Nationwide implementation of the non-invasive prenatal test: Evaluation of a blended learning program for counselors

Linda Martin; Janneke T. Gitsels-van der Wal; Caroline J. Bax; Mijntje J. Pieters; Jacqueline C. I. Y. Reijerink-Verheij; Robert-Jan Galjaard; Lidewij Henneman; Dutch NIPT Consortium

doi:10.1371/journal.pone.0267865

Peer Review History

Original SubmissionJanuary 25, 2022
9 Feb 2022 Decision Letter - Antonio Simone Laganà, Editor PONE-D-22-02421Concerning Submission of Research Nationwide implementation of the Non-Invasive Prenatal Test: Evaluation of a blended learning program for counselorsPLOS ONE Dear Dr. Martin, 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 Mar 26 2022 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. 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When you resubmit, please ensure that you provide the correct grant numbers for the awards you received for your study in the ‘Funding Information’ section. 4. Please ensure that you refer to Figure 1 in your text as, if accepted, production will need this reference to link the reader to the figure. Additional Editor Comments: The topic of the manuscript is interesting. Nevertheless, the reviewers raised several concerns: considering this point, I invite authors to perform the required major revisions. [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: Yes Reviewer #2: Yes ******** 2. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: Yes Reviewer #2: No ****** 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: No Reviewer #2: No ****** 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 ****** 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: Dear Authors, I found your paper interesting. I believe strong effort should be put in the implementation of campaign to acknowledge health care professionals regarding NIPT. This test has been a large diffusion, but, women do not perform an adequately informed choice. Your paper highlight the importance of programs to improve de adequacy of health care professionals counseling regarding NIPT, therefore good job! I have some minor revisions: 1) I would try to ameliorate the abstract readability, is a bit tortuous, abstract is the first insight into a paper therefore describe your job in an easier way. - I would have talk about prenatal aneuploidy screening not anomaly screening - I would have specified who do you mena by Duch prenatal counselors (obstetricians, midwives, family doctors - Sentence linke " were positively associated with more correctly answered knowledge questions" I would have said positively associated with a higher level of knowledge for example In the mean text The introduction 1) Again discuss about prenatal screening for aneuploidies not anomalies 2) line 62 is essential to facilitate informed choice for expectant parents regarding prenatal 63 screening" I would have said to allow an effective/adequate pregnant women's informed choice line 89 I would suggest to mention this paper PMID: 33111167, that again shows how an not-adequate counseling from "counselor" associate with a poor understanding of women regarding NIPT, often considered as "diagnostic" and not "screening" I would recommend your paper to be published with minor revisions Reviewer #2: This manuscript evaluations the impact of a training program. These kinds of evaluations are useful to inform other providers of similar educational initiatives. The manuscript can be improved by addressing the following issues: 1. Introduction: Provide the reader with a brief description of what Non-Invasive Prenatal Testing is. 2. Introduction: What are first and second-tiers? Anyone from a different health system does not know what this means. 3. Introduction: line 75 - I would not use the word 'unacceptable' - replace it with something like 'problematic' 4. Introduction: line 80 - what is interfering education by commercial test providers? 5. Introduction: line 86-87 It is not correct to say that little is know about how efficient and effective educational programs are for healthcare implementations. There is plenty of literature about education on other topics. 6. Materials and Methods: line 134 - what is RIVM-CvB and RC (spell out acronyms at first use) 7. Materials and Methods line 176 - an unpaired t-test is not the correct statistical test. The sample is presumably mostly the same participants at both assessments. I understand that they can't be matched, but the mean score on the first assessment could be used as the threshold score for the comparison of the posttest scores using a one-sample t test. 8. Materials and Methods line 178-179: Why are separate regressions used for the pre and post? ****** 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: 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.0267865.r001
Revision 1
