ArticlePediatrics2011
Parents' decisions to screen newborns for FMR1 gene expansions in a pilot research project.
Article in Pediatrics, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers, 2 of them syntheses that pooled it.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
26 citing papers in PubMed, 2 syntheses or guidelines pooled it.
- Parental Views of Facilitators and Barriers to Research Participation: Systematic Review.Pediatrics · 2023Pooled it
- Acceptability of childhood screening: a systematic narrative review.Public health · 2021Pooled it
- A qualitative study exploring young adult's attitudes towards adopting whole genome sequencing into newborn screening programs.European journal of human genetics : EJHG · 2026Article
- Psychosocial Issues Related to Newborn Screening: A Systematic Review and Synthesis.International journal of neonatal screening · 2022Review
- Attitudes toward population screening among people living with fragile X syndrome in the UK: 'I wouldn't wish him away, I'd just wish his fragile X syndrome away'.Journal of genetic counseling · 2021Article
- Parents' Perspectives and Societal Acceptance of Implementation of Newborn Screening for SCID in the Netherlands.Journal of clinical immunology · 2021Article
- "If He Has it, We Know What to Do": Parent Perspectives on Familial Risk for Autism Spectrum Disorder.Journal of pediatric psychology · 2020Article
- Ethical Dilemmas Linked to Fragile X Testing of Minors-a Preliminary Survey Among Professionals.Journal of molecular neuroscience : MN · 2020Article
- Parental interest in genomic sequencing of newborns: enrollment experience from the BabySeq Project.Genetics in medicine : official journal of the American College of Medical Genetics · 2019Article
- Molecular Correlates and Recent Advancements in the Diagnosis and Screening of FMR1-Related Disorders.Genes · 2016Review
- Biobank participant support of newborn screening for disorders with variable treatment and intervention options.Journal of community genetics · 2016Article
- Family Communication and Cascade Testing for Fragile X Syndrome.Journal of genetic counseling · 2016Article
- Developmental profiles of infants with anJournal of neurodevelopmental disorders · 2016Article
- Identification of a male with fragile X syndrome through newborn screening.Intractable & rare diseases research · 2015Article
- Article
- Framing optional genetic testing in the context of mandatory newborn screening tests.BMC medical informatics and decision making · 2015Article
- A mixed methods exploration of families' experiences of the diagnosis of childhood spinal muscular atrophy.European journal of human genetics : EJHG · 2015Article
- Fragile X protein in newborn dried blood spots.BMC medical genetics · 2014Article
- Longitudinal interviews of couples diagnosed with diminished ovarian reserve undergoing fragile X premutation testing.Journal of genetic counseling · 2014Article
- Developmental and behavioral pediatricians' attitudes toward screening for fragile X.American journal on intellectual and developmental disabilities · 2013Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
8 authors.
Funding
Abstract
objectiveThe goal of this study was to document rates of parental consent in a pilot study of newborn screening for FMR1 gene expansions, examine demographic characteristics of mothers who consented or declined, describe the reasons for their decision, and discuss ethical and social aspects of the consent process.
methodsA brief survey was used to record basic demographic data from mothers and an open-ended question was used to elicit parents' reasons for accepting or declining screening. A descriptive analysis was conducted on the number of mothers who consented to or declined screening, and a logistic regression model predicted mothers' likelihood to agree to screening based on demographic characteristics. Reasons for decisions were analyzed using content analysis. The study was conducted at University of North Carolina Hospitals. A total of 2137 mothers were approached.
resultsThe uptake rate for couples was 63%. Acceptance rates varied by race/ethnicity, with black respondents being less likely to accept screening. Primary reasons for accepting were "to know," "belief in research," and "the test was minimal/no risk." Reasons for declining included not wanting to know or worry, not being a good time, and issues with testing children or with genetic tests.
conclusionsFindings demonstrate that a majority of parents accepted newborn screening for FMR1 gene expansions, but decision rates and reasons for accepting or declining varied in part as a function of race/ethnicity and in part as a function of what parents most valued or feared in their assessment of risks and benefits.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.