Psychosocial Effect of Newborn Genomic Sequencing on Families in the BabySeq Project: A Randomized Clinical Trial
| Autor: | Heidi L. Rehm, Bethany Zettler, Amy L. McGuire, Sergei Roumiantsev, Dmitry Dukhovny, Kalotina Machini, Talia S. Schwartz, Ozge Ceyhan-Birsoy, Hadley Stevens Smith, Timothy W. Yu, Jill O. Robinson, Devan Petersen, Pankaj B. Agrawal, Chet Graham, Amy E. Roberts, Tiffany T. Nguyen Dolphyn, Tina K. Truong, Maegan Harden, Carrie L. Blout Zawatsky, Casie A. Genetti, Ingrid A. Holm, Shawn Fayer, Xingquan Lu, Harvey L. Levy, Vivek Ramanathan, Richard B. Parad, Leslie A. Frankel, Jaclyn B. Murry, Amanda M. Gutierrez, Wendi N. Betting, Kaitlyn B. Lee, Grace E. VanNoy, Susan E. Waisbren, Robert C. Green, Stacey Pereira, Alan H. Beggs, Matthew S. Lebo, Kurt D. Christensen, Medha Naik, Hayley A. Peoples, Rubaiya Islam, Uma Ramamurthy, Joel B. Krier |
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| Jazyk: | angličtina |
| Rok vydání: | 2021 |
| Předmět: |
Adult
Male Parents Pediatrics medicine.medical_specialty law.invention Neonatal Screening Randomized controlled trial law Intensive care Exome Sequencing Medicine Humans Genetic Predisposition to Disease Parent-Child Relations Family history Child Generalized estimating equation Original Investigation Newborn screening business.industry Infant Newborn Genomics Edinburgh Postnatal Depression Scale Pediatrics Perinatology and Child Health Anxiety Female medicine.symptom business Psychosocial |
| Zdroj: | JAMA Pediatr |
| Popis: | 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]; jointP = .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]; jointP = .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]; jointP = .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 |
| Databáze: | OpenAIRE |
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