1 Mar 2022 Author Response March, 1st 2022 Dear reviewers, Thank you for your expert review of our manuscript titled “Nationwide implementation of the Non-Invasive Prenatal Test: Evaluation of a blended learning program for counselors”, PONE-D-22-02421. We have used your comments to improve our paper further, and we are pleased to send you our revised manuscript. Our response after each comment in italics are provided below. Reviewer #1: Dear Authors, I found your paper interesting. I believe strong effort should be put in the implementation of campaign to acknowledge health care professionals regarding NIPT. This test has been a large diffusion, but, women do not perform an adequately informed choice. Your paper highlight the importance of programs to improve the adequacy of health care professionals counseling regarding NIPT, therefore good job! Thank you for your encouraging words! I have some minor revisions: 1) I would try to ameliorate the abstract readability, is a bit tortuous, abstract is the first insight into a paper therefore describe your job in an easier way. We do agree with the reviewer that the abstract could be made more readable. Therefore, we changed is according to the suggestions below (line 29-43 of the manuscript): “This study assesses the results of a mandatory blended learning-program for counselors (e.g. midwives, sonographers, obstetricians) guiding national implementation of the Non-Invasive Prenatal Test (NIPT). We assessed counselors' 1) knowledge about prenatal aneuploidy screening, 2) factors associated with their knowledge (e.g. counselors' characteristics, attitudes towards NIPT), and 3) counselors’ attitudes regarding the blended learning.” - I would have talk about prenatal aneuploidy screening not anomaly screening We do understand this suggestion. Since we measured not only knowledge about aneuploidy screening but also about structural anomaly screening we used the term prenatal anomaly screening. However, most knowledge items concerned aneuploidy screening and the article is about implementation of NIPT. Therefore, we changed our wording into prenatal aneuploidy screening (line 32; see inserted text comment 1). - I would have specified who do you mean by Duch prenatal counselors (obstetricians, midwives, family doctors). We implemented this suggestion (line 30): “This study assesses the results of a mandatory blended learning-program for counselors (e.g. midwives, sonographers, obstetricians) guiding national implementation of the Non-Invasive Prenatal Test (NIPT). We assessed counselors' 1) knowledge about prenatal aneuploidy screening, 2) factors associated with their knowledge (e.g. counselors' characteristics, attitudes towards NIPT), and 3) counselors’ attitudes regarding the blended learning.” - Sentence linke " were positively associated with more correctly answered knowledge questions". I would have said positively associated with a higher level of knowledge for example. Thank you for this more readable suggestion (see line 43): “Work experience and secondary care work-setting were positively associated with a higher level of knowledge.” In the mean text The introduction 1) Again discuss about prenatal screening for aneuploidies not anomalies. In line with our response to the earlier comment on this topic we changed our wording into prenatal aneuploidy screening thoughout the manuscript. 2) line 62 is essential to facilitate informed choice for expectant parents regarding prenatal 63 screening" I would have said to allow an effective/adequate pregnant women's informed choice We agree with the reviewer that the suggested text improves the manuscript. We therefore edited the text into: “Such knowledge, e.g. understanding the advantages and limitations of NIPT compared to other prenatal tests and knowing how to interpret the results, is essential to enable pregnant women making an adequate informed choice regarding prenatal screening (line 63).” 3) line 89 I would suggest to mention this paper PMID: 33111167, that again shows how an not-adequate counseling from "counselor" associate with a poor understanding of women regarding NIPT, often considered as "diagnostic" and not "screening" Thank you for putting our attention towards this interesting article. We now refer to this article at line 75-78: “The resulting knowledge gaps are problematic since insight into NIPT's test characteristics is fundamental for understanding the clinical implications of the test results and the fact that NIPT is not diagnostic, diagnostic options and thereby for providing adequate counseling that supports informed decisions [4,6,14,35].” Reviewer #2: This manuscript evaluations the impact of a training program. These kinds of evaluations are useful to inform other providers of similar educational initiatives. The manuscript can be improved by addressing the following issues: 1. Introduction: Provide the reader with a brief description of what Non-Invasive Prenatal Testing is. We thank the reviewer for this relevant suggestion. In lines 55-57 we briefly describe NIPT: “NIPT is a maternal blood test using cell-free DNA, mostly targeted at the common aneuploidies Down-, Edwards- and Patau syndrome. In most countries, NIPT is readily commercially available, although it has increasingly been offered as a second-tier pr first-tier an aneuploidy screening test, within nationwide healthcare programs, in which mostly primary maternity care providers offer accompanying pre-test counseling [1-3].” 