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Please use this identifier to cite or link to this item: http://hdl.handle.net/2262/53330

Title: Rate of familial amyotrophic lateral sclerosis: a systematic review and meta-analysis.
Author: HARDIMAN, ORLA
MC LAUGHLIN, RUSSELL
BEDE, PETER
WALSH, CATHAL DOMINIC
Sponsor: Health Research Board
Author's Homepage: http://people.tcd.ie/walshc
http://people.tcd.ie/hardimao
http://people.tcd.ie/mclaugr
http://people.tcd.ie/pbede
Keywords: Clinical Medicine
applied statistics
familial amyotrophic lateral sclerosis
Issue Date: 2011
Citation: Byrne S, Walsh C, Lynch C, Bede P, Elamin M, Kenna K, McLaughlin R, Hardiman O, Rate of familial amyotrophic lateral sclerosis: a systematic review and meta-analysis., Journal of neurology, neurosurgery, and psychiatry, 82, 6, 2011, 623 - 627
Series/Report no.: Journal of neurology, neurosurgery, and psychiatry
82
6
Abstract: Background The population rate of familial amyotrophic lateral sclerosis (FALS) is frequently reported as 10%. However, a systematic review and meta-analysis of the true population based frequency of FALS has never been performed. Method A Medline literature review identified all original articles reporting a rate of FALS. Studies were grouped according to the type of data presented and examined for sources of case ascertainment. A systematic review and meta-analysis of reported rates of FALS was then conducted to facilitate comparison between studies and calculate a pooled rate of FALS. Results 38 papers reported a rate of FALS. Thirty-three papers were included in analysis and the rate of FALS for all studies was 4.6% (95% CI 3.9% to 5.5%). Restricting the analysis to prospective population based registry data revealed a rate of 5.1% (95% CI 4.1% to 6.1%). The incidence of FALS was lower in southern Europe. There was no correlation between rate of FALS and reported SOD1 mutation rates. Conclusion The rate of FALS among prospective population based registries is 5.1% (CI 4.1 to 6.1%), and not 10% as is often stated. Further detailed prospective population based studies of familial ALS are required to confirm this rate.
Description: PUBLISHED
URI: http://hdl.handle.net/2262/53330
Related links: http://dx.doi.org/10.1136/jnnp.2010.224501
Appears in Collections:Statistics (Scholarly Publications)

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