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Reporting Adult-Onset Genomic Results to Pediatric Biobank Participants and Parents

Award Number: R01HG009671
ORGANIZATION: NATIONAL HUMAN GENOME RESEARCH INSTITUTE
OPDIV: NIH
AWARD CLASS: DISCRETIONARY
AWARD ACTIVITY TYPE: SCIENTIFIC/HEALTH RESEARCH (INCLUDES SURVEYS)
PERIOD OF PERFORMANCE START DATE: 09/19/2018
PERIOD OF PERFORMANCE END DATE: 06/30/2024

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Reporting Adult-Onset Genomic Results to Pediatric Biobank Participants and Parents - Project Summary/Abstract The potential benefits and harms of returning genomic results to children and their parents are matters of enduring controversy—especially genomic results for adult-onset conditions that are not medically actionable in childhood. Returning results for adult-onset conditions can spur life-saving preventive measures in the parents of affected children. However, there has been long-standing concern that children who receive a result for an adult-onset condition might experience negative psychosocial outcomes such as distress or altered family functioning. The lack of empirical data to support either position in this controversy prevents policy makers from making evidence-based judgments that balance benefits and harms. We propose to provide this urgently needed data by returning adult- and pediatric-onset genomic results through a supportive clinical program and assessing the associated psychosocial, behavioral and legal impact. Using our experience in returning medically actionable genomic findings to adults in Geisinger's MyCode Community Health Initiative, and with support from pediatric and adult clinical psychologists and clinician researchers with experience with adolescents at increased cancer risk, we will return results for two adult-onset and 28 pediatric-onset genomic conditions to MyCode pediatric participants and parents. We will conduct a longitudinal, mixed-methods cohort study to assess psychosocial outcomes and health behaviors among participants: with a pathogenic variant in an adult-onset gene (Group 1); with a pathogenic variant in a pediatric-onset gene (Group 2); and without a genomic result (Group 3). This study design will allow us to address three critical research questions: 1) Do children and parents who receive an adult-onset genomic result via a supportive clinical interaction have poorer psychosocial outcomes than their counterparts who receive a pediatric-onset result or no genomic result? 2) How does the receipt of children's genomic information impact their parents' cascade testing and risk reduction decisions? and 3) Does the loss of chance tort doctrine (which allows for recovery of damages when breach of duty caused a reduction in the chance of a favorable outcome) provide an applicable legal framework for deciding whether to return adult-onset genomic findings to children? Data will be gathered longitudinally via quantitative surveys using validated measures of distress, family functioning, and quality of life; qualitative interviews with children and parents in Groups 1 and 2; electronic health records review of parents' uptake of cascade testing and initiation of disease risk reduction procedures; and legal review of relevant case law and legislation. Quantitative analyses will determine whether change in psychosocial and behavioral outcomes from pre- to post-disclosure differs significantly among groups. Qualitative analyses will allow for comparison of themes among those with an adult-onset vs. a pediatric-onset result. Data from this innovative landmark study will guide national and international groups planning to perform genome-scale testing of children, as well as other groups considering population screening for genomic conditions.


Issue Date FY	Funding FY	Legal Entity Name	Legal Entity Address	Legal Entity City	Legal Entity State	Legal Entity Zip Code	Legal Entity COUNTY	Legal Entity COUNTRY	Assistance Listing	Award Code	Budget Year	Action Date	Action Type	Action Amount

Issue Date FY: 2025 ( Subtotal = -$1,723 )
2025	2022	GEISINGER CLINIC	100 N ACADEMY AVE # MC4970	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	000	5	11/1/2024	NON-COMPETING CONTINUATION	-$1,723
														Subtotal = -$1,723

Issue Date FY: 2023 ( Subtotal = $0 )
2023	2022	GEISINGER CLINIC	100 N ACADEMY AVE	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	000	5	2/16/2023	NON-COMPETING CONTINUATION	$0
														Subtotal = $0

Issue Date FY: 2022 ( Subtotal = $591,298 )
2022	2022	GEISINGER CLINIC	100 N ACADEMY AVE	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	000	5	6/24/2022	NON-COMPETING CONTINUATION	$591,298
														Subtotal = $591,298

Issue Date FY: 2021 ( Subtotal = $627,524 )
2021	2021	GEISINGER CLINIC	100 N ACADEMY AVE	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	001	4	8/19/2021	NON-COMPETING CONTINUATION	$0
2021	2021	GEISINGER CLINIC	100 N ACADEMY AVE	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	000	4	7/15/2021	NON-COMPETING CONTINUATION	$627,524
														Subtotal = $627,524

Issue Date FY: 2020 ( Subtotal = $698,079 )
2020	2020	GEISINGER CLINIC	100 N ACADEMY AVE	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	000	3	6/19/2020	NON-COMPETING CONTINUATION	$698,079
														Subtotal = $698,079

Issue Date FY: 2019 ( Subtotal = $804,822 )
2019	2019	GEISINGER CLINIC	100 N ACADEMY AVE	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	001	2	6/11/2019	NON-COMPETING CONTINUATION	$804,822
2019	2018	GEISINGER CLINIC	100 N ACADEMY AVE	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	000	1	11/16/2018	NEW	$0
														Subtotal = $804,822

Issue Date FY: 2018 ( Subtotal = $819,711 )
2018	2018	GEISINGER CLINIC	100 N ACADEMY AVE	DANVILLE	PA	17822	MONTOUR	USA	Human Genome Research	000	1	9/19/2018	NEW	$819,711
														Subtotal = $819,711

Grand Total All Awards = $3,539,711

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Reporting Adult-Onset Genomic Results to Pediatric Biobank Participants and Parents

Award Number: R01HG009671

ORGANIZATION: NATIONAL HUMAN GENOME RESEARCH INSTITUTE

OPDIV: NIH

AWARD CLASS: DISCRETIONARY

AWARD ACTIVITY TYPE: SCIENTIFIC/HEALTH RESEARCH (INCLUDES SURVEYS)

PERIOD OF PERFORMANCE START DATE: 09/19/2018

PERIOD OF PERFORMANCE END DATE: 06/30/2024

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