title: Parent Experience With False-Positive Newborn Screening Results for Cystic Fibrosis.
abstract: The risk of psychosocial harm in families of infants with false-positive (FP) newborn bloodspot screening (NBS) results for cystic fibrosis (CF) is a longstanding concern. Whether well designed retrieval and confirmatory testing systems can mitigate risks remains unknown.
  
  
  
  Using a mixed-methods cohort design, we obtained prospective self-report data from mothers of infants with FP CF NBS results 2 to 3 months after confirmatory testing at Ontario's largest follow-up center, and from a randomly selected control sample of mothers of screen negative infants from the same region. Mothers completed a questionnaire assessing experience and psychosocial response. A sample of mothers of FP infants completed qualitative interviews.
  
  
  
  One hundred thirty-four mothers of FP infants (response rate, 55%) and 411 controls (response rate, 47%) completed questionnaires; 54 mothers of FP infants were interviewed. Selected psychosocial response measures did not detect psychosocial distress in newborns or 1 year later (P > .05). Mothers recalled distress during notification of the positive result and in the follow-up testing period related to fear of chronic illness, but valued the screening system of care in mitigating concerns.
  
  
  
  Although immediate distress was reported among mothers of FP infants, selected psychometric tools did not detect these concerns. The NBS center from which mothers were recruited minimizes delay between notification and confirmatory testing and ensures trained professionals are communicating results and facilitating follow-up. These factors may explain the presence of minimal psychosocial burden. The screening system reflected herein may be a model for NBS programs working to minimize FP-related psychosocial harm.
authors.affiliation: Child Health Evaluative Sciences, Research Institute, The Hospital for Sick Children, Toronto, Canada; Institute of Health Policy, Management and Evaluation, robin.hayeems@sickkids.ca.
authors.forename: Robin Z
authors.initials: RZ
authors.lastname: Hayeems
authors.affiliation: Institute of Health Policy, Management and Evaluation.
authors.forename: Fiona A
authors.initials: FA
authors.lastname: Miller
authors.affiliation: Institute of Health Policy, Management and Evaluation.
authors.forename: Carolyn J
authors.initials: CJ
authors.lastname: Barg
authors.affiliation: Institute of Health Policy, Management and Evaluation, Li Ka Shing Knowledge Institute of St. Michael's Hospital, Toronto, Canada;
authors.forename: Yvonne
authors.initials: Y
authors.lastname: Bombard
authors.affiliation: Psychology, Departments of Pediatrics and.
authors.forename: Elizabeth
authors.initials: E
authors.lastname: Kerr
authors.affiliation: Divisions of Clinical and Metabolic Genetics.
authors.forename: Karen
authors.initials: K
authors.lastname: Tam
authors.affiliation: Family and Community Medicine, Sinai Health System, University of Toronto, Toronto, Canada;
authors.forename: June C
authors.initials: JC
authors.lastname: Carroll
authors.affiliation: School of Epidemiology, Public Health & Preventive Medicine.
authors.forename: Beth K
authors.initials: BK
authors.lastname: Potter
authors.affiliation: Department of Pediatrics, Faculty of Medicine, University of Ottawa, Ottawa, Canada; Newborn Screening Ontario, Children's Hospital of Eastern Ontario, Ottawa, Canada;
authors.forename: Pranesh
authors.initials: P
authors.lastname: Chakraborty
authors.affiliation: Newborn Screening Ontario, Children's Hospital of Eastern Ontario, Ottawa, Canada;
authors.forename: Christine
authors.initials: C
authors.lastname: Davies
authors.affiliation: Newborn Screening Ontario, Children's Hospital of Eastern Ontario, Ottawa, Canada;
authors.forename: Jennifer
authors.initials: J
authors.lastname: Milburn
authors.affiliation: Institute of Health Policy, Management and Evaluation.
authors.forename: Sarah
authors.initials: S
authors.lastname: Patton
authors.affiliation: Institute of Health Policy, Management and Evaluation, Department of Public Health, University of Helsinki, Helsinki, Finland;
authors.forename: Jessica P
authors.initials: JP
authors.lastname: Bytautas
authors.affiliation: Pediatric Medicine, and.
authors.forename: Louise
authors.initials: L
authors.lastname: Taylor
authors.affiliation: Children's Hospital of Western Ontario, London, Ontario, Canada; and.
authors.forename: April
authors.initials: A
authors.lastname: Price
authors.affiliation: Pediatric Medicine, and.
authors.forename: Tanja
authors.initials: T
authors.lastname: Gonska
authors.affiliation: Pediatric Medicine, and.
authors.forename: Katherine
authors.initials: K
authors.lastname: Keenan
authors.affiliation: Departments of Pediatrics and Pediatric Medicine, and Respiratory Medicine, Departments of Pediatrics and.
authors.forename: Felix
authors.initials: F
authors.lastname: Ratjen
authors.affiliation: Child Health Evaluative Sciences, Research Institute, The Hospital for Sick Children, Toronto, Canada; Institute of Health Policy, Management and Evaluation, Departments of Pediatrics and Pediatric Medicine, and Institute for Clinical Evaluative Sciences, Toronto, Canada.
authors.forename: Astrid
authors.initials: A
authors.lastname: Guttmann
mesh_terms: D000328:Adult; D016022:Case-Control Studies; D003550:Cystic Fibrosis / Q000175:diagnosis*; D005189:False Positive Reactions; D005260:Female; D006801:Humans; D007223:Infant; D007231:Infant, Newborn; D009035:Mothers / Q000523:psychology*; D015997:Neonatal Screening; D009864:Ontario; D011446:Prospective Studies; D013315:Stress, Psychological / Q000209:etiology; D011795:Surveys and Questionnaires