Journal of Inherited Metabolic Disease

, Volume 35, Issue 1, pp 169–176 | Cite as

Putting a value on the avoidance of false positive results when screening for inherited metabolic disease in the newborn

  • Simon Dixon
  • Phil Shackley
  • Jim Bonham
  • Rachel Ibbotson
Original Article

Abstract

Despite the increase in the number of inherited metabolic diseases that can be detected at birth using a single dried blood spot sample, the impact of false positive results on parents remains a concern. We used an economic approach - the contingent valuation method – which asks parents to give their maximum willingness to pay for an extension in a screening programme and the degree to which the potential for false positive results diminishes their valuations. 160 parents of a child or children under the age of 16 years were surveyed and given descriptions of the current screening programme in the UK, an extended programme and an extended programme with no false positives. 148 (92.5%) respondents said they would accept the screen for the five extra conditions in an expanded screening programme whilst 10 (6.3%) said they would not and two were unsure. When asked to indicate if they would choose to be screened under an expanded screening programme with no false positive results, 152 (95%) said they would, five (3.1%) said they would not, two were unsure, and there was one non-response. 151 (94.4%) said they preferred the hypothetical test with no false-positives. The mean willingness to pay for the expanded programme was £178 compared to £219 for the hypothetical expanded programme without false positives (p > 0.05). The results suggest that there is widespread parental support for extended screening in the UK and that the number of false-positives is a relatively small issue.

References

  1. Arrow K et al (1993) Report of the NOAA Panel on Contingent Valuation. Fed Regist 58(10):4601–4614Google Scholar
  2. Bodamer OA, Hoffmann GF, Lindner M (2007) Expanded screening in Europe 2007. J Inherit Metab Dis 30:439–444PubMedCrossRefGoogle Scholar
  3. Cookson R (2003) Willingness to pay methods in health care: a sceptical view. Health Econ 12:891–894PubMedCrossRefGoogle Scholar
  4. Dixon S, Shackley P (2003) The use of willingness to pay to assess public preferences towards the fortification of foodstuffs with folic acid. Health Expect 6:140–148PubMedCrossRefGoogle Scholar
  5. Gold M, Siegel J, Russell L, Weinstein M (1996) Cost-effectiveness in health and medicine. Oxford University Press, New YorkGoogle Scholar
  6. Hewlett J, Waisbren SE (2006) A review of the psychosocial effects of false-positive results on parents and current communication practices in newborn screening. J Inherit Metab Dis 29:677–682PubMedCrossRefGoogle Scholar
  7. Kai J, Ulph F, Cullinan T, Qureshi N (2009) Communication of carrier status information following universal newborn screening for sickle cell disorders and cystic fibrosis: qualitative study of experience and practice. Health Technol Assess 13(57):1–82PubMedGoogle Scholar
  8. Lloyd A (2003) Threats to the estimation of benefit: are preferences elicitation methods accurate. Health Econ 12:393–402PubMedCrossRefGoogle Scholar
  9. National Institute for Health and Clinical Excellence (2008) Guide to the Methods of Technology Appraisal. National Institute for Health and Clinical Excellence, LondonGoogle Scholar
  10. Olsen J, Donaldson C, Pereira J (2004) The insensitivity of 'willingness-to-pay' to the size of the good: new evidence for health care. J Econ Psychol 25:445–460CrossRefGoogle Scholar
  11. Prosser LA, Ladapo JA, Rusinak D, Waisbren SE (2008) Parental tolerance of false-positive newborn screening results. Arch Pediatr Adolesc Med 162(9):870–876PubMedCrossRefGoogle Scholar
  12. Ryan M, Farrar S (2000) Using conjoint analysis to elicit preferences for health care. BMJ 320:1530–1533PubMedCrossRefGoogle Scholar
  13. Ryan M, Scott DA, Donaldson C (2004) Valuing health care using willingness to pay: a comparison of the payment card and dichotomous choice methods. J Health Econ 23(2):237–258PubMedCrossRefGoogle Scholar
  14. Shackley P, Dixon S (2000) Using contingent valuation to elicit public preferences for water fluoridation. Appl Econ 32:777–787CrossRefGoogle Scholar
  15. Therrell B, Adams J (2007) Newborn screening in North America. J Inherit Metab Dis 30:447–465PubMedCrossRefGoogle Scholar
  16. Waisbren SE, Albers S, Amato S et al (2003) Effect of expanded newborn screening for biochemical genetic disorders on child outcomes and parental stress. JAMA 290:2564–2572PubMedCrossRefGoogle Scholar

Copyright information

© SSIEM and Springer 2011

Authors and Affiliations

  • Simon Dixon
    • 1
  • Phil Shackley
    • 1
  • Jim Bonham
    • 2
  • Rachel Ibbotson
    • 3
  1. 1.School of Health and Related ResearchUniversity of SheffieldSheffieldUK
  2. 2.Sheffield Children’s Hospital NHS Foundation TrustSheffieldUK
  3. 3.Centre for Health and Social Care ResearchSheffield Hallam UniversitySheffieldUK

Personalised recommendations