The Methodological Quality and Challenges in Conducting Economic Evaluations of Newborn Screening: A Scoping Review
Introduction: Cost-effectiveness (CEA) and cost–utility analyses (CUA) have become popular types of economic evaluations (EE) used for evidence-based decision-making in healthcare resource allocation. Newborn screening programs (NBS) can have significant clinical benefits for society, and cost-effec...
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doaj-226a99d273e747da920fdc00298da8e92020-11-25T04:12:34ZengMDPI AGInternational Journal of Neonatal Screening2409-515X2020-11-016949410.3390/ijns6040094The Methodological Quality and Challenges in Conducting Economic Evaluations of Newborn Screening: A Scoping ReviewPasquale Cacciatore0Laurenske A. Visser1Nasuh Buyukkaramikli2Catharina P. B. van der Ploeg3M. Elske van den Akker-van Marle4Sezione di Igiene, Istituto di Sanità Pubblica, Università Cattolica del Sacro Cuore, 00168 Rome, ItalyErasmus School of Health Policy & Management, Erasmus University Rotterdam, 3062 PA Rotterdam, The NetherlandsErasmus School of Health Policy & Management, Erasmus University Rotterdam, 3062 PA Rotterdam, The NetherlandsDepartment of Child Health, TNO, 2316 ZL Leiden, The NetherlandsUnit Medical Decision Making, Department of Biomedical Datasciences, Leiden University Medical Center, 2333 ZA Leiden, The NetherlandsIntroduction: Cost-effectiveness (CEA) and cost–utility analyses (CUA) have become popular types of economic evaluations (EE) used for evidence-based decision-making in healthcare resource allocation. Newborn screening programs (NBS) can have significant clinical benefits for society, and cost-effectiveness analysis may help to select the optimal strategy among different screening programs, including the no-screening option, on different conditions. These economic analyses of NBS, however, are hindered by several methodological challenges. This study explored the methodological quality in recent NBS economic evaluations and analyzed the main challenges and strategies adopted by researchers to deal with them. Methods: A scoping review was conducted according to PRISMA methodology to identify CEAs and CUAs of NBS. The methodological quality of the retrieved studies was assessed quantitatively using a specific guideline for the quality assessment of NBS economic evaluations, by calculating a general score for each EE. Challenges in the studies were then explored using thematic analysis as a qualitative synthesis approach. Results: Thirty-five studies met the inclusion criteria. The quantitative analysis showed that the methodological quality of NBS economic evaluations was heterogeneous. Lack of clear description of items related to results, discussion, and discounting were the most frequent flaws. Methodological challenges in performing EEs of neonatal screenings include the adoption of a long time horizon, the use of quality-adjusted life years as health outcome measure, and the assessment of costs beyond the screening interventions. Conclusions: The results of this review can support future economic evaluation research, aiding researchers to develop a methodological guidance to perform EEs aimed at producing solid results to inform decisions for resource allocation in neonatal screening.https://www.mdpi.com/2409-515X/6/4/94economic evaluationsnewborn screeningdecision analysismethodology |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Pasquale Cacciatore Laurenske A. Visser Nasuh Buyukkaramikli Catharina P. B. van der Ploeg M. Elske van den Akker-van Marle |
spellingShingle |
Pasquale Cacciatore Laurenske A. Visser Nasuh Buyukkaramikli Catharina P. B. van der Ploeg M. Elske van den Akker-van Marle The Methodological Quality and Challenges in Conducting Economic Evaluations of Newborn Screening: A Scoping Review International Journal of Neonatal Screening economic evaluations newborn screening decision analysis methodology |
author_facet |
Pasquale Cacciatore Laurenske A. Visser Nasuh Buyukkaramikli Catharina P. B. van der Ploeg M. Elske van den Akker-van Marle |
author_sort |
Pasquale Cacciatore |
title |
The Methodological Quality and Challenges in Conducting Economic Evaluations of Newborn Screening: A Scoping Review |
title_short |
The Methodological Quality and Challenges in Conducting Economic Evaluations of Newborn Screening: A Scoping Review |
title_full |
The Methodological Quality and Challenges in Conducting Economic Evaluations of Newborn Screening: A Scoping Review |
title_fullStr |
The Methodological Quality and Challenges in Conducting Economic Evaluations of Newborn Screening: A Scoping Review |
title_full_unstemmed |
The Methodological Quality and Challenges in Conducting Economic Evaluations of Newborn Screening: A Scoping Review |
title_sort |
methodological quality and challenges in conducting economic evaluations of newborn screening: a scoping review |
publisher |
MDPI AG |
series |
International Journal of Neonatal Screening |
issn |
2409-515X |
publishDate |
2020-11-01 |
description |
Introduction: Cost-effectiveness (CEA) and cost–utility analyses (CUA) have become popular types of economic evaluations (EE) used for evidence-based decision-making in healthcare resource allocation. Newborn screening programs (NBS) can have significant clinical benefits for society, and cost-effectiveness analysis may help to select the optimal strategy among different screening programs, including the no-screening option, on different conditions. These economic analyses of NBS, however, are hindered by several methodological challenges. This study explored the methodological quality in recent NBS economic evaluations and analyzed the main challenges and strategies adopted by researchers to deal with them. Methods: A scoping review was conducted according to PRISMA methodology to identify CEAs and CUAs of NBS. The methodological quality of the retrieved studies was assessed quantitatively using a specific guideline for the quality assessment of NBS economic evaluations, by calculating a general score for each EE. Challenges in the studies were then explored using thematic analysis as a qualitative synthesis approach. Results: Thirty-five studies met the inclusion criteria. The quantitative analysis showed that the methodological quality of NBS economic evaluations was heterogeneous. Lack of clear description of items related to results, discussion, and discounting were the most frequent flaws. Methodological challenges in performing EEs of neonatal screenings include the adoption of a long time horizon, the use of quality-adjusted life years as health outcome measure, and the assessment of costs beyond the screening interventions. Conclusions: The results of this review can support future economic evaluation research, aiding researchers to develop a methodological guidance to perform EEs aimed at producing solid results to inform decisions for resource allocation in neonatal screening. |
topic |
economic evaluations newborn screening decision analysis methodology |
url |
https://www.mdpi.com/2409-515X/6/4/94 |
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