the author, including concerns about research or publication ethics.

You may also provide optional suggestions and comments to authors that they might find helpful in planning their study.

(Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: This is a very clearly written study protocol. The proposed research is appropriate for the Niue context (which is highly unique) and should lead to information that benefits the national LF elimination effort - hopefully confirming that elimination status is maintained.

I would request that the authors address the following minor issues:

Study hypotheses do not appear to be unique. Particularly hypotheses 1 & 3. I suggest removing hypothesis 3, it feels like a subset of hypothesis 1.

Line 129 has a pair of errant words

Line 150: were should be "where"

Line 155: please state the age group sampled in the 'C survey'

Line 164: missing a space in the date

Line 260: I suggest you use "the house" and not "a house"; I would imagine most people slept in a house (unless you really do mean to distinguish between people sleeping in doors vs. outdoors)

Can you add a little more commentary in the discussion around the applicability of integrating with STEPS surveys? Are they always census surveys in the Pacific? To what other countries would you anticipate this methodology would extend?

Several times you mention priority diseases. Please share this list of diseases for Niue to give the reader an idea of what will be assessed, and possibly inspire readers with the types of diseases that can be assessed in an integrated manner.

Reviewer #2: The resurgence of LF following validation of elimination can happen, although it may take time. The authors for this study describe a protocol aimed at LF PVS through integration with other activities. Addressing the following comments will strengthen the paper.

Line 76. Ae. Cooki. cooki should be with a small "c".

Line 113: correct the spelling of ESRI ArcGIS

Line 125: Change remined to remained.

Lines 105/128/133/139/149: Check the population number. In the abstract and in line 105, a population of 1600 is reported. In line 128, 4,408 people were tested in 1972. Has the population declined? What accounts for that?

Line 129: Remove "nor" after whether.

Line 196: Correct the spelling of "Fourth"

- Why has the sampling population been restricted to those aged 15 - 69 years? A clarification for this will be useful.

- A description of the STEPS survey and how the LF PVS fits in will be useful.

- Are there other opportunities for integration that could be explored beyond the STEP survey

- Portions of the text described in the "Field Logistics" section should be moved to the "Informed Consent" section. There is some repetitions e.g. consenting procedures.

- Knowing that molecular xenomonioting results in higher infection prevalence in mosquitoes compared to humans, is there any consideration for this in the PVS surveys?

- It would be useful to provide copies of the tools used (e.g. questionnaires) as supplementary files to guide the reproducibility of the work by others.

- Some potential benefits of integration were listed. It might also be helpful for the authors to describe any challenges as part of the integration of the LF survey and STEP, even at the planning stage.

Reviewer #3: Comments on the paper entitled “Integrating post-validation surveillance of lymphatic filariasis with a non-communicable disease program in Niue, a study protocol”

PLOS ONE accepts submissions for publication of Study Protocols for any study type within the journal’s scope. This study Protocols describe detailed plans for conducting research, including the background, rationale, objectives, methodology, statistical plan, and organization of a research project. Publication of this protocol can be considered as the protocol plans to integrate and generate data on various indicators and interpretation of the results of LF-PVS.

Niue with a population of about 1600 people and antigenemia based mapping was done in 1999, repeat antigenemia survey in 2001, 2004 and 2009. The surveys covering population aged 2 years and above showed a decline trend in antigenemia prevalence from 3.1 to 0.5. MDA for LF elimination was carried out from 2000-2004. WHO validation was done in 2016. The country has a long history of LF control/elimination, since 1954 and monitoring and evaluation by conducting whole population survey. Therefore it is pertinent to trial with the new study protocol of PVS. The protocol is well written and presented. However, the following are the minor comments:

Comments

Abstract

1. Consider revising the title, specifying the disease for integration with LF. It can be “Integrating post-validation surveillance of lymphatic filariasis with a standard STEPs survey in Niue, a study protocol”

2. Line 24-29: Can be deleted. Start with LF situation in Niue.

3. Line 32-34: Rephrase the sentence as “However, no surveillance was conducted ever since validation to confirm infection rates have remained below WHO’s elimination threshold”.

4. Line 42: survey can be deleted

5. Line 47: Consider replacing “for ongoing LF transmission” with “for LF”. Prevalence of antigenemia not necessary indicate the ongoing transmission. It indicates both past and recent exposure.

6. Line 49: Justify antibody assay when we collect data on antigen and Mf. It may not reflect the ongoing transmission.

Background and study rationale

7. Line 64:what is the parasite species other than Wuchereria bancrofti prevalent in South Pacific countries? W.bancrofti is probably the only LF parasite prevalent in Niue [WHO. The PacELF way: towards the elimination of lymphatic filariasis from the Pacific, 1999-2005. Manila: WHO Western Pacific Region; 2006].

8. Line 73: this article “Zeldenryk, L.M., Gray, M., Speare, R., Gordon, S., Melrose, W., 2011. The Emerging Story of Disability Associated with Lymphatic Filariasis: A Critical Review. PLOS Neglected Tropical Diseases 5, e1366.. https://doi.org/10.1371/journal.pntd.0001366” can be cited as reference for LF is the second leading cause of chronic disability.

9. Line 76: Correct Ae. Cooki as Ae. cooki

10. Line 116: cite the reference of Carlingford [7]

11. Line 128-130: Suggest to rephrase the sentence “The survey was reported to have included 98.4% of a population, although nor whether this was for the entire population or a subset (e.g., those above a certain age) is unclear.”

12. Line 140-142: Give prevalence data, if available instead of number of cases. [A relatively high number (n=13) of Ag-positive individuals were found in two locations (the villages of Tamakautonga and Hikutavake) that were distant to each other (i.e., not bordering each othe].

