PONE-D-24-03539The liminal space between hope and grief: the phenomenon of uncertainty as experienced by people living with relapsing-remitting multiple sclerosisPLOS 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: Partly

Reviewer #3: Yes

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

Reviewer #1: N/A

Reviewer #2: N/A

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: No

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: * General aspects

As far as originality is concerned, the article can be considered to make a new and relevant contribution to this field of study by proposing to understand the experience of uncertainty lived by people recently diagnosed with multiple sclerosis. The text is coherent, but excessively long. This contributes to an uneasy reading within and between the sections of the article.

The uncertainty experienced by people with multiple sclerosis is a complex subject and deserves special attention from the academic community. We appreciate the opportunity to review this article, as it is indeed a very relevant topic.

The manuscript is clearly and coherently written and the topic is very important, especially if we consider that societies are ageing rapidly and that it is essential to pay attention to the aspects that people value when faced with a medical diagnosis and the inherent uncertainty (especially in pathologies such as multiple sclerosis). Here are some aspects that could strengthen the arguments presented and improve the article:

*Title:

It is coherent with the content presented, clear and objective. It arouses the reader's interest.

* Summary:

The abstract is clear and well organized, following the structure of the article. However, the section on results could be improved by presenting the main results of the study more clearly and objectively. The way this section is written is a little vague and does not allow a clear link to be established with what is presented in the conclusion section. In turn, it is too focused on health professionals (rather than the experience of existential uncertainty by people with multiple sclerosis). It might also be useful to include more clearly the contribution/implications of the study for research in the field.

*Introduction:

The introduction adequately presents the context and importance of the study. It is clearly written, but perhaps a little briefer than is desirable in terms of the existing research on the subject. In other words, the introduction gives a reasonably clear account of the implications of uncertainty related to medical diagnosis on patients' well-being and quality of life, but it is more vague when it comes to research into the specific scope of this study - the uncertainty experienced by people with multiple sclerosis. The article would advance significantly if the authors presented a more complete overview of the relevant research in this field, in order to make the research problem more explicit. In other words, the article would benefit from a more detailed analysis of recent research, and the results obtained, in this specific field of study.

That said, it is important that the introduction situates the existing knowledge on the topic under study, helping to formulate the hypotheses/arguments that will be developed in the article.

It is also necessary to make a link between previous work and this article. For example, if the following is stated: “A phenomenological study of the lifeworld of people with MS is not a new venture”, it would be interesting to explore further why the approach adopted is innovative. In other words, it could be useful to clarify the originality and relevance of the study, since the innovative nature of the study is not entirely clear.

The theoretical approach used seems adequate for reading and interpreting the results obtained and the introduction ends with a presentation of the study's objective - which is very appropriate.

*Methodology:

The authors adequately describe the methodology used, however this section is quite extensive. The article would benefit from a shorter, less detailed section. For example, the description of the study design and the participant selection process could be more succinct. Omitting the names of the hospitals might also be appropriate in order to guarantee the anonymity of the participants.

Given the nature of the study, the authors seem uncomfortable with “pre-defined” designations, as they state: “For practical reasons, this study upholds these concepts while acknowledging their problematic nature”. It is therefore suggested that a structure more in keeping with a phenomenological study be adopted. Furthermore, it doesn't seem to make much sense to mention the tasks that were done (or not) by the first author of the study (or by others). Similarly, the ethical considerations should be shorter (for example, it doesn't seem relevant to mention that a participant became emotional during the interview, or what the researcher's behavior was at that moment). Thus, the section on the methodology used should describe the methods and techniques used clearly and objectively, and non-essential aspects should be eliminated or summarized.

*Results

The results section begins with a sentence indicating the bibliographic reference and page. I don't understand why this is: “In this section, the essence of the researched phenomenon and its constituents will be described [31 p.255].”

This is followed by a paragraph in which the authors frame what will be presented next, but it is not clear whether these ideas derive specifically from the interviews conducted. I don't think a framework of this length is justified. If it is to be kept, it should be shorter.

The results section is around 17 pages long and that seems too long to me. As has been suggested throughout the article, we recommend reducing this section. The content is very interesting, but it could clearly focus on the essentials and present the main results of the study carried out - illustrated, if possible, with extracts from the interviews conducted. This would make the reading more objective, richer and more interesting, as the transcripts are adequate and useful for understanding the participants' perspective.

