Parental Preferences about Policy Options Regarding Disclosure of Incidental Genetic Findings in Newborn Screening: Using Videos and the Internet to Educate and Obtain Input
Abstract
:1. Introduction
1.1. Cystic Fibrosis Newborn Screening
1.2. Next Generation Sequencing in Newborn Screening
1.3. Policy Options for Disclosure
2. Materials and Methods
2.1. Design
2.2. Methodologic Elements to Support the Objectives
2.2.1. Embedded Explanatory Videos
2.2.2. Substitution of a Generic Disease X instead of CF
2.2.3. Vignettes and Complementary Modes for Preference Questions
2.3. Sample and Recruitment
2.4. Data Management and Statistical Analyses
3. Results
3.1. Sample Characteristics
3.2. Reaction to Animated Video Survey Format
3.3. Disclosure Preferences
3.4. Value Comparison
4. Discussion
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Acknowledgments
Conflicts of Interest
Financial Disclosure
Abbreviations
cystic fibrosis | CF |
CFTR-related metabolic syndrome | CRMS |
cystic fibrosis screen positive inconclusive diagnosis | CFSPID |
newborn screening | NBS |
next generation sequencing | NGS |
incidental findings | IFs |
confidence interval | CI |
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Policy | Definition | Advantages (Benefits) | Disadvantages | Added Value a |
---|---|---|---|---|
Fully Informed | Incidental findings always disclosed to parents | No information is withheld from parents | Requires more time that could be devoted to other health care needs Potential for confusion, misconceptions, emotional stress | Being fully informed |
Withholding Information | Incidental findings with very minimal or no risk to health are withheld from parents | Reduces parental confusion and risk of emotional problems | Parents not consulted about information that is part of them and their family | Reducing emotional stress |
Parents Decide | Parents are counseled just prior to results communication, and then make a decision about how much information to receive about incidental genetic findings | Parents are in control of the screening results that affect their baby and can determine if they want full details if they are willing to risk emotional distress | Requires more time that could be devoted to other health care needs Curiosity may lead to the parents asking questions about details they don’t actually want or need to know | Autonomy is preserved (freedom of choice) b |
Characteristic | N | (%) |
---|---|---|
Married or stable relationship | 123 | (96.1) |
First-time parent | 93 | (72.7) |
Infant born >2 weeks before due date | 19 | (14.8) |
NICU stay >2 days | 11 | (8.6) |
Race and/or ethnicity of respondent | ||
White | 112 | (87.5) |
Non-White | 11 | (8.6) |
Missing response | 5 | (3.9) |
Education | ||
High school | 11 | (9) |
Undergraduate degree | 62 | (48.4) |
Postgraduate degree | 52 | (40.6) |
Missing response | 3 | (2.3) |
Health literacy (need help reading…) | ||
never | 86 | (67.2) |
rarely | 28 | (21.9) |
sometimes | 8 | (6.3) |
often | 1 | (0.8) |
always | 1 | (0.8) |
not answered | 4 | (3.1) |
First-Rank Policy Group | p-Value * | |||
---|---|---|---|---|
Fully Informed | Withholding Information | Parents Decide | ||
Comparison A (comparing the importance of being fully informed to reducing distress) | ||||
Mild X scenario | ||||
mean (std. dev.) | 0.28 (0.33) | 0.76 (0.78) | 0.58 (0.44) | <0.001 * |
median (IRQ) | 0.17 (0.42) | 0.73 (1.55) | 0.48 (0.52) | |
Disease X carrier status scenario | ||||
mean (std. dev.) | 0.32 (0.38) | 0.96 (0.61) | 0.55 (0.38) | <0.001 * |
median (IRQ) | 0.19 (0.40) | 1.0 (0.92) | 0.47 (0.44) | |
Comparison B (comparing the importance of autonomy in decision-making versus reducing emotional distress) | ||||
Mild X scenario | ||||
mean (std. dev.) | 1.61 (0.50) | 0.76 (0.74) | 1.37 (0.46) | <0.001 * |
median (IRQ) | 1.76 (0.63) | 0.58 (1.10) | 1.40 (0.61) | |
Disease X carrier status scenario | ||||
mean (std. dev.) | 1.51 (0.56) | 1.03 (0.40) | 1.45 (0.43) | 0.015 * |
median (IRQ) | 1.69 (0.71) | 1.05 (0.66) | 1.46 (0.58) | |
Comparison C (comparing autonomous Decision-making without all details to allowing experts to make decisions) | ||||
Mild X scenario | ||||
mean (std. dev.) | 0.97 (0.62) | 0.95 (0.83) | 1.27 (0.51) | 0.008 * |
median (IRQ) | 1.01 (1.00) | 1.01 (1.60) | 1.33 (0.61) | |
Disease X carrier status scenario | ||||
mean (std. dev.) | 1.04 (0.61) | 1.08 (0.61) | 1.25 (0.54) | 0.092 |
median (IRQ) | 1.13 (1.01) | 1.23 (0.92) | 1.33 (0.65) |
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Farrell, M.H.; Mooney, K.E.; Laxova, A.; Farrell, P.M. Parental Preferences about Policy Options Regarding Disclosure of Incidental Genetic Findings in Newborn Screening: Using Videos and the Internet to Educate and Obtain Input. Int. J. Neonatal Screen. 2022, 8, 54. https://doi.org/10.3390/ijns8040054
Farrell MH, Mooney KE, Laxova A, Farrell PM. Parental Preferences about Policy Options Regarding Disclosure of Incidental Genetic Findings in Newborn Screening: Using Videos and the Internet to Educate and Obtain Input. International Journal of Neonatal Screening. 2022; 8(4):54. https://doi.org/10.3390/ijns8040054
Chicago/Turabian StyleFarrell, Michael H., Katherine E. Mooney, Anita Laxova, and Philip M. Farrell. 2022. "Parental Preferences about Policy Options Regarding Disclosure of Incidental Genetic Findings in Newborn Screening: Using Videos and the Internet to Educate and Obtain Input" International Journal of Neonatal Screening 8, no. 4: 54. https://doi.org/10.3390/ijns8040054
APA StyleFarrell, M. H., Mooney, K. E., Laxova, A., & Farrell, P. M. (2022). Parental Preferences about Policy Options Regarding Disclosure of Incidental Genetic Findings in Newborn Screening: Using Videos and the Internet to Educate and Obtain Input. International Journal of Neonatal Screening, 8(4), 54. https://doi.org/10.3390/ijns8040054