2. Introduction: What are first and second-tiers? Anyone from a different health system does not know what this means. NIPT can be offered as a first-tier screening test to all pregnant women regardless of their risk level, or as a second-tier test to women with increased risk for fetal aneuploidies only. This has been added to the introduction (line 97-102): “In 2017, alongside the transition of NIPT from a second-tier test, only available for women with a high risk pregnancy based on the First trimester Combined Test results or medical indication, to a first-tier screening test for all pregnant women, the Dutch government set new educational requirements for healthcare professionals who provide counseling for prenatal aneuploidy screening [18,19]” 3. Introduction: line 75 - I would not use the word 'unacceptable' - replace it with something like 'problematic' We replaced the word ‘unacceptable’ for ‘problematic’, according to the reviewer’s suggestion (line 75-76). 4. Introduction: line 80 - what is interfering education by commercial test providers? By intefering education we ment biased education and changed the text accordingly (see line 81). 5. Introduction: line 86-87 It is not correct to say that little is know about how efficient and effective educational programs are for healthcare implementations. There is plenty of literature about education on other topics. We agree with the reviewer that line 86-87 is not correct. We corrected into (line 89): “Furthermore, relatively little is known about efficient and effective educational programs accompanying implementation of NIPT, ….etc.” 6. Materials and Methods: line 134 - what is RIVM-CvB and RC (spell out acronyms at first use) In line 141, in the revised manuscript, we spell out the acronym RIVM-CvB (Centre for Population Screening at the National Institute for Public Health and the Environment). In 123 we already spell out RC (Regionals Centers for prenatal screening). 7. Materials and Methods line 176 - an unpaired t-test is not the correct statistical test. The sample is presumably mostly the same participants at both assessments. I understand that they can't be matched, but the mean score on the first assessment could be used as the threshold score for the comparison of the posttest scores using a one-sample t test. Thank you for this remark! In line with the reviewers comment we did discuss which statistical test we should use. By reading the manuscript again we do see a discrepance in our work. In line 185 we wrote: Mean scores at T0 and T1 were statistically evaluated using an unpaired sample t-test. However, in line 194 we wrote: One sample proportion tests were performed to identify significant differences between the results of T0 and T1. We did the latter, but unfortunately we did not remove line 185 from the manuscript. Now we did. 8. Materials and Methods line 178-179: Why are separate regressions used for the pre and post? By using separate models for T0 and T1 we took a conservative approach, because we did not want to lean on assumptions about overlapping groups within T0 and T1. To clarify this to the readers we edited the text (line 188-190) into: “Separate models were analyzed for T0 and T1, because we did not want to lean on the assumption that the same participants were included at both assessments.” Attachments Attachment Submitted filename: Response to Reviewers_LH.docx https://doi.org/10.1371/journal.pone.0267865.r002
18 Apr 2022 Decision Letter - Antonio Simone Laganà, Editor Concerning Submission of Research Nationwide implementation of the Non-Invasive Prenatal Test: Evaluation of a blended learning program for counselors PONE-D-22-02421R1 Dear Dr. Martin, 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 for payment will follow shortly after the formal acceptance. To ensure an efficient process, please log into Editorial Manager at http://www.editorialmanager.com/pone/, click the 'Update My Information' link at the top of the page, and double check that your user information is up-to-date. 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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 #2: All comments have been addressed ******** 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 #2: Yes ****** 3. Has the statistical analysis been performed appropriately and rigorously? Reviewer #1: Yes Reviewer #2: Yes ****** 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: Yes Reviewer #2: (No Response) ****** 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 ****** 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: Well done, I think the paper has been improved. The topic as I've already commented is actual and interesting therefore i will recommend it for publication Reviewer #2: The authors have done a good job of responding to the review feedback. I have no further suggestions. ****** 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 #1: No Reviewer #2: No https://doi.org/10.1371/journal.pone.0267865.r003
Formally Accepted
22 Apr 2022 Acceptance Letter - Antonio Simone Laganà, Editor PONE-D-22-02421R1 Nationwide implementation of the Non-Invasive Prenatal Test: Evaluation of a blended learning program for counselors Dear Dr. Martin: I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department. 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 plosone@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. Antonio Simone Laganà Academic Editor PLOS ONE https://doi.org/10.1371/journal.pone.0267865.r004

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