* Discussion

The discussion begins with a summary of the main results, which is appropriate. Nevertheless, some repetition could be eliminated. There is room for a more in-depth discussion about the uncertainty of people diagnosed with Multiple Sclerosis and the role of health professionals.

What follows is a set of arguments that would make more sense in the introduction of the article rather than in the discussion. At this point, one would expect a more in-depth critical analysis of the main results of the study. What often happens is that one of the results obtained is presented and compared with one or more bibliographic sources, but there is a lot of information about these studies (e.g. how they were conducted, limitations, etc.), which makes reading difficult and not very fluid.

It is therefore suggested that the theoretical and empirical framework be more robust in the introduction so that the discussion can adopt a more analytical and critical lens on the results obtained. There is also a need for greater fluidity in the reading of this section (and the whole article) which could be achieved by significantly reducing the size of the article.

With regard to the section with recommendations for healthcare practices, we suggest eliminating the first few paragraphs which repeat what has already been mentioned above and keeping only the authors' recommendations based on the research carried out (duly supported by theory and research).

As far as the main limitations of the study are concerned, the constitution of the sample may be a weakness (especially the fact that most of the participants were recruited by health professionals) as it is not a study with a random sample. As the authors point out, this has implications for the generalizability of the results. It might also be useful to analyze the cultural influence on the experience of uncertainty, since we are analyzing a specific country.

It is also essential to make a clear statement about the contribution and/or implications of the study in relation to existing knowledge. What, in fact, are the main conclusions of the study and what implications does it have for the advancement of knowledge in this field? What implications does a study, albeit somewhat exploratory, on individual perceptions (of people with multiple sclerosis) of uncertainty have?

*Conclusion

Based on the results of the study, it can be concluded that uncertainty is a multifaceted experience and that people newly diagnosed with multiple sclerosis have to deal with various levels of uncertainty. In this context, it is up to healthcare professionals to support these patients - not only with clear and objective medical information, but also with a greater understanding of their doubts, difficulties and uncertainties. Given that these are the main conclusions of the study, what implications do they have for the advancement of knowledge in this field?

It is also very important to elaborate a little more on what is recommended in the final paragraph (“Training in this field, reflective discussions, and supervision might help healthcare professionals better support their patients.”).

Reviewer #2: The topic chosen by the authors is worth investigating. The uncertainty and unpredictability associated with managing conditions such as relapsing-remitting Multiple Sclerosis (RRMS) are most often inadequately addressed, leading to misunderstandings about the condition.

Patients with RRMS frequently experience unpredictable or unfamiliar symptoms, which can cause uncertainty and challenges in their daily lives. Such unpredictability and uncertainty may also lead healthcare providers to overlook patients' experiences of the condition. They may also overlook essential aspects of care. However, different aspects of uncertainty explored in this study can help enhance awareness among healthcare practitioners and patients. This can, in turn, result in developing strategies that empower individuals to navigate their condition with greater confidence, thereby improving patient care.

While the manuscript addresses an important topic, it is currently somewhat difficult to follow. The authors have used several technical terms and jargon, which could make it challenging for the broader scientific community to fully understand. It is essential that the manuscript be presented in a clear format to ensure the research is understood, and its implications are fully recognised. Examples include ‘attunement’, ‘uniquely’, ‘antecedents’, ‘irreconcilable’ etc

Line 117, the statement "[uncertainty] is a lack of knowledge of which the individual is aware" is somewhat unclear. Please improve clarity.

The final paragraph of the introduction regarding phenomenology may be shifted to the methods section.

Study Design

The study design is too lengthy and may cause confusion. Please revise it for clarity and focus.

“The gap between on the hand hand a descriptive and on the other hand an interpretive approach was closed by Dahlberg 216 and Dahlberg’s third way.” Does not read well.

“Second, a medical student who worked as an intern in both the 231 Elisabeth-TweeSteden Hospital and on this research project, actively searched 232 patient files and reached out to possible candidates after consulting with their 233 physician.”

• Did the candidates/participants know that their information was being accessed for research purposes? And how was patient confidentiality maintained when accessing their files?

