PONE-D-21-22428

Applying Citizen Science to engage families affected by ovarian cancer in developing genetic service outreach strategies

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?

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: Yes

Reviewer #2: I Don't Know

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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: The authors aim to evaluate the feasibility of a Citizen Science quality framework, recruiting survivors of ovarian cancer and their close relatives, suggesting that CS engagement is fesible. The manuscript is well written but requries some minor revisions in order to be more comprehensive. Please find my comments as follows:

Introduction

Page 3 line 69, there is no reference for the sentence

“In the initial 63 planning year of a four-year NCI-funded study entitled “Your Family Connects” we applied citizen 64 science to generate intervention content for a message-based outreach intervention to encourage 65 families affected by ovarian cancer to consider genetic service options.”

Page 4, line 75. There is no reference for the sentence “Nunn and colleagues concluded that genomics- and 75 related translation researchers could benefit from standardized methods to apply PPI”. This regards the ref 16, which should be reported also in this sentence, otherwise it seems that is the continuum of ref 17.

Page 4, line 77-78, the sentence “Up to 20% of ovarian 78 cancers are linked to inherited genetic mutations in cancer predisposing genes”, should not be underlined.

Page 4, line 89 It is not clear the transition to concerns, i would suggest to add a sentence on the general concerns before describing each of them in details.

Page 5, line 107-108, i would suggest to include Table 1 in the Methods section and leave in the introduction the reference.

The introduction is quite long and should be shortened, with a focus on Citizens sceicne use in cancer and genetic outreach sterategies

Methods

Page 7, line 121, the authors should add the Protocol Approval Number from the Echics Committe

Page 8, line 137, Table 2 contains the results of data compelteness and should be included only in the results.

There is not a description of data statistical analysis process, eg using mean and SD

Results

In the academic writing, number less than 10 are suggested to be written in letters. The authors might conider writing these number is letters.

Discussion

The authors did not report an strenghts and limitation of their study.

The authors should describe the practical implication of their study.

Reviewer #2: Summary

The was an interesting paper, thank you for the chance to review it. Some small pick ups on consistent terminology will help improve this excellent paper.

Major issues: No major issues

Minor issues: The following points are mainly around consistency language, which if improved, will improve the readability of this paper.

Abstract:

‘nonprofessionals’ – should read non-professional researchers – as they may be professionals in other fields?

Data was/were line 38/40 – consistent?

Line 31 – 40 was complex and could be summarised more clearly or succinctly, for example, adding a heading ‘method’ and ‘results’.

Intro:

Line 54: the word citizen appears here for the first time and hasn’t been defined previously. Suggest clarification of terminology in first paragraph when defining patient and public involvement.

59: PPI collaboration or just PPI, if introducing the term collaboration does this need to be defined? Same with the word engagement.

61: ‘higher order citizen engagement’ – again, does this mean PPI? Consider using a task based approach rather than levels to describe involvement – as in ‘involvement in research design’ (high level) or just participation in survey responses (low level). A lot of this terminology will be unfamiliar to some readers so it’s important to be consistent in the terminology you are using.

63: ‘applied citizen science’ I would contest that you can ‘apply’ citizen science as a method, you could say ‘we involved people using methods informed by citizen science’ or words to that effect, if you see the distinction. At the very least you could say ‘we applied citizen science methodology’ but you’d need to carefully define what exactly you mean by that before using the shorthand later.

66: ‘Citizen science (CS) approaches involve nonprofessionals (citizens)’ suggest rewording as ‘Citizen science (CS) methods [metholodiges/paradigms?] involve non-professional researchers (citizens)

67: shouldn’t you add ‘collect data’ here too, as that is what happened with this project?

72: again, consider the word ‘approaches’ and defining it sooner. Methods, modes and tasks had very clear definitions in the review you are citing and I think this paper would be improved if terms like ‘citizen science approaches’ or ‘citizen science methods’ we used more consistently. I acknowledge this a wider issue of defining woolly terms such as CS (see my later comment about STARDIT), and you could consider using ‘participatory action research’ as an alternative term to CS.