13. Line 150: replace “were” by “where”

14. Line 156-157: add “in individuals older than 2 years” before [23]

15. Line 159-160: rephrase the sentence as “The seven individuals who tested positive in 2009 were re-tested for Ag and treated the positives until testing Ag-negative”.

Methods

16. Line 216: add “in all 14 villages” after diseases

17. Line 228: a column “total population” can be added before “Target sample size”. Add “in the age class 15-69 years after target sample size”

18. Line 231-232: Migrants can also be excluded

Study implementation

19. Line 241: the target sample size for whole population (excluding below 15 years of age) LF survey can be mentioned.

20. Line 245 & 258: Consider providing a supplement table for “A standard STEPs survey questionnaire”

21. Line 253: preferably witness from either the community or health staff and witness by a survey team member can be avoided

22. Line 254: Assent form for individuals 15-17 years may be required

23. Line 263: Whether GPS of the household will be recorded?

24. Line 270: add “for LF survey” after blood collection tubes

25. Line 285: will the DBS samples be sufficient to carry out a panel of tests of other NTDs, VPDs, and VBDs?

Discussion

Discussion brings out possible outcome of the study. However, discussion should focus on the timeline and how long these LF-PVS surveys if demonstrated feasible should be carried out. What changes are required when it is translated into a larger population? Discussion may also indicate the frequency and duration of these surveys and interpretation of the results. The implications of showing Ag/Ab/Mf positives and any possible thresholds of each indicator may be discussed.

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If you choose “no”, your identity will remain anonymous but your review may still be made public.

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Reviewer #1: No

Reviewer #2: Yes: Dziedzom K de Souza

Reviewer #3: Yes: Krishnamoorthy

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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.]

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PONE-D-24-36104R1Integrating post-validation surveillance of lymphatic filariasis with the WHO STEPwise approach to non-communicable disease risk factor surveillance in Niue, a study protocolPLOS ONE

Dear Dr. Craig,

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 have agreed with most of the responses to their comments except the one of the comments of reviewer #2. As you will note from reviewer #2's comments, they are requesting that the questionnaires being used in the study are provided with the protocol at this point rather than with any subsequent research article for transparency purposes. From the IRB-approved protocol, there appears to be (i) a standard STEPs household questionnaire and (ii) the addition of one question “To which countries have you travelled in the last 5 years?”, which is being added to this. In view of this, please could you provide a copy of the STEPs household questionnaire or a link to the questionnaire, and add into the text details of the additional question, along the lines of  the 'Survey-based data collection' section of the IRB-approved protocol."

Please submit your revised manuscript by Jan 03 2025 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,

Swaminathan Subramanian, Ph.D.

Academic Editor

PLOS ONE

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Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. Does the manuscript provide a valid rationale for the proposed study, with clearly identified and justified research questions?

The research question outlined is expected to address a valid academic problem or topic and contribute to the base of knowledge in the field.

Reviewer #2: Yes

Reviewer #3: Yes

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2. Is the protocol technically sound and planned in a manner that will lead to a meaningful outcome and allow testing the stated hypotheses?

The manuscript should describe the methods in sufficient detail to prevent undisclosed flexibility in the experimental procedure or analysis pipeline, including sufficient outcome-neutral conditions (e.g. necessary controls, absence of floor or ceiling effects) to test the proposed hypotheses and a statistical power analysis where applicable. As there may be aspects of the methodology and analysis which can only be refined once the work is undertaken, authors should outline potential assumptions and explicitly describe what aspects of the proposed analyses, if any, are exploratory.

Reviewer #2: Yes

Reviewer #3: Yes

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3. Is the methodology feasible and described in sufficient detail to allow the work to be replicable?

Descriptions of methods and materials in the protocol should be reported in sufficient detail for another researcher to reproduce all experiments and analyses. The protocol should describe the appropriate controls, sample size calculations, and replication needed to ensure that the data are robust and reproducible.

Reviewer #2: No

Reviewer #3: Yes

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4. Have the authors described where all data underlying the findings will be made available when the study is complete?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception, at the time of publication. 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: No

Reviewer #3: 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

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 and, if applicable, provide comments about issues authors must address before this protocol can be accepted for publication. You may also include additional comments for the author, including concerns about research or publication ethics.

You may also provide optional suggestions and comments to authors that they might find helpful in planning their study.

(Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #2: I thank the authors for addressing my comments. However, on the comment of providing the survey tools, the authors' response is that these tools are now being drafted and will be made available as part of the results manuscripts.

If this is the case, then the submission should be placed on hold until the tools which are an integral part of the protocol are ready and submitted alongside. The principal reason for insisting on the tools is that there is no guarantee that the results manuscript will be submitted for publication or submitted to this journal. Also, the readers looking at this current submission need to be able to access all the tools without having to wait for a couple of years for the results manuscript nor look for it in another publication. Thus, there is no point submitting an incomplete study protocol for publication.

Reviewer #3: Reviewed the revised manuscript and the response to the comments (Reviewer 3). The revison is satisfactory. Except a comment, all the other comments have been accpeted by the authors and revised the manuscript accordingly. It is recommended that the manuscript in the present form may be accepted for publication.

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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: No

Reviewer #3: Yes: Kaliannagounder Krishnamoorthy

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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.

Integrating post-validation surveillance of lymphatic filariasis with the WHO STEPwise approach to non-communicable disease risk factor surveillance in Niue, a study protocol

PONE-D-24-36104R2

Dear Dr. Craig,

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,

Swaminathan Subramanian, Ph.D.

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Reviewers' comments: