Literature DB >> 15178985

Explorative study of costs, effects and savings of screening for female fragile X premutation and full mutation carriers in the general population.

M F Wildhagen1, T A van Os, J J Polder, L P ten Kate, J D Habbema.   

Abstract

OBJECTIVE: Evaluation of the costs, effects and savings of three strategies for female fragile X premutation and full mutation carrier screening in the general population.
METHODS: We calculated the costs, effects and savings by using a general model for prenatal, preconceptional, and school carrier screening. Assumptions were based on literature data, expert opinions, prices and tariffs.
RESULTS: Prenatal screening will detect most carriers and will lead to the highest number of avoided fragile X syndrome patients. The costs per detected carrier are quite similar for all screening programmes (around USD 45,000). All screening strategies have a favourable cost-savings balance (USD 14 million for prenatal screening, USD 9 million for preconceptional screening and USD 2 million for school screening).
CONCLUSIONS: From an economic point of view, there is no obstacle to fragile X screening. The decision to screen or not can (and should) therefore concentrate on discussion of medical, social, psychological and ethical considerations.

Entities:  

Year:  1998        PMID: 15178985     DOI: 10.1159/000016133

Source DB:  PubMed          Journal:  Community Genet        ISSN: 1422-2795


  7 in total

1.  Carrier screening in preconception consultation in primary care.

Authors:  Sylvia A Metcalfe
Journal:  J Community Genet       Date:  2011-12-20

2.  Noninvasive test for fragile X syndrome, using hair root analysis.

Authors:  R Willemsen; B Anar; Y De Diego Otero; B B de Vries; Y Hilhorst-Hofstee; A Smits; E van Looveren; P J Willems; H Galjaard; B A Oostra
Journal:  Am J Hum Genet       Date:  1999-07       Impact factor: 11.025

3.  Fragile-X carrier screening and the prevalence of premutation and full-mutation carriers in Israel.

Authors:  H Toledano-Alhadef; L Basel-Vanagaite; N Magal; B Davidov; S Ehrlich; V Drasinover; E Taub; G J Halpern; N Ginott; M Shohat
Journal:  Am J Hum Genet       Date:  2001-07-06       Impact factor: 11.025

4.  Prenatal screening for fragile x: carriers, controversies, and counseling.

Authors:  Julie F Gutiérrez; Komal Bajaj; Susan D Klugman
Journal:  Rev Obstet Gynecol       Date:  2013

5.  Social/economic costs and health-related quality of life in patients with fragile X syndrome in Europe.

Authors:  Karine Chevreul; Coralie Gandré; Karen Berg Brigham; Julio López-Bastida; Renata Linertová; Juan Oliva-Moreno; Pedro Serrano-Aguilar; Manuel Posada-de-la-Paz; Domenica Taruscio; Arrigo Schieppati; Georgi Iskrov; László Gulácsi; Johann Matthias Graf von der Schulenburg; Panos Kanavos; Ulf Persson; Giovani Fattore
Journal:  Eur J Health Econ       Date:  2016-04-12

Review 6.  FMR1 and the fragile X syndrome: human genome epidemiology review.

Authors:  D C Crawford; J M Acuña; S L Sherman
Journal:  Genet Med       Date:  2001 Sep-Oct       Impact factor: 8.822

Review 7.  Evaluating Cost-effectiveness of Interventions That Affect Fertility and Childbearing: How Health Effects Are Measured Matters.

Authors:  Jeremy D Goldhaber-Fiebert; Margaret L Brandeau
Journal:  Med Decis Making       Date:  2015-04-29       Impact factor: 2.749

  7 in total

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