Parental Preferences about Policy Options Regarding Disclosure of Incidental Genetic Findings in Newborn Screening: Using Videos and the Internet to Educate and Obtain Input
Round 1
Reviewer 1 Report
Overall, the paper is well-written and the information is somewhat helpful, although there are significant limitations of the study, particularly the oversampling of well-educated Whites. The videos were interesting, although it would have been preferable to have their content and that of any other related written discussions evaluated as to educational level. Likewise, there were some instances of unnecessary wording that may have influenced the responses (ie. In movie 2 – “… the experts who propose this policy will know better than the parents what is good for them to know …” This sentence is presented in such a way as to alienate the viewer towards this choice (in my opinion).
Since the paper is also about he use of the internet and videos for assistance in obtaining parental input, and is noted in this way in the abstract, I suggest changing the title to: Using Videos and the Internet to Obtain Parental Input into Policies Regarding Disclosure of Genetic Findings in Newborn Screening: A Possible Model.
The information below is intended to assist in improving the paper:
Line 89 – To better reflect reality, please substitute “inaccurate or insufficient” into this sentence, “…know the family because of inaccurate or insufficient labeling of …”.
Line 112 – For improved readability, Table 1 needs to be reformatted to leave spaces between columns and better agreement of successive columns with column, especially with respect to the last two definitions.
Line 294 – In Table 3, it is interesting to note that 47.7% of parents remembered receiving an ‘abnormal’ result and an additional 33.6% recalled that the screen showed ‘something,’ for a total of 80% with a “not normal” newborn screen. This is a rather astonishing number given that 63% (50.0 + 13.3] were ‘definitely’ or ‘somewhat certain’ that they were not told their newborn’s screening result, and that newborn screening in general has a very low number of abnormal results. These findings should be at least mentioned in the discussion.
Line 296 – Table 4 not listed in the text, as far as I could tell.
Author Response
Overall, the paper is well-written and the information is somewhat helpful, although there are significant limitations of the study, particularly the oversampling of well-educated Whites. The videos were interesting, although it would have been preferable to have their content and that of any other related written discussions evaluated as to educational level. Likewise, there were some instances of unnecessary wording that may have influenced the responses (ie. In movie 2 – “… the experts who propose this policy will know better than the parents what is good for them to know …” This sentence is presented in such a way as to alienate the viewer towards this choice (in my opinion). Thank you. We acknowledged the limitations and described our determination of the educational level of the videos.
Since the paper is also about the use of the internet and videos for assistance in obtaining parental input, and is noted in this way in the abstract, I suggest changing the title to: Using Videos and the Internet to Obtain Parental Input into Policies Regarding Disclosure of Genetic Findings in Newborn Screening: A Possible Model. We appreciate this thoughtful suggestion and are agreeable to a title change. It does seem appropriate to highlight the use of videos and the Internet since this is a novel feature of our study. However, we prefer not to include “A Possible Model” and keep the emphasis on “Parental Preferences about Policy Options.” Consequently, we propose: “Parental Preferences about Policy Options Regarding Disclosure of Genetic Findings in Newborn Screening: Using Videos and the Internet to Educate and Obtain Input.” The videos were intended to both educate and obtain input.” We feel that this title describes the study well fits better with the theme of the Special Issue on Ethical and Psychosocial Aspects of Genomics in the Neonatal Period. Thanks again for your suggestion.
The information below is intended to assist in improving the paper:
Line 89 – To better reflect reality, please substitute “inaccurate or insufficient” into this sentence, “…know the family because of inaccurate or insufficient labeling of …”. This revision was made.
Line 112 – For improved readability, Table 1 needs to be reformatted to leave spaces between columns and better agreement of successive columns with column, especially with respect to the last two definitions. Done— thank you.
Line 294 – In Table 3, it is interesting to note that 47.7% of parents remembered receiving an ‘abnormal’ result and an additional 33.6% recalled that the screen showed ‘something,’ for a total of 80% with a “not normal” newborn screen. This is a rather astonishing number given that 63% (50.0 + 13.3] were ‘definitely’ or ‘somewhat certain’ that they were not told their newborn’s screening result, and that newborn screening in general has a very low number of abnormal results. These findings should be at least mentioned in the discussion. We agree and have been concerned about some of the data on “personal experiences.” Because the submitted manuscript included four tables and 5 figures, we have elected to delete the original Table 3 and cite only the relevant data in the text, namely the limited knowledge of newborn screening.
Line 296 – Table 4 not listed in the text, as far as I could tell. Thank you. We have ensured that all tables and figures are referred to in the revised manuscript.
Reviewer 2 Report
In Video #1: Why propagate the "PKU test" misnomer which is incomplete and incorrect information?
Video #2 has a typographical error in ‘height’ visible at 1 minute 20 seconds.
Has the language, in the videos, been vetted for an eight-grade education?
What would you propose to overcome the issues of recruiting a more diverse parental population as your study participants?
Line 267 refers to Table 4 rather than Table 3. An explanation of the term ‘value score’ is essential for a reader who is not as familiar with the statistical analyses performed. The terms in Table 4, comparison A, comparison B, comparison C, are not defined either as a footnote to the table nor in the text at the point of the initial referral to table 4. The portions of the table corresponding to each comparison need to be better set off, potentially by a simple maneuver as making the comparison A text left justified and bold.
