Longitudinal change in dysarthria associated with Friedreich ataxia: a potential clinical endpoint

Rosen, Kristin M., Folker, Joanne E., Vogel, Adam P., Corben, Louise A., Murdoch, Bruce E. and Delatycki, Martin B. (2012) Longitudinal change in dysarthria associated with Friedreich ataxia: a potential clinical endpoint. Journal of Neurology, 259 11: 2471-2477. doi:10.1007/s00415-012-6547-x


Author Rosen, Kristin M.
Folker, Joanne E.
Vogel, Adam P.
Corben, Louise A.
Murdoch, Bruce E.
Delatycki, Martin B.
Title Longitudinal change in dysarthria associated with Friedreich ataxia: a potential clinical endpoint
Journal name Journal of Neurology   Check publisher's open access policy
ISSN 0340-5354
1432-1459
Publication date 2012-11
Sub-type Article (original research)
DOI 10.1007/s00415-012-6547-x
Volume 259
Issue 11
Start page 2471
End page 2477
Total pages 7
Place of publication Heidelberg, Germany
Publisher Springer Medizin
Collection year 2013
Language eng
Abstract CNS functions that show change across short periods of time are particularly useful clinical endpoints for Friedreich ataxia. This study determined whether there is measurable acoustical change in the dysarthria associated with Friedreich ataxia across yearly intervals. A total of 29 participants diagnosed with Friedreich ataxia were recorded across 4 years at yearly intervals. A repeated measures ANOVA was used to determine which acoustic measures differed across time, and pairwise t tests were used to assess the consistency of the change across the time intervals. The relationship between the identified measures with perceptual severity was assessed with stepwise regression. Significant longitudinal change was observed with four measures that relate to the utterance duration and spectral changes in utterances. The spectral measures consistently detected change across time intervals of two or more years. The four measures combined moderately predicted perceptual severity. Together, the results implicate longitudinal change in speaking rate and utterance duration. Changes in speech associated with Friedreich ataxia can be measured across intervals of 2 years and therefore show rich potential for monitoring disease progression and therapy outcomes.
Keyword Friedreich ataxia
Speech
Biomarkers
Genetics
Degenerative disease
Longitudinal
Q-Index Code C1
Q-Index Status Confirmed Code
Institutional Status UQ
Additional Notes Published online: 6 June 2012.

Document type: Journal Article
Sub-type: Article (original research)
Collections: Official 2013 Collection
School of Health and Rehabilitation Sciences Publications
 
Versions
Version Filter Type
Citation counts: TR Web of Science Citation Count  Cited 5 times in Thomson Reuters Web of Science Article | Citations
Scopus Citation Count Cited 6 times in Scopus Article | Citations
Google Scholar Search Google Scholar
Created: Sun, 02 Dec 2012, 01:09:46 EST by System User on behalf of School of Health & Rehabilitation Sciences