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1.
Adv Exp Med Biol ; 1031: 267-281, 2017.
Artigo em Inglês | MEDLINE | ID: mdl-29214578

RESUMO

Nowadays, health funding decisions must be supported by sound arguments in terms of both effectiveness and economic criteria. After more than half a century of newborn screening for rare diseases, the appropriate economic evaluation framework for these interventions is still challenging. The validity of standard methods for economic evaluation heavily relies on the availability of robust evidence, but collection of such evidence is precluded by the rareness of the conditions that may benefit from screening. Furthermore, there are a series of conceptual and methodological limitations that warrant further careful consideration when assessing the cost-effectiveness of newborn screening programs. In this chapter we provide a general overview of current economic evaluation methods and the challenges for their application to newborn screening programs.


Assuntos
Custos de Cuidados de Saúde , Triagem Neonatal/economia , Triagem Neonatal/métodos , Doenças Raras/diagnóstico , Doenças Raras/economia , Deficiência de Biotinidase/diagnóstico , Deficiência de Biotinidase/economia , Deficiência de Biotinidase/terapia , Análise Custo-Benefício , Humanos , Incidência , Recém-Nascido , Modelos Econômicos , Valor Preditivo dos Testes , Prevalência , Prognóstico , Anos de Vida Ajustados por Qualidade de Vida , Doenças Raras/terapia
2.
Pediatrics ; 136(2): e424-32, 2015 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-26169436

RESUMO

BACKGROUND AND OBJECTIVES: There are conflicting views as to whether testing for biotinidase deficiency (BD) ought to be incorporated into universal newborn screening (NBS) programs. The aim of this study was to evaluate the cost-effectiveness of adding BD to the panel of conditions currently screened under the national NBS program in Spain. METHODS: We used information from the regional NBS program for BD that has been in place in the Spanish region of Galicia since 1987. These data, along with other sources, were used to develop a cost-effectiveness decision model that compared lifetime costs and health outcomes of a national birth cohort of newborns with and without an early detection program. The analysis took the perspective of the Spanish National Health Service. Effectiveness was measured in terms of quality-adjusted life years (QALYs). We undertook extensive sensitivity analyses around the main model assumptions, including a probabilistic sensitivity analysis. RESULTS: In the base case analysis, NBS for BD led to higher QALYs and higher health care costs, with an estimated incremental cost per QALY gained of $24,677. Lower costs per QALY gained were found when conservative assumptions were relaxed, yielding cost savings in some scenarios. The probability that BD screening was cost-effective was estimated to be >70% in the base case at a standard threshold value. CONCLUSIONS: This study indicates that NBS for BD is likely to be a cost-effective use of resources.


Assuntos
Deficiência de Biotinidase/diagnóstico , Deficiência de Biotinidase/economia , Triagem Neonatal/economia , Análise Custo-Benefício , Árvores de Decisões , Humanos , Recém-Nascido , Programas Nacionais de Saúde , Espanha
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