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Neurocognitive Signs in Prodromal Huntington Disease
Journal article   Open access   Peer reviewed

Neurocognitive Signs in Prodromal Huntington Disease

Julie C Stout, Jane S Paulsen, Sarah Queller, Andrea C Solomon, Kathryn B Whitlock, J. Colin Campbell, Noelle Carlozzi, Kevin Duff, Leigh J Beglinger, Douglas R Langbehn, …
Neuropsychology, Vol.25(1), pp.1-14
01/2011
DOI: 10.1037/a0020937
PMCID: PMC3017660
PMID: 20919768
url
https://admin.research-repository.uwa.edu.au/en/publications/8886580f-0059-4eb0-94ed-461d7101ad3dView
Open Access

Abstract

Objective: PREDICT-HD is a large-scale international study of people with the Huntington disease (HD) CAG-repeat expansion who are not yet diagnosed with HD. The objective of this study was to determine the stage in the HD prodrome at which cognitive differences from CAG-normal controls can be reliably detected. Method: For each of 738 HD CAG-expanded participants, we computed estimated years to clinical diagnosis and probability of diagnosis in 5 years based on age and CAG-repeat expansion number (Langbehn, Brinkman, Falush, Paulsen, & Hayden, 2004). We then stratified the sample into groups: NEAR, estimated to be ≤9 years; MID, between 9 and 15 years; and FAR, ≥15 years. The control sample included 168 CAG-normal participants. Nineteen cognitive tasks were used to assess attention, working memory, psychomotor functions, episodic memory, language, recognition of facial emotion, sensory-perceptual functions, and executive functions. Results: Compared with the controls, the NEAR group showed significantly poorer performance on nearly all of the cognitive tests and the MID group on about half of the cognitive tests (p = .05, Cohen's d NEAR as large as −1.17, MID as large as −0.61). One test even revealed significantly poorer performance in the FAR group (Cohen's d = −0.26). Individual tasks accounted for 0.2% to 9.7% of the variance in estimated proximity to diagnosis. Overall, the cognitive battery accounted for 34% of the variance; in comparison, the Unified Huntington's Disease Rating Scale motor score accounted for 11.7%. Conclusions: Neurocognitive tests are robust clinical indicators of the disease process prior to reaching criteria for motor diagnosis of HD.
Neuropsychology presymptomatic cognitive assessment prediagnosis psychomotor

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