@article{0b4b91b76d324293b2dd269b3f225979,
title = "Parental interest in genomic sequencing of newborns: enrollment experience from the BabySeq Project",
abstract = "Purpose: Newborn genomic sequencing (nGS) has great potential to improve pediatric care. Parental interest and concerns about genomics are relatively unexplored. Understanding why parents decline research consent for nGS may reveal implementation barriers. Methods: We evaluated parental interest in a randomized trial of nGS in well-baby and intensive care unit nursery settings. Interested families attended an informational enrollment session (ES) with a genetic counselor prior to consenting. Reason(s) for declining participation and sociodemographic associations were analyzed. Results: Of 3860 eligible approached families, 10% attended ES, 67% of whom enrolled. Of 1760 families queried for decline reasons, 58% were uninterested in research. Among 499 families considering research, principal reasons for decline prior to ES included burdensome study logistics (48%), feeling overwhelmed postpartum (17%), and lack of interest/discomfort with genetic testing (17%). Decliners after ES more often cited concerns about privacy/insurability (41%) and uncertain/unfavorable results (23%). Conclusion: Low interest in research and study logistics were major initial barriers to postpartum enrollment and are likely generic to many postpartum research efforts. Concerns over privacy and result implications were most commonly cited in decliners after ES. Understanding parental concerns around research nGS may inform future integration of nGS into newborn screening, predictive testing, and pediatric diagnostics.",
keywords = "Consent for genomic testing, Genomic sequencing, Newborn, Newborn screening, Parental concerns",
author = "{The BabySeq Project Team} and Genetti, {Casie A.} and Schwartz, {Talia S.} and Robinson, {Jill O.} and VanNoy, {Grace E.} and Devan Petersen and Stacey Pereira and Shawn Fayer and Peoples, {Hayley A.} and Agrawal, {Pankaj B.} and Betting, {Wendi N.} and Holm, {Ingrid A.} and McGuire, {Amy L.} and Waisbren, {Susan E.} and Yu, {Timothy W.} and Green, {Robert C.} and Beggs, {Alan H.} and Parad, {Richard B.} and Agrawal, {Pankaj B.} and Beggs, {Alan H.} and Betting, {Wendi N.} and Ozge Ceyhan-Birsoy and Christensen, {Kurt D.} and Dmitry Dukhovny and Shawn Fayer and Frankel, {Leslie A.} and Genetti, {Casie A.} and Chet Graham and Green, {Robert C.} and Gutierrez, {Amanda M.} and Maegan Harden and Holm, {Ingrid A.} and Krier, {Joel B.} and Lebo, {Matthew S.} and Levy, {Harvey L.} and Xingquan Lu and Kalotina Machini and McGuire, {Amy L.} and Murry, {Jaclyn B.} and Medha Naik and Nguyen, {Tiffany T.} and Parad, {Richard B.} and Peoples, {Hayley A.} and Devan Petersen and Uma Ramamurthy and Vivek Ramanathan and Rehm, {Heidi L.} and Amy Roberts and Robinson, {Jill O.} and Serguei Roumiantsev and Schwartz, {Talia S.}",
note = "Funding Information: The authors thank the families and clinicians for their participation in the BabySeq Project. Special thanks to the current and former research assistants, genetic counselors, and research nurses involved in the BabySeq Project recruitment at Brigham and Women's Hospital, Boston Children{\textquoteright}s Hospital, and Massachusetts General Hospital. This work was supported by grants U19 HD077671 and R01 HD075802 from the National Institute of Child Health and Human Development and National Human Genome Research Institute of the National Institutes of Health, as well as by the Manton Center for Orphan Disease Research of Boston Children{\textquoteright}s Hospital. Publisher Copyright: {\textcopyright} 2018, American College of Medical Genetics and Genomics.",
year = "2019",
month = mar,
day = "1",
doi = "10.1038/s41436-018-0105-6",
language = "English (US)",
volume = "21",
pages = "622--630",
journal = "Genetics in Medicine",
issn = "1098-3600",
publisher = "Lippincott Williams and Wilkins",
number = "3",
}