Imparting carrier status results detected by universal newborn screening for sickle cell and cystic fibrosis in England: a qualitative study of current practice and policy challenges

BMC Health Serv Res. 2007 Dec 13:7:203. doi: 10.1186/1472-6963-7-203.

Abstract

Background: Universal newborn screening for early detection of children affected by sickle cell disorders and cystic fibrosis is currently being implemented across England. Parents of infants identified as carriers of these disorders must also be informed of their baby's result. However there is a lack of evidence for most effective practice internationally when doing so. This study describes current or proposed models for imparting this information in practice and explores associated challenges for policy.

Methods: Thematic analysis of semi-structured interviews with Child Health Coordinators from all English Health Regions.

Results: Diverse methods for imparting carrier results, both within and between regions, and within and between conditions, were being implemented or planned. Models ranged from result by letter to in-person communication during a home visit. Non-specialists were considered the best placed professionals to give results and a similar approach for both conditions was emphasised. While national guidance has influenced choice of models, other factors contributed such as existing service structures and lack of funding. Challenges included uncertainty about guidance specifying face to face notification; how best to balance allaying parental anxiety by using familiar non-specialist health professionals with concerns about practitioner competence; and extent of information parents should be given. Inadequate consideration of resource and service workload was seen as the main policy obstacle. Clarification of existing guidance; more specific protocols to ensure consistent countrywide practice; integration of the two programmes; and 'normalising' carrier status were suggested as improvements.

Conclusion: Differing models for communicating carrier results raise concerns about equity and clinical governance. However, this variation provides opportunity for evaluation. Timely and more detailed guidance on protocols with clarification of existing recommendations is needed.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Carrier State*
  • Cystic Fibrosis / diagnosis
  • Cystic Fibrosis / genetics*
  • England
  • Genetic Testing* / methods
  • Health Policy*
  • Humans
  • Infant, Newborn
  • Models, Organizational
  • National Health Programs*
  • Neonatal Screening* / methods
  • Parental Notification
  • Parents / psychology*
  • Professional-Family Relations*
  • Sickle Cell Trait / diagnosis
  • Sickle Cell Trait / genetics*
  • Truth Disclosure*