Line 257, Data Collection

• First paragraph: The authors introduce the concept of data within the framework of phenomenology; however, this section could benefit from being simplified or even removed. Supporting references could enhance its relevance.

Please remove duplicates

• e.g. line 241-42 and line 267-68 cover the same content

Line 284: follow-up questions or probing questions?

Please be consistent in using either 'participant' or 'candidate.'

Line 287-288: This implies that the interview guide has not been the core component of the interview process or perhaps a secondary resource. Please clarify.

The data analysis and Ethics sections could benefit from further refinement, as they currently appear lengthy. A more concise presentation will enhance clarity and strengthen the manuscript's overall impact. It might be helpful to focus on key points while ensuring that essential details are still communicated effectively.

Results Section

There is a lot of information in the content, which shows the richness of the result. However, it might be easier to understand if it were reviewed and shortened. For instance, the section on the "search for definition" could be revised by highlighting a few key issues that directly address the main point while summarising the remaining content. This would enhance focus and readability.

Given the points mentioned above, I recommend revising the manuscript before resubmitting it for another review. Therefore, I will pause my review of the results and discussion sections at this time. Thank you

Reviewer #3: Thank you for a well-prepared manuscript which I had the joy to read. The manuscript addresses a significant and often overlooked aspect of living with relapsing-remitting multiple sclerosis (RRMS). I have read the manuscript carefully. Generally, your manuscript is well-written and well-structured. To improve your manuscript, I have a few comments for you.

Introduction: The introduction is well-written and describes the previous research on the topic and defines the scope of the research well. However, I think it would benefit from a more explicit specification of the research question.

Methods: The methods are described very well and in sufficient detail. However, the study design section is somewhat lengthy and a bit confusing. It would be clearer if the authors focused on describing what the study is, rather than what it is not.

Recruitment: The anonymity of informants should be observed carefully.

The relevance of “Chosen medication” and “medication and usage” is unclear. It might be more pertinent to include information about social support instead.

There is an overlap between Table 1 and Table 2; choosing one would streamline the presentation.

The phrase “As described previously” leads to repetition of information and should be omitted.

The statement “interviews that were conducted immediately after inclusion” needs clarification, as it does not align with the description on page 16, lines 337-338, where it states that “all participants were briefed by the interviewer (EvR), first by email or phone and second in person, preceding the actual interview.” Please elaborate on what “immediately” means.

What is the significance of informing about how long the interviews lasted?

How do the interviewers relate to the informants?

The use of journals to “capture non-verbal communication, atmosphere, and other relevant observations” should be explained in terms of how these observations were utilized.

Results: The results are well analyzed and written. However, the subtheme “Painful memory of the past” does not accurately describe the content, which seems to focus more on the participants’ difficulties in adjusting to their new situation in everyday life.

Discussion: The heading “Discussion” competes with the subheading “Reflections on the findings.” Please specify whether this section is a discussion or a reflection, and introduce what is to be discussed or reflected on more precisely.

Recommendations for Healthcare Practice: This paragraph is quite long. I suggest you to focus on the implications for practice derived from the findings, rather than referencing other works.

Limitations and Strengths: Please discuss whether it is a limitation or a strength that the focus on the experience of uncertainty in people with RRMS is at such an early stage. Consider discussing the pros and cons of this. Additionally, why is future research at an earlier stage recommended? Could a longitudinal study be explicitly recommended for future research?

Thank you for considering my feedback. I believe these revisions will enhance the clarity and impact of the manuscript.

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Reviewer #1: No

Reviewer #2: No

Reviewer #3: Yes: Lise Sæstad Beyene

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The liminal space between hope and grief: the phenomenon of uncertainty as experienced by people living with relapsing-remitting multiple sclerosis

PONE-D-24-03539R1

Dear Dr. van Reenen,

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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Kind regards,

Alejandro Botero Carvajal, MD

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

Reviewer #3: 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

Reviewer #3: Yes

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

Reviewer #1: N/A

Reviewer #3: 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: (No Response)

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 #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. 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)

Reviewer #3: Thank you for the opportunity to review your revised manuscript. I acknowledge the significant progress you have made since the initial submission. The improvements enhance the clarity and depth of your research. I recommend this manuscript to be accepted for publication. It contributes valuable insights to the field and advances our understanding of the topic.

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Reviewer #1: No

Reviewer #3: No

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