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An analysis of disease severity based on SMN2 copy number in adults with spinal muscular atrophy

Authors :
Xiaoli Zhang
Wendy King
Deborah F. Gelinas
Bakri Elsheikh
Robert G. Miller
Thomas W. Prior
Stephen J. Kolb
Richard J. Barohn
D. A N Moore
Jerry R. Mendell
Barry S. Russman
Walter G. Bradley
Susan T. Iannaccone
Michelle Mendoza
Stephen W. Smith
John T. Kissel
Wilson W. Bryan
Robert T. Leshner
Source :
Muscle & Nerve. 40:652-656
Publication Year :
2009
Publisher :
Wiley, 2009.

Abstract

To evaluate the effect of SMN2 copy number on disease severity in spinal muscular atrophy (SMA), we stratified 45 adult SMA patients based on SMN2 copy number (3 vs. 4 copies). Patients with 3 copies had an earlier age of onset and lower spinal muscular atrophy functional rating scale (SMAFRS) scores and were more likely to be non-ambulatory. There was, however, no difference between the groups in quantitative muscle strength or pulmonary function testing. Functional scale may be a more discriminating outcome measure for SMA clinical trials.

Details

ISSN :
10974598 and 0148639X
Volume :
40
Database :
OpenAIRE
Journal :
Muscle & Nerve
Accession number :
edsair.doi.dedup.....e1d1a2c6fc06db6e976ce6e9a5e7844f
Full Text :
https://doi.org/10.1002/mus.21350