Bessey, A. orcid.org/0000-0001-7677-0309, Chilcott, J. orcid.org/0000-0003-1231-7817, Pandor, A. orcid.org/0000-0003-2552-5260 et al. (1 more author) (2020) The cost-effectiveness of expanding the UK newborn bloodspot screening programme to include five additional inborn errors of metabolism. International Journal of Neonatal Screening, 6 (4). 93.
Abstract
Glutaric aciduria type 1, homocystinuria, isovaleric acidaemia, long-chain hydroxyacyl CoA dehydrogenase deficiency and maple syrup urine disease are all inborn errors of metabolism that can be detected through newborn bloodspot screening. This evaluation was undertaken in 2013 to provide evidence to the UK National Screening Committee for the cost-effectiveness of including these five conditions in the UK Newborn Bloodspot Screening Programme. A decision-tree model with lifetable estimates of outcomes was built with the model structure and parameterisation informed by a systematic review and expert clinical judgment. A National Health Service/Personal Social Services perspective was used, and lifetime costs and quality-adjusted life years (QALYs) were discounted at 1.5%. Uncertainty in the results was explored using expected value of perfect information analysis methods together with a sensitivity analysis using the screened incidence rate in the UK from 2014 to 2018. The model estimates that screening for all the conditions is more effective and cost saving when compared to not screening for each of the conditions, and the results were robust to the updated incidence rates. The key uncertainties included the sensitivity and specificity of the screening test and the estimated costs and QALYs.
Metadata
Item Type: | Article |
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Authors/Creators: |
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Copyright, Publisher and Additional Information: | © 2020 The Authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/). |
Keywords: | glutaric aciduria type 1; homocystinuria; isovaleric acidaemia; long-chain hydroxyacyl CoA dehydrogenase deficiency; maple syrup urine disease; inborn errors of metabolism; cost-effectiveness; economic; newborn screening; neonatal screening |
Dates: |
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Institution: | The University of Sheffield |
Academic Units: | The University of Sheffield > Faculty of Medicine, Dentistry and Health (Sheffield) > School of Health and Related Research (Sheffield) > ScHARR - Sheffield Centre for Health and Related Research |
Depositing User: | Symplectic Sheffield |
Date Deposited: | 26 Nov 2020 12:03 |
Last Modified: | 26 Nov 2020 12:33 |
Status: | Published |
Publisher: | MDPI AG |
Refereed: | Yes |
Identification Number: | 10.3390/ijns6040093 |
Open Archives Initiative ID (OAI ID): | oai:eprints.whiterose.ac.uk:168443 |