Prevalence and help-seeking for infertility in a population with a low fertility rate

Mei-Chuan Lee; Pei-Shan Chien; Yue Zhou; Tsung Yu

doi:10.1371/journal.pone.0306572

Peer Review History

Original SubmissionMarch 26, 2024
5 Jun 2024 Decision Letter - Patrick Ifeanyi Okonta, Editor PONE-D-24-11968Prevalence and Help-Seeking for Infertility in a Population with a Low Fertility RatePLOS ONE Dear Dr. Yu, 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 Jul 20 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, Patrick Ifeanyi Okonta, MBBCh, MPH, FWACS, FMCOG, MD, DRH 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. We suggest you thoroughly copyedit your manuscript for language usage, spelling, and grammar. If you do not know anyone who can help you do this, you may wish to consider employing a professional scientific editing service. The American Journal Experts (AJE) (https://www.aje.com/) is one such service that has extensive experience helping authors meet PLOS guidelines and can provide language editing, translation, manuscript formatting, and figure formatting to ensure your manuscript meets our submission guidelines. Please note that having the manuscript copyedited by AJE or any other editing services does not guarantee selection for peer review or acceptance for publication. Upon resubmission, please provide the following: The name of the colleague or the details of the professional service that edited your manuscript A copy of your manuscript showing your changes by either highlighting them or using track changes (uploaded as a supporting information file) A clean copy of the edited manuscript (uploaded as the new manuscript file)” 3. Thank you for stating the following financial disclosure: "The study was funded by the National Science and Technology Council in Taiwan." 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. 4. Thank you for stating the following in your Competing Interests section: "None declared." Please complete your Competing Interests on the online submission form to state any Competing Interests. If you have no competing interests, please state ""The authors have declared that no competing interests exist."", as detailed online in our guide for authors at http://journals.plos.org/plosone/s/submit-now This information should be included in your cover letter; we will change the online submission form on your behalf. 5. In the online submission form, you indicated that "Please contact Prof. Tsung Yu for the study data and statistical codes." All PLOS journals now require all data underlying the findings described in their manuscript to be freely available to other researchers, either 1. In a public repository, 2. Within the manuscript itself, or 3. Uploaded as supplementary information. This policy applies to all data except where public deposition would breach compliance with the protocol approved by your research ethics board. If your data cannot be made publicly available for ethical or legal reasons (e.g., public availability would compromise patient privacy), please explain your reasons on resubmission and your exemption request will be escalated for approval. 6. 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. Additional Editor Comments: Author(s) should address the review comments of the three reviewers. In particular, authors should give 1. More details on the construction of the interview questionnaire 2. The reasons for the response rate and its probable effects on the validity of the results 3. Discussion and Conclusion based on the data from this study [Note: HTML markup is below. Please do not edit.] 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: No Reviewer #2: Yes Reviewer #3: Yes ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: No Reviewer #2: Yes Reviewer #3: Yes ****** 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 ****** 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: No Reviewer #2: Yes Reviewer #3: Yes ****** 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: A. THE SOURCE OF THE SURVEY QUESTIONNAIRE IS NOT ATTACHED. IS IT A VALIDATED ONE OR NOT? -ALSO NOT MENTIONED! B. (27.9% response rate is too small for a survey of this importance and to deduce any meaningful and representative conclusion or results.) C. A response rate of 70% or more would have been more appropriate. D. The conclusion is biased and inappropriate. E. Infertility definitions 1 and 2; similar and very confusing. What is the relevance of this dichotomy in the definition? Could the authors elaborate on why they did not use only one definition for their analysis? F. Outlandish discussion–not conclusively relevant to the analysis based on the data. G. A need to correct all grammatical errors. Reviewer #2: Good and relevant research. It was a cross sectional study done through telephone interviews which is adequate to get prevalence of a disease condition. The author however did not explore possible cultural and other sociological factors which may be the reason for the low fertility rate in Taiwan. Reviewer #3: The manuscript is well written. technically sound and the statistical analysis is appropriate. However can the conclusion be made to state whether the prevalence of infertility, based on the two operational definitions used for the study, was high or low in comparison to global, regional or national statistics. Anecdotal evidence from the press might not suffice. ****** 6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files. If you choose “no”, your identity will remain anonymous but your review may still be made public. Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy. Reviewer #1: No Reviewer #2: Yes: HAJARATU UMAR SULAYMAN Reviewer #3: 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. Attachments Attachment Submitted filename: Review for Prevalence and Help seeking for infertility.docx https://doi.org/10.1371/journal.pone.0306572.r001
