Parent Experience With False-Positive Newborn Screening Results for Cystic Fibrosis.

Autor:	Hayeems RZ; Child Health Evaluative Sciences, Research Institute, The Hospital for Sick Children, Toronto, Canada; Institute of Health Policy, Management and Evaluation, robin.hayeems@sickkids.ca., Miller FA; Institute of Health Policy, Management and Evaluation., Barg CJ; Institute of Health Policy, Management and Evaluation., Bombard Y; Institute of Health Policy, Management and Evaluation, Li Ka Shing Knowledge Institute of St. Michael's Hospital, Toronto, Canada;, Kerr E; Psychology, Departments of Pediatrics and., Tam K; Divisions of Clinical and Metabolic Genetics., Carroll JC; Family and Community Medicine, Sinai Health System, University of Toronto, Toronto, Canada;, Potter BK; School of Epidemiology, Public Health & Preventive Medicine., Chakraborty P; Department of Pediatrics, Faculty of Medicine, University of Ottawa, Ottawa, Canada; Newborn Screening Ontario, Children's Hospital of Eastern Ontario, Ottawa, Canada;, Davies C; Newborn Screening Ontario, Children's Hospital of Eastern Ontario, Ottawa, Canada;, Milburn J; Newborn Screening Ontario, Children's Hospital of Eastern Ontario, Ottawa, Canada;, Patton S; Institute of Health Policy, Management and Evaluation., Bytautas JP; Institute of Health Policy, Management and Evaluation, Department of Public Health, University of Helsinki, Helsinki, Finland;, Taylor L; Pediatric Medicine, and., Price A; Children's Hospital of Western Ontario, London, Ontario, Canada; and., Gonska T; Pediatric Medicine, and., Keenan K; Pediatric Medicine, and., Ratjen F; Departments of Pediatrics and Pediatric Medicine, and Respiratory Medicine, Departments of Pediatrics and., Guttmann A; 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.
Jazyk:	angličtina
Zdroj:	Pediatrics [Pediatrics] 2016 Sep; Vol. 138 (3). Date of Electronic Publication: 2016 Aug 02.
DOI:	10.1542/peds.2016-1052
Abstrakt:	Background: 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. Methods: 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. Results: 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. Conclusions: 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. (Copyright © 2016 by the American Academy of Pediatrics.)
Databáze:	MEDLINE
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