When to return genomic newborn screening results: health care professional perspectives
Lewis, A. C. F.; Buchanan, A. H.; Goldenberg, A. J.; Knoppers, B. M.; McGuire, A. L.; Green, R. C.; Holm, I. A.
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Background and Objectives: Dozens of projects around the world are sequencing the genomes of healthy babies near birth. Genomic information becomes actionable across infancy, childhood and adulthood, making timing of return a design choice for programs. We report the first study of health care professionals' views on timing. Methods: We conducted a qualitative interview study of US-based clinical geneticists, genetic counselors, laboratory personnel, pediatric primary care clinicians and genomic screening implementers. Participants responded to three strategies: staged throughout childhood when information becomes actionable, all at birth with adult-actionable results deferred, and all at birth. Transcripts were analyzed using framework analysis. Results: We interviewed 52 individuals; 39 were asked directly about timing. Giving parents a choice, raised by participants rather than presented, was the most endorsed position (18), ahead of staging across childhood (12). Many viewed staging as preferable in theory, but feasibility concerns weighed against it, including that 'actionability' was not a robust enough concept. A further concern was that parents would not grasp the distinction between data generated and data examined, on which staging depends. Where information is staged, participants saw a role for adolescent assent; where it is not, disclosure to the developing child becomes important, and participants identified a lack of support for parents. Conclusions: Tying the return of information to the age of actionability is intuitive but hard to operationalize. Parental choice was the most endorsed position but will only be viable with decision support and guidance for disclosure to children.
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