Exercise as a therapy for sickle cell-associated musculoskeletal pain among children: Healthcare professionals’ perspectives

Britney Pinamang Gyampo; Isaac Mensah Bonsu

doi:10.1371/journal.pone.0327183

Peer Review History

Original SubmissionJune 10, 2025
30 Sep 2025 Decision Letter - Tomasz Kaminski, Editor Dear Dr. Mensah Bonsu, 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. Please submit your revised manuscript by Nov 14 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. 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'. 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Please include captions for your Supporting Information files at the end of your manuscript, and update any in-text citations to match accordingly. Please see our Supporting Information guidelines for more information: http://journals.plos.org/plosone/s/supporting-information . 4. If the reviewer comments include a recommendation to cite specific previously published works, please review and evaluate these publications to determine whether they are relevant and should be cited. There is no requirement to cite these works unless the editor has indicated otherwise. Additional Editor Comments: Dear Authors, Both reviewers have recommended major revisions, and I agree with their assessment. Please revise the manuscript accordingly and carefully address all reviewer comments in your response. Best regards, Tomasz W Kaminski [Note: HTML markup is below. Please do not edit.] Reviewer's Responses to Questions Comments to the Author 1. Is the manuscript technically sound, and do the data support the conclusions? Reviewer #1: Yes Reviewer #2: Partly ******** 2. Has the statistical analysis been performed appropriately and rigorously? -->?> Reviewer #1: N/A Reviewer #2: N/A ****** 3. Have the authors made all data underlying the findings in their manuscript fully available??> The PLOS Data policy Reviewer #1: Yes Reviewer #2: No ****** 4. Is the manuscript presented in an intelligible fashion and written in standard English??> Reviewer #1: Yes Reviewer #2: Yes ****** Reviewer #1: This manuscript describes a structured interview study of healthcare providers knowledge and attitudes toward the use of exercise as a therapeutic strategy for managing SCD-associated MSK pain in children with SCD. The healthcare providers were chosen from two healthcare facilities in the Ashanti Region of Ghana. The introduction adequately describes the background literature supporting the study rationale. The study methodology and qualitative analysis are appropriate, and the interview guide is provided as supplemental materials. The discussion adequately addresses the study strengths and limitations. I had a number of questions/suggestions to improve this manuscript: 1. Please provide more detail about the pediatricians and nurses who took part in the interviews. Table 1 provides data on the years of clinical experience, but it is unclear how much of that clinical experience included participating in the care of children with SCD. Further, the context of care is important to the interpretation of these results. I assume these healthcare providers were providing in-patient care for acute vaso-occlusive pain, rather than addressing the management of chronic pain, improving physical activity, or attempting to reduce the frequency of acute pain in an outpatient setting. 2. The introduction and discussion with their associate references also need to be clear about these contexts, as the role of exercise in speeding improvement in the resolution of acute pain may be quite different from its role in improving cardiovascular health, improving physical activity, or moderating the effects of chronic pain. Reviewer #2: The manuscript addresses an important gap in the literature by exploring healthcare professionals’ perspectives on exercise as a therapeutic option for musculoskeletal pain in children with sickle cell disease (SCD). The qualitative design is well-justified, and the findings highlight both perceived benefits and practical barriers, which will be valuable for clinical practice and future program design. Strengths: Original and contextually relevant research (first such study in Ghana). Strong methodology: purposive sampling, ethical clearance, data saturation, and trustworthiness criteria. Practical insights into barriers (pain, family attitudes, limited resources) and facilitators (collaboration, family involvement). Areas for improvement: Clarity of writing: Some sentences are long and could be streamlined for readability. A thorough language edit would improve flow. Discussion depth: The link between identified barriers (e.g., kinesiophobia, family concerns) and proposed interventions could be expanded with more practical recommendations. Limitations: The authors acknowledge the limited scope (two hospitals, only nurses and pediatricians). It would be useful to suggest explicitly how including physiotherapists, hematologists, and families in future studies could enrich findings. Implications for practice: The conclusion could more clearly emphasize actionable recommendations (e.g., integrating physiotherapists into clinic days, caregiver education programs). ****** 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: No Reviewer #2: No ******** [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. https://doi.org/10.1371/journal.pone.0327183.r001
Revision 1