75: regarding the line ‘Nunn and colleagues concluded that genomics- and related translation researchers could benefit from standardized methods to apply PPI’ you might be interested to learn about the project Standardised Data on Initiatives (STARDIT) which followed based on the recommendations of this scoping review and is now in a Beta stage of development. It allows stanardised reporting of involvement and the impacts of involvement (among much else). Pre-print here: https://doi.org/10.31219/osf.io/w5xj6 - and a friendly invitation to all authors to get involved with the STARDIT project if it’s of interest

82: do not advise the use of the word racial in the line ‘racial/ethnic minorities’, as race is generally considered a social construction. Ethnic acceptable as a short hand (if defined - .e.g is it cultural or are you specifically referring to inherited characteristics – e.g. groupings of people more pre-disposed to certain cancers with more awareness in those associated communities?) but perhaps also noting language communities and other cultural influences which might affect uptake. The point is you are grouping people by race in this comment, and people may not self-identify with such groupings. I note the papers you cite use this term, but in a health context I’d avoid it unless you are specifically talking about people’s lived experience of racism, which is distinct from reinforcing socially constructed racial categories. This article explains better what I’m trying to say: https://en.wikipedia.org/wiki/Race_(human_categorization)

85: This is a fantastic point and I note you’re now also introducing ‘co-create’. Again I suggest you chose a term at the start, mention all the different things it can be called, and then use that consistently as co-creation as a method might also need more explanation?

92: ‘Survivors’ is this a term that was co-defined by the people you involved, do they self-identify with that? Perhaps ‘people affected by cancer….’ Which also then includes loved-ones/carers of people with cancer etc

94: ‘Additionally, the credibility of researchers who share their passion for expanding reach could engage CS members more deeply in seeing the value of research.’ I think I know what you’re trying to say here, and I think it’s a good point. Could it be made into two sentences and made a little more explicit, perhaps with a ‘for example’? I think it’s an important point and worth expanding on.

96: ‘Individuals who learn they are at high risk for carrying inherited genetic mutations such as BRCA1/2 have the responsibility of (i.e., “duty to warn”) informing first- and second-degree blood relatives that they too may be mutation carriers’ – I would qualify this with a ‘Some individuals….may feel’ – otherwise you are prescribing a moral and ethical stance, which is dangerous waters. I know for example many people who feel it is not their duty and this might be a huge breach of ethics – unless they had asked if someone wanted to know such things first. Naturally this is a very complex area so you may wish to consult a qualified genetic councillor on writing about this accurately. If you wanted to take a CS/PAR approach to this, you’d actually say ‘CS can help understand different people’s attitudes towards sharing information about risk with relatives, and co-create learning and development interventions to support people to make informed decisions when communicating about such risk’ – or words to that effect if you see where I’m coming from?

103: typo – in sum.

107: ‘We based the procedures for the approach on implementation quality criteria

108 described by Heigl and colleagues’ – seeing as this is the basis for a large and complex table, a line or two about what this criteria is and how you applied it to this work might help the reader make more sense of the table (I had to go back and forth a couple of times and read the original paper before it clicked for me – but I might be slow!). line 175 explains it in more detail, and parts of this section could be moved earlier?

Methods and other sections

124: ‘Survivors’ – again, this term is used – is it distinct from other people involved as non-professional researchers (referred to on line 154 as ‘CS members’. Suggest this term is defined, or changed to ‘people affected by cancer (which can include people with, survivors of, people pre-disposed to, family of…etc – define as you wish). Later the acronym OCS is also used – being consistent as early as possible will help the reader navigate these complex terms.

125: the para FITD might need further review by someone more familiar with this method – this short section felt like it might need to be integrated further? In other words, why am I reading about it? Because this method was used? Could a sentence like ‘our method was informed by the FITD….because….) before hand make the link clearer?

146: stylistically, starting a new section with ‘thus’ might need looking at. I think it’s fine after a set up clause, but this is a matter of style, so take or leave.

161: ‘snowball sampling’ – while I know what you mean here, but noting that some readers might not (aside from the fact that some citizen science projects might actually sample actual snow!) perhaps a reference to this method for more information, or a short explanation of what that means?

218: ‘All CS members were female’ – does this paper require some commentary on this? Perhaps a line in the discussion around 284?

225: ‘listservs’ – define this term

252: does this line and others need putting in inverted commas ‘gaining insights about the experiences of ovariancancer survivors that will be useful to me,’?

286: ‘using probing techniques’ suggest you define this

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Reviewer #1: Yes: Ilda Hoxhaj

Reviewer #2: Yes: Jack S Nunn

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Applying Citizen Science to engage families affected by ovarian cancer in developing genetic service outreach strategies

PONE-D-21-22428R1

Dear Dr. McBride,

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.

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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: 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 #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

**********

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: Thank you for the opportunity to read such an interesting manuscript , I wish you all the best and hope we can stay in touch about http://scienceforall.world/STARDIT

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Reviewer #2: Yes: Jack S Nunn