Positive parental attitudes to participating in research involving newborn screened infants with CF

• Published in: Journal of cystic fibrosis 2013-05, Vol.12 (3), p.234-240
• Main Authors: Chudleigh, Jane, Hoo, Ah-Fong, Ahmed, Deeba, Prasad, Ammani, Sheehan, Denise, Francis, Jackie, Buckingham, Sarah, Cowlard, Jacqui, Thia, Lena, Nguyen, The Thanh Diem, Stocks, Janet
• Format: Article
• Language: English
• Subjects: Attitude; Biomedical Research; Community Participation; Cystic Fibrosis - diagnosis; Feasibility Studies; Humans; Infant; Infant, Newborn; Neonatal Screening; Newborn screening; Parents - psychology; Patient Selection; Pulmonary function tests; Pulmonary/Respiratory; Recruitment; Surveys and Questionnaires
• ISSN: 1569-1993; 1873-5010
• DOI: 10.1016/j.jcf.2012.09.001

Abstract Background Information regarding recruitment of infants to research studies following the diagnosis of cystic fibrosis (CF) via newborn screening (NBS) is not currently available. This study aimed to assess parental attitudes and the feasibility of recruiting and retaining both NBS infants with CF and healthy control infants to a longitudinal, observational study. Methods All infants underwent pulmonary function tests (PFTs) at ~ 3 and ~ 12 months of age. Infants with CF had additional combined chest high resolution computed tomography (HRCT), bronchoscopy and broncho-alveolar lavage (BAL) at ~ 12 months of age. Parental attitude questionnaires (PAQs) were administered to all parents following the ~ 3 month PFTs and to parents of infants with CF after completion of all tests at ~ 12 months. Results 86% (92/107) of families whose infant had CF consented to participate, of whom 92% had PFTs at ~ 3 months of age with 99% of these having PFTs at ~ 12 months of age. Recruitment of healthy controls was feasible but more challenging; 29% of those contacted agreed to participate; 73% of these had PFTs at ~ 3 months of age; of whom 83% had repeated PFTs at ~ 12 months of age. Completed PAQs were received from 71% of parents, (both of CF and healthy infants) at ~ 3 months and from 58% parents of infants with CF at ~ 12 months. Responses from the PAQs were generally positive, 95% of parents indicated they would recommend participation in such studies to other families. Discrepancies between responses at 3 and 12 months suggested that parental understanding of what the research entailed developed during the course of the study. Conclusions The high recruitment and retention rates for newly diagnosed CF NBS infants to this observational study are encouraging. These findings will help inform future study design both in the field of CF and other conditions diagnosed by NBS.