title: Parents' decisions to screen newborns for FMR1 gene expansions in a pilot research project.
abstract: OBJECTIVE
  The 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.
  
  
  METHODS
  A 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.
  
  
  RESULTS
  The 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.
  
  
  CONCLUSIONS
  Findings 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.
authors.affiliation: FPG Child Development Institute, University of North Carolina at Chapel Hill, Carrboro, NC 27510, USA. debra.skinner@unc.edu
authors.forename: Debra
authors.initials: D
authors.lastname: Skinner
authors.forename: Summer
authors.initials: S
authors.lastname: Choudhury
authors.forename: John
authors.initials: J
authors.lastname: Sideris
authors.forename: Sonia
authors.initials: S
authors.lastname: Guarda
authors.forename: Allen
authors.initials: A
authors.lastname: Buansi
authors.forename: Myra
authors.initials: M
authors.lastname: Roche
authors.forename: Cynthia
authors.initials: C
authors.lastname: Powell
authors.forename: Donald B
authors.initials: DB
authors.lastname: Bailey
mesh_terms: D000328:Adult; D003657:Decision Making; D005260:Female; D051860:Fragile X Messenger Ribonucleoprotein 1 / Q000032:analysis*; D005600:Fragile X Syndrome / Q000175:diagnosis* / Q000235:genetics; D005820:Genetic Testing / Q000523:psychology*; D006801:Humans; D007231:Infant, Newborn; D008297:Male; D033282:Parental Consent / Q000941:ethics / Q000523:psychology*; D010290:Parents / Q000523:psychology*; D010865:Pilot Projects; D012189:Retrospective Studies
chemical_list: D051860:Fragile X Messenger Ribonucleoprotein 1; C497683:FMR1 protein, human