8 Oct 2025 Author Response Response to Reviewer’s comments Dear Editor, We genuinely appreciate the reviewer’s valuable feedback on improving the clarity and specificity of the manuscript. We recognize the importance of refining certain aspects to strengthen the overall quality of the study. Please find our detailed responses below for each comment. Reviewer #1: Comment: Please provide more details about the pediatricians and nurses who took part in the interviews. Table 1 provides data on the years of clinical experience, but it is unclear how much of that clinical experience included participating in the care of children with SCD. Response: We thank the reviewer for this important comment. We have now clarified in the manuscript that “All nineteen (19) pediatric nurses and pediatricians who participated in the interviews were directly involved in the treatment of children with SCD. As these hospitals run dedicated SCD clinics weekly, the participants' clinical experience, as reported in Table 1, includes regular and active participation in the care of children with SCD.” Highlighted in RED on page 5. Comment: Further, the context of care is important to the interpretation of these results. I assume these healthcare providers were providing inpatient care for acute vaso-occlusive pain, rather than addressing the management of chronic pain, improving physical activity, or attempting to reduce the frequency of acute pain in an outpatient setting. Response: We thank the reviewer. The reviewer's assumption is understandable; however, our study was specifically designed to explore perspectives on exercise, not in the context of acute inpatient pain management, but as a therapeutic strategy for managing persistent musculoskeletal pain and preventing pain crises in the outpatient setting. We intended to investigate a gap in the literature, moving beyond the paradigm of purely pharmacological pain management during crises to explore how pediatricians and pediatric nurses view a proactive, non-pharmacological intervention like exercise for improving long-term health outcomes. Comment: The introduction and discussion with their associated references also need to be clear about these contexts, as the role of exercise in speeding improvement in the resolution of acute pain may be quite different from its role in improving cardiovascular health, improving physical activity, or moderating the effects of chronic pain. Response: We thank the reviewer for this critically important observation. We agree with the reviewer that the distinct roles of exercise in an acute pain context versus a chronic health context are fundamentally different. We have addressed these in the manuscript to reflect the chronic musculoskeletal pain and long-term morbidity in children with SCD, separating it from the acute vaso-occlusive crisis model. Reviewer #2: Comment: Clarity of writing: Some sentences are long and could be streamlined for readability. A thorough language edit would improve flow. Response: We thank the reviewer for this feedback. We agree that enhancing the clarity and readability of the manuscript is important. We have performed a thorough language edit throughout the manuscript, focusing on streamlining long sentences and improving overall flow. Comment: Discussion depth: The link between identified barriers (e.g., kinesiophobia, family concerns) and proposed interventions could be expanded with more practical recommendations. Response: These have been revised to read as “A key psychological barrier is kinesiophobia, the fear of movement due to pain [18]. In SCD, where pain is unpredictable and severe, this fear is particularly understandable. Children may develop avoidance behaviors, which can be unintentionally reinforced by caregivers and clinicians who prioritize safety and pain prevention [19]. This cycle of fear and inactivity paradoxically worsens long-term outcomes by promoting musculoskeletal deconditioning. To break this cycle, clinicians can adopt several practical strategies. A foundational approach is to structure activity program pacing, co-creating an activity plan with the child that begins with short, manageable sessions of low-impact exercise (e.g., 5-10 minutes of walking) and gradually increases in a patient-directed manner. Using an activity diary can provide visual proof of progress and help desensitize the fear that activity always leads to pain. For more complex cases, a proactive referral to a physiotherapist specializing in pediatric chronic pain can facilitate a tailored, graded exposure program, while a clinical health psychologist can address the underlying challenging thoughts using techniques like cognitive behavioral therapy”. Highlighted in RED on page 15. Comment: Limitations: The authors acknowledge the limited scope (two hospitals, only nurses and pediatricians). It would be useful to suggest explicitly how including physiotherapists, hematologists, and families in future studies could enrich findings. Response: We have revised the limitation section to read as “The findings are based on data from two hospitals and the perspectives of pediatric nurses and physicians only, which may limit the transferability of the results. Future research should actively include the voices of other critical stakeholders to enrich the findings. For instance, physiotherapists could provide essential expertise on practical exercise prescription and graded movement strategies. Hematologists could offer insights into integrating physical activity with complex medical regimens. Most crucially, including patients with SCD and their families is fundamental to ensuring that any future exercise interventions are feasible, acceptable, and aligned with their lived experiences and priorities”. Highlighted in