Self-paced timing detects and tracks change in prodromal Huntington disease.
| Autor: | Rowe KC; Department of Psychiatry, University of Iowa Carver College of Medicine, Iowa City, IA 52242, USA., Paulsen JS, Langbehn DR, Duff K, Beglinger LJ, Wang C, O'Rourke JJ, Stout JC, Moser DJ |
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| Jazyk: | angličtina |
| Zdroj: | Neuropsychology [Neuropsychology] 2010 Jul; Vol. 24 (4), pp. 435-42. |
| DOI: | 10.1037/a0018905 |
| Abstrakt: | Objective: This study compares self-paced timing performance (cross-sectionally and longitudinally) between participants with prodromal Huntington's disease (pr-HD) and a comparison group of gene non-expanded participants from affected families (NC). Method: Participants (747 pr-HD: 188 NC) listened to tones presented at 550-ms intervals, matched that pace by tapping response keys and continued the rhythm (self-paced) after the tone had stopped. Standardized cross-sectional and longitudinal linear models examined the relationships between self-paced timing precision and estimated proximity to diagnosis, and other demographic factors. Results: Pr-HD participants showed significantly less timing precision than NC. Comparison of pr-HD and NC participants showed a significant performance difference on two task administration conditions (dominant hand: p < .0001; alternating thumbs: p < .0001). Additionally, estimated proximity to diagnosis was related to timing precision in both conditions, (dominant hand: t = -11.14, df = 920, p < .0001; alternating thumbs: t = -11.32, df = 918, p < .0001). Longitudinal modeling showed that pr-HD participants worsen more quickly at the task than the NC group, and rate of decline increases with estimated proximity to diagnosis in both conditions (dominant hand: t = -2.85, df = 417, p = .0045; alternating thumbs: t = -3.56, df = 445, p = .0004). Effect sizes based on adjusted mean annual change ranged from -0.34 to 0.25 in the longitudinal model. Conclusions: The self-paced timing paradigm has potential for use as a screening tool and outcome measure in pr-HD clinical trials to gauge therapeutically mediated improvement or maintenance of function. |
| Databáze: | MEDLINE |
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