Revision 1
13 Jun 2024 Author Response Dear Prof. Okonta, Thank you for reviewing our manuscript. The comments were very helpful for us to revise and improve the manuscript. Hereby please find below point-by-point responses to the reviewers’ comments. We also attached the revised manuscript with tracked changes and a clean version. We look forward to seeing your further evaluation and response. Sincerely yours, Authors Point-by-point response to the reviewers’ comments Reviewer #1: 1. The source of the survey questionnaire is not attached. Is it a validated one or not? Also not mentioned. Response: Our survey questions were adapted from the National Survey of Family Growth in the United States and the Knowledge, Attitude, and Practice of Contraception Survey in Taiwan. We clarified this point in the Methods section (see lines 101-102 on page 8 in the manuscript with tracked changes). 2. 27.9% response rate is too small for a survey of this importance and to deduce any meaningful and representative conclusion or results. A response rate of 70% or more would have been more appropriate. Response: We acknowledged the low response rate of our survey in the Discussion section (see lines 246-249 on page 15). It was indeed very difficult to get a high response rate in a telephone survey in Taiwan. 3. The conclusion is biased and inappropriate. Response: We do not fully understand this comment. 4. Infertility definitions 1 and 2; similar and very confusing. What is the relevance of this dichotomy in the definition? Could the authors elaborate on why they did not use only one definition for their analysis? Response: As what we explained in the Methods section (see lines 119-123 on page 9), when we designed the questionnaire, we also asked respondents to report how long they had been trying to become pregnant, and we intended to use such information to perform the “current duration” analysis for the prevalence of infertility. However, most of these data that we collected seemed incorrect or were missing responses, and therefore we did not proceed with the current duration analysis. As a result, we used the available data related to how long they had been trying to become pregnant to define infertility in another way, which was the definition 2 of our study. 5. Outlandish discussion–not conclusively relevant to the analysis based on the data. Response: We do not fully understand this comment. 6. A need to correct all grammatical errors. Response: We thank the reviewer for this comment. Accordingly, we asked for help of professional service to edit our manuscript. Reviewer #2: 7. Good and relevant research. It was a cross sectional study done through telephone interviews which is adequate to get prevalence of a disease condition. The author however did not explore possible cultural and other sociological factors which may be the reason for the low fertility rate in Taiwan. Response: We thank the reviewer for this excellent suggestion. To explore the possible cultural and sociological factors that cause the low fertility rate in Taiwan was not the objective of the present study. In fact, we are also working on this issue in another manuscript. 8. Summary and Background: The study was a cross sectional population-based study conducted through telephone interviews which sought to assess the prevalence of infertility as well as the number of people seeking help for fertility. This was a commendable study as it sought to determine the current state of fertility and fertility treatments in Taiwan given their low total fertility rate per woman which is less than 1. Though Taiwan was noted as being one of the countries facing a rapid decline in fertility, the study only concentrated on determining the prevalence of primary infertility; secondary infertility was not mentioned. The background of the study was silent about the cultural and religious beliefs regarding fertility in Taiwan as this would have given more insights the fertility seeking behavior of the people. Response: Because of the questionnaire design we did not estimate the prevalence of secondary infertility, which was one of our study limitations. We provided a reference in the Background section relevant to the social determinants of infertility diagnosis and treatment in Taiwan (see lines 64-69 on page 5). 