RED on page 18. Comment: Implications for practice: The conclusion could more clearly emphasize actionable recommendations (e.g., integrating physiotherapists into clinic days, caregiver education programs). Response: The implication for practice has been revised and highlighted in RED on page 19. “Based on our findings, we propose incorporating physiotherapists or exercise specialists into SCD clinic days to provide real-time assessment, counseling, and the development of individualized activity plans. Again, establish formal channels for pediatricians and nurses to collaborate with hematologists and mental health professionals, ensuring physical activity is addressed as a core component of comprehensive SCD care. Implementing these strategies can help transform exercise from a perceived risk into a standard, managed therapy for improving health outcomes in this population.” Attachments Attachment Submitted filename: RESPONSE TO REVIEWERS (Plos one).docx https://doi.org/10.1371/journal.pone.0327183.r002
5 Nov 2025 Decision Letter - Tomasz Kaminski, Editor Dear Dr. Mensah Bonsu, 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. Please submit your revised manuscript by Dec 20 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. 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 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, Tomasz W. Kaminski Academic Editor PLOS ONE Journal Requirements: If the reviewer comments include a recommendation to cite specific previously published works, please review and evaluate these publications to determine whether they are relevant and should be cited. There is no requirement to cite these works unless the editor has indicated otherwise. 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. Additional Editor Comments: Dear Authors, I apologize for the longer waiting time during the review process. Reviewer 1 changed their recommendation from major revision to accept after the revisions were submitted. Reviewer 2 was unavailable to re-evaluate the manuscript, but their main concerns were, in my opinion, properly addressed. To ensure fairness, I invited a third reviewer, who suggested only minor comments. Based on all feedback, the decision has been updated to minor revision. Thank you for your patience and for the thoughtful improvements made to the manuscript. Best regards, Tomasz W. Kaminski [Note: HTML markup is below. Please do not edit.] Reviewers' comments: Reviewer's Responses to Questions Comments to the Author Reviewer #1: All comments have been addressed Reviewer #3: (No Response) ******** 2. Is the manuscript technically sound, and do the data support the conclusions??> Reviewer #1: Yes Reviewer #3: Yes ****** 3. Has the statistical analysis been performed appropriately and rigorously? -->?> Reviewer #1: Yes Reviewer #3: Yes ****** 4. Have the authors made all data underlying the findings in their manuscript fully available??> The PLOS Data policy Reviewer #1: Yes Reviewer #3: Yes ****** 5. Is the manuscript presented in an intelligible fashion and written in standard English??> Reviewer #1: Yes Reviewer #3: Yes ****** Reviewer #1: (No Response) Reviewer #3: I really enjoyed reading your manuscript on healthcare professionals’ perspectives on exercise as a therapy for musculoskeletal pain in children with sickle cell disease. This is a very timely and important topic, especially given the growing interest in non-drug approaches in SCD care. Authors clearly shows the clinical relevance of exercise and provide thoughtful insights into what helps or hinders its use. The qualitative approach is well chosen and seems to have been conducted carefully, with attention to COREQ guidelines and ethical considerations. I do have a few suggestions that could make the manuscript even stronger: • Add practical exercise examples: Including specific exercises suitable for children with SCD—like low-impact or graded activities—would give clinicians clearer takeaways and link perceptions to real-world practice. • You discuss physiotherapists in the limitations, but acknowledging their role earlier could make the discussion of implementation barriers clearer. • Check flow and readability: A few of the longer sentences could be tightened for smoother reading throughout the manuscript. • Clean up references: There are a few repeated or inconsistently formatted references; standardizing them would improve overall presentation. Overall, this is a strong and valuable manuscript, and addressing these points could make it even more reader-friendly and impactful. ****** 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: Carlton Dampier MD Reviewer #3: Yes: Marta Wolosowicz ******** [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.] To ensure your figures meet our technical requirements, please review our figure guidelines: https://journals.plos.org/plosone/s/figures You may also use PLOS’s free figure tool, NAAS, to help you prepare publication quality figures: https://journals.plos.org/plosone/s/figures#loc-tools-for-figure-preparation. NAAS will assess whether your figures meet our technical requirements by comparing each figure against our figure specifications. https://doi.org/10.1371/journal.pone.0327183.r003
Revision 2