The data underlying during the statement on line 286 that respondents reported being the primary caregiver for the baby… is not supported by the information provided in Tables 2 or 3. This potentially biases the subsequent analyses because what is not obvious, to a reader, is who is the medical decision maker within the parental dyad. How did you account for the dynamics of decision making?
Table 3 would suggest that newborn screening was not fresh in the minds of these parents if 50% do not remember being told about their infant's newborn screening result. Where there enough respondents in the category that recalled something about newborn screening to do a sub-analysis?
Since there are multiple jurisdictions in which any notation within the medical record is immediately available to a patient/their duly authorized representative (parent), information about carrier status (withhold information policy) may cause additional utilization of healthcare resources and even the potential generation of a lack of trust in the healthcare provider. Parents and may not remember which decision they previously made nor all of the implications of that decision. Have you considered the extension of the testing to look at parental retention of the information presented within the videos?
Author Response
In Video #1: Why propagate the "PKU test" misnomer which is incomplete and incorrect information? This is a good question. Whether appropriate or not, it’s clear that the maternity ward nurses who explain to parents (usually just mothers) about newborn screening refer to the tests as the “PKU test” but this needs to change.
Video #2 has a typographical error in ‘height’ visible at 1 minute 20 seconds. Thank you for viewing the video and identifying this error.
Has the language, in the videos, been vetted for an eight-grade education? Yes. This is mentioned in the revised manuscript.
What would you propose to overcome the issues of recruiting a more diverse parental population as your study participants? This is a difficult challenge. It would be labor intensive to recruit more minority parents. Instead of flyers in clinics, we believe that it would be necessary to meet with individual families and perhaps offer other incentives. In our study, we had resource limitations that precluded such tactics.
Line 267 refers to Table 4 rather than Table 3. An explanation of the term ‘value score’ is essential for a reader who is not as familiar with the statistical analyses performed. The terms in Table 4, comparison A, comparison B, comparison C, are not defined either as a footnote to the table nor in the text at the point of the initial referral to table 4. The portions of the table corresponding to each comparison need to be better set off, potentially by a simple maneuver as making the comparison A text left justified and bold. We agree and have added descriptors to the table, which is now Table 3.
The data underlying during the statement on line 286 that respondents reported being the primary caregiver for the baby… is not supported by the information provided in Tables 2 or 3. This potentially biases the subsequent analyses because what is not obvious, to a reader, is who is the medical decision maker within the parental dyad. How did you account for the dynamics of decision making? This is difficult to address because we have limited information on the parental dynamics of decision-making to reach conclusions about potential biases. However, by knowing who is the primary caregiver and who takes the baby to the PCP we have some indication about which parent will be making decisions. We feel that it will of interest interesting to repote that “Respondents who had reported being the primary caregiver for the baby were more likely to vote for the Parents Decide policy (p=0.006, Wilcoxon) or full disclosure policy (p=0.035, Wilcoxon).”
Table 3 would suggest that newborn screening was not fresh in the minds of these parents if 50% do not remember being told about their infant's newborn screening result. Where there enough respondents in the category that recalled something about newborn screening to do a sub-analysis? Unfortunately, no.
Since there are multiple jurisdictions in which any notation within the medical record is immediately available to a patient/their duly authorized representative (parent), information about carrier status (withhold information policy) may cause additional utilization of healthcare resources and even the potential generation of a lack of trust in the healthcare provider. Parents and may not remember which decision they previously made nor all of the implications of that decision. Have you considered the extension of the testing to look at parental retention of the information presented within the videos? Yes, but resource limitations made this impossible.
Reviewer 3 Report
The authors described their online survey study on the parental views about the disclosure policy options for IF in NBS. It is an interesting study with the first time internet-based educational video used in a NBS-related survey.
The findings are helpful to the readers to understand the parental views on this important emerging ethical issues about how to handle IF on NBS.
The authors should add some discussion that the interests of the minors should not be determined only or mainly by the parents. The same ethical principal applies to IF in NBS as well as presymptomatic genetic tests on minor or NBS for adult onset diseases. The perspectives of public (e.g. people considering for their own whether they would like their parents to decide for them when they were babies for such conditions) can be very different.
In Table 1, as the authors stated that withholding information with IF very minimal or no risk to health, then the wording of disadvantages as "may affect their baby" should be rephrased to tune down the adverse effect instead.
Author Response
The authors described their online survey study on the parental views about the disclosure policy options for IF in NBS. It is an interesting study with the first time internet-based educational video used in a NBS-related survey.
The findings are helpful to the readers to understand the parental views on this important emerging ethical issues about how to handle IF on NBS. Thank you.
The authors should add some discussion that the interests of the minors should not be determined only or mainly by the parents. The same ethical principal applies to IF in NBS as well as presymptomatic genetic tests on minor or NBS for adult onset diseases. The perspectives of public (e.g. people considering for their own whether they would like their parents to decide for them when they were babies for such conditions) can be very different. Thank you. We added two sentences to the Discussion to address this concern.
In Table 1, as the authors stated that withholding information with IF very minimal or no risk to health, then the wording of disadvantages as "may affect their baby" should be rephrased to tune down the adverse effect instead. This was rephrased.
