WS21.2 Newborn screening for cystic fibrosis with an IRT-PAP protocol

Abstract

Objectives: 1. Assess feasibility of recruiting and retaining a. NBS infants with CF to an invasive observational study involving pulmonary function tests (PFTs), CT and bronchoscopy, b. healthy controls (HC) to a longitudinal study of PFTs. 2. Investigate parental attitudes to participating in such a study. Methods: PFTs were undertaken at 3 mths and 1 yr in both CF and healthy infants between Jan 2010-Dec 2011. CT and bronchoscopy was also undertaken in those with CF at 1 yr. Parental Attitude Questionnaires (PAQ) were administered to all parents at the 3mth PFT. A further questionnaire was completed anonymously by parents of CF infants following the 1 yr assessments. Results: Of 107 eligible CF infants, 79% (84) attended the 3 mth PFT. By Dec 2011, 72 were at least 1 yr of age, of whom 90% (65) completed the 1 yr assessments. Of the 270 HC invited to participate, 21% (56) had PFTs at 3mth. By Dec 2011, 46 were at least 1 yr of age, of whom 78% (36) completed PFTs. Responses from the PAQ indicated that most parents of infants with CF chose to be involved in the study to help their own and other infants in the future. Parents of HC cited altruistic reasons. Most (95%) parents of CF infants would recommend involvement in research to other parents. Conclusions: Recruitment and retention of infants with CF was excellent, providing encouraging data for future interventional trials. While recruitment of HC was more difficult, retention was similar. Parental attitudes were very positive, with the majority of parents willing to be involved in such studies for the good of their own and other children.