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Incidence of Fragile X syndrome in Ireland

Authors :
Deirdre E. Donnelly
David E. Barton
Michael Sweeney
Deborah M. Lambert
Sally Ann Lynch
Celine M. Gervin
James J. O'Byrne
Eleanor D. Beattie
Source :
American Journal of Medical Genetics Part A. 173:678-683
Publication Year :
2017
Publisher :
Wiley, 2017.

Abstract

Described as the commonest single gene cause of learning disability internationally, the incidence of Fragile X syndrome (FXS) has never previously been determined in Ireland. The aim of this work was to determine the observed incidence of FXS in the island of Ireland; the Republic of Ireland (ROI) and Northern Ireland (NI) separately and combined. Ascertainment was achieved for a cross-sectional study by a retrospective, clinical and laboratory database review of positive FXS cases, born in either ROI or NI, between years 2000-2009 inclusive. The observed incidence of FXS per 10,000 live births in the island of Ireland in males was 0.94 (95%CI: 0.75-1.13) or ∼1:10,600 and in females was 0.23 (95%CI: 0.14-0.32) or ∼1:43,000. Comparable testing rates for FXS are present in ROI and NI, with on average 1.48% (1.30% in ROI, 1.96% in NI) of live male births and 0.4% (0.35% in ROI, 0.55% in NI) of live female births undergoing analysis which is comparable to other centres internationally. This study demonstrates the observed incidence of FXS in the island of Ireland is (i) approximately half the estimated worldwide incidence in males and is not explained by low levels of testing, and (ii) approximately one quarter the estimated worldwide incidence in females which may be explained by low levels of testing. © 2017 Wiley Periodicals, Inc.

Details

ISSN :
15524825 and 20002009
Volume :
173
Database :
OpenAIRE
Journal :
American Journal of Medical Genetics Part A
Accession number :
edsair.doi.dedup.....b352f538f2b7d32a50ad9ec7cf49bef8
Full Text :
https://doi.org/10.1002/ajmg.a.38081