6 Nov 2025 Author Response Response to Reviewer’s comments Dear Editor, We appreciate the reviewer’s valuable feedback on improving the clarity and specificity of the manuscript. Please find our detailed responses below for each comment. Reviewer #3: comment: Add practical exercise examples: Including specific exercises suitable for children with SCD—like low-impact or graded activities—would give clinicians clearer takeaways and link perceptions to real-world practice. Response: We thank the reviewer for this important comment. However, we have added practical exercise examples. Highlighted on page 14 and read as “Such a program, like 'play-based & social activities' (indoor recreational games, light ball play, dancing, and performing daily chores under supervision), Comment: You discuss physiotherapists in the limitations, but acknowledging their role earlier could make the discussion of implementation barriers clearer. Response: We appreciate the reviewer’s insightful observation. The current study specifically explored healthcare professionals' (pediatric nurses’ and pediatricians’) perspectives on exercise as a therapy for sickle cell–associated musculoskeletal pain among children. As such, the inclusion of physiotherapists was beyond the original scope and participant design of the study. The intention was to capture how frontline medical and nursing professionals perceive, recommend, or integrate exercise within pediatric sickle cell care, given their frequent patient contact in clinical and ward settings. Participants acknowledged that their availability will enhance the implementation of the exercise in the management of MSK in SCD. However, we fully acknowledge that physiotherapists play an important role in exercise prescription, movement retraining, and graded activity planning for children with sickle cell disease. Their perspectives would indeed deepen understanding of implementation feasibility and enhance interdisciplinary insight. In line with this, we have clarified their potential contribution earlier in the discussion to strengthen the connection between professional collaboration and implementation barriers. We have also noted the need for future studies to include physiotherapists and other rehabilitation professionals to provide a more holistic view of exercise-based interventions for this population. Comment: Check flow and readability: A few of the longer sentences could be tightened for smoother reading throughout the manuscript. Response: Thank you for the observation. The longer sentences have been reviewed and revised throughout the manuscript to improve flow, clarity, and readability. Comment: Clean up references: There are a few repeated or inconsistently formatted references; standardizing them would improve overall presentation. Response: Thank you for the observation. The reference list has been thoroughly reviewed, and repeated or inconsistently formatted entries have been corrected. Attachments Attachment Submitted filename: Response to Reviewer (PLOS ONE ).docx https://doi.org/10.1371/journal.pone.0327183.r004
11 Nov 2025 Decision Letter - Tomasz Kaminski, Editor Exercise as a therapy for sickle cell associated musculoskeletal pain among children: Healthcare professionals’ perspectives. PONE-D-25-29804R2 Dear Dr. Mensah Bonsu, 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. An invoice will be generated when your article is formally accepted. Please note, if your institution has a publishing partnership with PLOS and your article meets the relevant criteria, all or part of your publication costs will be covered. Please make sure your user information is up-to-date by logging into Editorial Manager at Editorial Manager® and clicking the ‘Update My Information' link at the top of the page. For questions related to billing, please contact billing support . 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, Tomasz W. Kaminski Academic Editor PLOS ONE Additional Editor Comments: The Authors adequately addressed the minor comments raised by the Reviewer 3. https://doi.org/10.1371/journal.pone.0327183.r005
Formally Accepted
Acceptance Letter - Tomasz Kaminski, Editor PONE-D-25-29804R2 PLOS ONE Dear Dr. Mensah Bonsu, I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now being handed over to our production team. At this stage, our production department will prepare your paper for publication. This includes ensuring the following: * All references, tables, and figures are properly cited * All relevant supporting information is included in the manuscript submission, * There are no issues that prevent the paper from being properly typeset You will receive further instructions from the production team, including instructions on how to review your proof when it is ready. Please keep in mind that we are working through a large volume of accepted articles, so please give us a few days to review your paper and let you know the next and final steps. Lastly, if your institution or institutions have a press office, please let them know about your upcoming paper now to help maximize its impact. If they'll be preparing press materials, please inform our press team within the next 48 hours. 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. You will receive an invoice from PLOS for your publication fee after your manuscript has reached the completed accept phase. If you receive an email requesting payment before acceptance or for any other service, this may be a phishing scheme. Learn how to identify phishing emails and protect your accounts at https://explore.plos.org/phishing. If we can help with anything else, please email us at customercare@plos.org. Thank you for submitting your work to PLOS ONE and supporting open access. Kind regards, PLOS ONE Editorial Office Staff on behalf of Dr. Tomasz W. Kaminski Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0327183.r006

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