Including ELSI research questions in newborn screening pilot studies

for the Bioethics and Legal Workgroup of the Newborn Screening Translational Research Network

doi:10.1038/s41436-018-0101-x

Including ELSI research questions in newborn screening pilot studies

for the Bioethics and Legal Workgroup of the Newborn Screening Translational Research Network

Pediatrics - Genetics and Metabolism

Research output: Contribution to journal › Article › peer-review

19 Scopus citations

Abstract

Background: The evidence review processes for adding new conditions to state newborn screening (NBS) panels rely on data from pilot studies aimed at assessing the potential benefits and harms of screening. However, the consideration of ethical, legal, and social implications (ELSI) of screening within this research has been limited. This paper outlines important ELSI issues related to newborn screening policy and practices as a resource to help researchers integrate ELSI into NBS pilot studies. Approach: Members of the Bioethics and Legal Workgroup for the Newborn Screening Translational Research Network facilitated a series of professional and public discussions aimed at engaging NBS stakeholders to identify important existing and emerging ELSI challenges accompanying NBS. Results: Through these engagement activities, we identified a set of key ELSI questions related to (1) the types of results parents may receive through newborn screening and (2) the initiation and implementation of NBS for a condition within the NBS system. Conclusion: Integrating ELSI questions into pilot studies will help NBS programs to better understand the potential impact of screening for a new condition on newborns and families, and make crucial policy decisions aimed at maximized benefits and mitigating the potential negative medical or social implications of screening.

Original language	English (US)
Pages (from-to)	525-533
Number of pages	9
Journal	Genetics in Medicine
Volume	21
Issue number	3
DOIs	https://doi.org/10.1038/s41436-018-0101-x
State	Published - Mar 1 2019

Bibliographical note

Funding Information:
We would like to thank Joanne Adelberg for her support on this manuscript. The work presented in this manuscript funded in part by the Newborn Screening Translational Research Network (funded by the National Institutes of Health, Eunice Kennedy Shriver National Institute of Child Health & Human Development —275201300011C-6-0-1).

Publisher Copyright:
© 2018, American College of Medical Genetics and Genomics.

Keywords

ELSI
Ethics
Newborn screening
Pilot studies
Research

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http://orca.cf.ac.uk/115766/1/Including%20ELSI%20Research%20Questions%20in%20Newborn%20Screening%20Pilots%20Studies.pdf

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title = "Including ELSI research questions in newborn screening pilot studies",

abstract = "Background: The evidence review processes for adding new conditions to state newborn screening (NBS) panels rely on data from pilot studies aimed at assessing the potential benefits and harms of screening. However, the consideration of ethical, legal, and social implications (ELSI) of screening within this research has been limited. This paper outlines important ELSI issues related to newborn screening policy and practices as a resource to help researchers integrate ELSI into NBS pilot studies. Approach: Members of the Bioethics and Legal Workgroup for the Newborn Screening Translational Research Network facilitated a series of professional and public discussions aimed at engaging NBS stakeholders to identify important existing and emerging ELSI challenges accompanying NBS. Results: Through these engagement activities, we identified a set of key ELSI questions related to (1) the types of results parents may receive through newborn screening and (2) the initiation and implementation of NBS for a condition within the NBS system. Conclusion: Integrating ELSI questions into pilot studies will help NBS programs to better understand the potential impact of screening for a new condition on newborns and families, and make crucial policy decisions aimed at maximized benefits and mitigating the potential negative medical or social implications of screening.",

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author = "{for the Bioethics and Legal Workgroup of the Newborn Screening Translational Research Network} and Goldenberg, {Aaron J.} and Michele Lloyd-Puryear and Brosco, {Jeffrey P.} and Bradford Therrell and Lynn Bush and Susan Berry and Amy Brower and Natasha Bonhomme and Bruce Bowdish and Denise Chrysler and Angus Clarke and Thomas Crawford and Edward Goldman and Sally Hiner and Howell, {R. Rodney} and David Orren and Wilfond, {Benjamin S.} and Michael Watson",

note = "Funding Information: We would like to thank Joanne Adelberg for her support on this manuscript. The work presented in this manuscript funded in part by the Newborn Screening Translational Research Network (funded by the National Institutes of Health, Eunice Kennedy Shriver National Institute of Child Health & Human Development —275201300011C-6-0-1). Publisher Copyright: {\textcopyright} 2018, American College of Medical Genetics and Genomics.",

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AU - for the Bioethics and Legal Workgroup of the Newborn Screening Translational Research Network

AU - Goldenberg, Aaron J.

AU - Lloyd-Puryear, Michele

AU - Brosco, Jeffrey P.

AU - Therrell, Bradford

AU - Bush, Lynn

AU - Berry, Susan

AU - Brower, Amy

AU - Bonhomme, Natasha

AU - Bowdish, Bruce

AU - Chrysler, Denise

AU - Clarke, Angus

AU - Crawford, Thomas

AU - Goldman, Edward

AU - Hiner, Sally

AU - Howell, R. Rodney

AU - Orren, David

AU - Wilfond, Benjamin S.

AU - Watson, Michael

N1 - Funding Information: We would like to thank Joanne Adelberg for her support on this manuscript. The work presented in this manuscript funded in part by the Newborn Screening Translational Research Network (funded by the National Institutes of Health, Eunice Kennedy Shriver National Institute of Child Health & Human Development —275201300011C-6-0-1). Publisher Copyright: © 2018, American College of Medical Genetics and Genomics.

PY - 2019/3/1

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N2 - Background: The evidence review processes for adding new conditions to state newborn screening (NBS) panels rely on data from pilot studies aimed at assessing the potential benefits and harms of screening. However, the consideration of ethical, legal, and social implications (ELSI) of screening within this research has been limited. This paper outlines important ELSI issues related to newborn screening policy and practices as a resource to help researchers integrate ELSI into NBS pilot studies. Approach: Members of the Bioethics and Legal Workgroup for the Newborn Screening Translational Research Network facilitated a series of professional and public discussions aimed at engaging NBS stakeholders to identify important existing and emerging ELSI challenges accompanying NBS. Results: Through these engagement activities, we identified a set of key ELSI questions related to (1) the types of results parents may receive through newborn screening and (2) the initiation and implementation of NBS for a condition within the NBS system. Conclusion: Integrating ELSI questions into pilot studies will help NBS programs to better understand the potential impact of screening for a new condition on newborns and families, and make crucial policy decisions aimed at maximized benefits and mitigating the potential negative medical or social implications of screening.

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Including ELSI research questions in newborn screening pilot studies

Abstract

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Keywords

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