The role of information provision in economic evaluations of non-invasive prenatal testing: a systematic review

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ORIGINAL PAPER

The role of information provision in economic evaluations of non‑invasive prenatal testing: a systematic review Nikita M. John1 · Stuart J. Wright1   · Sean P. Gavan1   · Caroline M. Vass1  Received: 6 December 2018 / Accepted: 13 June 2019 © The Author(s) 2019

Abstract Background  Technological progress has led to changes in the antenatal screening programmes, most significantly the introduction of non-invasive prenatal testing (NIPT). The availability of a new type of testing changes the type of information that the parent(s) require before, during and after screening to mitigate anxiety about the testing process and results. Objectives  To identify the extent to which economic evaluations of NIPT have accounted for the need to provide information alongside testing and the associated costs and health outcomes of information provision. Methods  A systematic review of economic evaluations of NIPTs (up to February 2018) was conducted. Medline, Embase, CINAHL and PsychINFO were searched using an electronic search strategy combining a published economic search filter (from NHS economic evaluations database) with terms related to NIPT and screening-related technologies. Data were extracted using the Consolidated Health Economic Evaluation Reporting Standards framework and the results were summarised as part of a narrative synthesis. Results  A total of 12 economic evaluations were identified. The majority of evaluations (n = 10; 83.3%) involved cost effectiveness analysis. Only four studies (33.3%) included the cost of providing information about NIPT in their economic evaluation. Two studies considered the impact of test results on parents’ quality of life by allowing utility decrements for different outcomes. Some studies suggested that the challenges of valuing information prohibited their inclusion in an economic evaluation. Conclusion  Economic evaluations of NIPTs need to account for the costs and outcomes associated with information provision, otherwise estimates of cost effectiveness may prove inaccurate. Keywords  Aneuploidy · Down’s syndrome · NIPT · Prenatal testing · Information · Cost effectiveness JEL Classification  I19 · D61

Introduction Prenatal testing involves the use of a range of tests to screen and diagnose abnormalities in pregnancy. Prenatal “screening” is usually used to determine the risk of delivering a baby with Electronic supplementary material  The online version of this article (https​://doi.org/10.1007/s1019​8-019-01082​-x) contains supplementary material, which is available to authorized users. * Caroline M. Vass [email protected] 1



Manchester Centre for Health Economics, Division of Population Health, Health Services Research and Primary Care, The University of Manchester, Jean McFarlane Building, Oxford Road, Manchester M13 9PL, UK

an abnormality [1]. Prenatal “diagnosis” techniques, such as amniocentesis and chorionic villus sampling (CVS), are then offered to confirm or reject a diagnosis of foetal abnormalities [1]. Conditions that can be detecte