9. Methodology: Cross sectional studies are good in determining point prevalence and the number of people reached were above 40,000 which is commendable. However, the response rate of 27.9% was low and clear explanation for this was not given. In the first and second definitions of infertility used in this study, it was not clear if the use of contraception was the differentiating factor. Is it possible for ‘childless with no pregnancy attempt’ to have its own place in the definitions of terms in this study; it appeared in both groups? Response: The response rate was not high although we tried our best to conduct the telephone survey. Please see our response to point 2. As for the definition of infertility, please see details in Figure 1. Contraception use was not the differentiating factor. “Childless with no pregnancy attempt” appeared in both definitions. We generated this group because this is a group of people who did not have children and were not trying to become pregnant. We were unable to know whether they were infertile or not. 10. Results and Discussion: With regard to professional help-seeking, 11.6% of men and 10.7% of women had ever consulted a doctor about getting pregnant; 10.4% of men and 9.5% of women had ever received diagnostic tests/treatment to help with conceiving; 3.1% of men and 2.1% of women were currently receiving diagnostic tests/treatment to help with conceiving’. In the above statement does it mean that male infertility is high in Taiwan? How does this compare to other Asian countries? Response: Given that infertility is a “disease of couple”, our results do not imply that male infertility is higher than female infertility in Taiwan. Instead, our results suggested that the prevalence estimates of infertility obtained from male respondents were higher than female respondents in our study samples. 11. In the final recommendations in this study, the author wrote that: These findings also suggest that there is a gap between those who are currently experiencing infertility and those who are currently being treated; hence, we recommend raising awareness of infertility and improving access to infertility healthcare.’ I think the recommendation should go beyond this as there may be some other sociologic factors; be it cultural or based on the norm, which is hindering people from having children. This calls for a wider survey to get to the root cause why people who do not have children are reluctant to do so. Response: We thank the reviewer for this excellent suggestion. As what we responded to point 7, to explore the possible cultural and sociological factors that cause the low fertility rate in Taiwan was not the objective of the present study. In fact, we are also working on this issue in another manuscript. 12. TO THE EDITOR: On a general note, this is a good study considering the paucity of studies from Taiwan and it can be considered for publication if the above issues are addressed. Thank you. Response: We thank the reviewer for these positive comments. Reviewer #3: 13. The manuscript is well written. Technically sound and the statistical analysis is appropriate. However can the conclusion be made to state whether the prevalence of infertility, based on the two operational definitions used for the study, was high or low in comparison to global, regional or national statistics. Anecdotal evidence from the press might not suffice. Response: We thank the reviewer for this point. Accordingly, we added this point to the Discussion section (see lines 262-264 on page 16). Attachments Attachment Submitted filename: point-by-point.docx https://doi.org/10.1371/journal.pone.0306572.r002
20 Jun 2024 Decision Letter - Patrick Ifeanyi Okonta, Editor Prevalence and Help-Seeking for Infertility in a Population with a Low Fertility Rate PONE-D-24-11968R1 Dear Dr. Yu, 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. If you have any questions relating to publication charges, please contact our Author Billing department directly at authorbilling@plos.org. 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, Patrick Ifeanyi Okonta, MBBCh, MPH, FWACS, FMCOG, MD, DRH Academic Editor PLOS ONE Additional Editor Comments (optional): Reviewers' comments: https://doi.org/10.1371/journal.pone.0306572.r003
Formally Accepted
10 Jul 2024 Acceptance Letter - Patrick Ifeanyi Okonta, Editor PONE-D-24-11968R1 PLOS ONE Dear Dr. Yu, 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 If revisions are needed, the production department will contact you directly to resolve them. If no revisions are needed, you will receive an email when the publication date has been set. At this time, we do not offer pre-publication proofs to authors during production of the accepted work. Please keep in mind that we are working through a large volume of accepted articles, so please give us a few weeks 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. 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 Professor Patrick Ifeanyi Okonta Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0306572.r004

Open letter on the publication of peer review reports

PLOS recognizes the benefits of transparency in the peer review process. Therefore, we enable the publication of all of the content of peer review and author responses alongside final, published articles. Reviewers remain anonymous, unless they choose to reveal their names.

We encourage other journals to join us in this initiative. We hope that our action inspires the community, including researchers, research funders, and research institutions, to recognize the benefits of published peer review reports for all parts of the research system.

Learn more at ASAPbio .