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- Title
Psychosocial Effect of Newborn Genomic Sequencing on Families in the BabySeq Project: A Randomized Clinical Trial.
- Authors
Pereira, Stacey; Smith, Hadley Stevens; Frankel, Leslie A.; Christensen, Kurt D.; Islam, Rubaiya; Robinson, Jill Oliver; Genetti, Casie A.; Blout Zawatsky, Carrie L.; Zettler, Bethany; Parad, Richard B.; Waisbren, Susan E.; Beggs, Alan H.; Green, Robert C.; Holm, Ingrid A.; McGuire, Amy L.
- Abstract
Key Points: Question: What is the psychosocial effect of newborn genomic sequencing results on families? Findings: In this randomized clinical trial that included 325 families, scores on measures of psychosocial impact in 3 family domains—parent-child relationship, parents' psychological distress, and parents' relationship—did not show sustained statistical differences between study groups over time. Meaning: This study found no evidence that returning newborn genomic sequencing information has an unfavorable psychosocial effect on families. Importance: Newborn genomic sequencing (nGS) may provide health benefits throughout the life span, but there are concerns that it could also have an unfavorable (ie, negative) psychosocial effect on families. Objective: To assess the psychosocial effect of nGS on families from the BabySeq Project, a randomized clinical trial evaluating the effect of nGS on the clinical care of newborns from well-baby nurseries and intensive care units. Design, Setting, and Participants: In this randomized clinical trial conducted from May 14, 2015, to May 21, 2019, at well-baby nurseries and intensive care units at 3 Boston, Massachusetts, area hospitals, 519 parents of 325 infants completed surveys at enrollment, immediately after disclosure of nGS results, and 3 and 10 months after results disclosure. Statistical analysis was performed on a per-protocol basis from January 16, 2019, to December 1, 2019. Intervention: Newborns were randomized to receive either standard newborn screening and a family history report (control group) or the same plus an nGS report of childhood-onset conditions and highly actionable adult-onset conditions (nGS group). Main Outcomes and Measures: Mean responses were compared between groups and, within the nGS group, between parents of children who received a monogenic disease risk finding and those who did not in 3 domains of psychosocial impact: parent-child relationship (Mother-to-Infant Bonding Scale), parents' relationship (Kansas Marital Satisfaction Scale), and parents' psychological distress (Edinburgh Postnatal Depression Scale anxiety subscale). Results: A total of 519 parents (275 women [53.0%]; mean [SD] age, 35.1 [4.5] years) were included in this study. Although mean scores differed for some outcomes at singular time points, generalized estimating equations models did not show meaningful differences in parent-child relationship (between-group difference in adjusted mean [SE] Mother-to-Infant Bonding Scale scores: postdisclosure, 0.04 [0.15]; 3 months, –0.18 [0.18]; 10 months, –0.07 [0.20]; joint P =.57) or parents' psychological distress (between-group ratio of adjusted mean [SE] Edinburgh Postnatal Depression Scale anxiety subscale scores: postdisclosure, 1.04 [0.08]; 3 months, 1.07 [0.11]; joint P =.80) response patterns between study groups over time for any measures analyzed in these 2 domains. Response patterns on one parents' relationship measure differed between groups over time (between-group difference in adjusted mean [SE] Kansas Marital Satisfaction Scale scores: postdisclosure, –0.19 [0.07]; 3 months, –0.04 [0.07]; and 10 months, –0.01 [0.08]; joint P =.02), but the effect decreased over time and no difference was observed on the conflict measure responses over time. We found no evidence of persistent negative psychosocial effect in any domain. Conclusions and Relevance: In this randomized clinical trial of nGS, there was no persistent negative psychosocial effect on families among those who received nGS nor among those who received a monogenic disease risk finding for their infant. Trial Registration: ClinicalTrials.gov Identifier: NCT02422511 This randomized clinical trial assesses the psychosocial effect of newborn genomic sequencing on families of newborns in the BabySeq Project.
- Subjects
MASSACHUSETTS; FAMILIES &; psychology; NEWBORN screening; EVALUATION of medical care; INTENSIVE care units; SEQUENCE analysis; PSYCHOLOGY of parents; GENETIC testing; RANDOMIZED controlled trials; PSYCHOLOGICAL tests; GENOMICS; INTERPERSONAL relations; PARENT-child relationships; PARENTS; EDINBURGH Postnatal Depression Scale; PSYCHOLOGICAL stress; CHILDREN
- Publication
JAMA Pediatrics, 2021, Vol 175, Issue 11, p1132
- ISSN
2168-6203
- Publication type
Article
- DOI
10.1001/jamapediatrics.2021.2829