Evaluation of longitudinal 12 and 24 month cognitive outcomes in premanifest and early Huntington's disease

被引:109
作者
Stout, Julie C. [1 ]
Jones, Rebecca [2 ]
Labuschagne, Izelle [1 ]
O'Regan, Alison M. [1 ]
Say, Miranda J. [3 ]
Dumas, Eve M. [4 ]
Queller, Sarah [1 ,5 ]
Justo, Damian [6 ,7 ]
Santos, Rachelle Dar [8 ]
Coleman, Allison [8 ]
Hart, Ellen P. [4 ]
Duerr, Alexandra [6 ,7 ]
Leavitt, Blair R. [8 ]
Roos, Raymund A. [4 ]
Langbehn, Doug R. [9 ,10 ]
Tabrizi, Sarah J. [3 ]
Frost, Chris [2 ]
机构
[1] Monash Univ, Sch Psychol & Psychiat, Melbourne, Vic 3800, Australia
[2] Univ London London Sch Hyg & Trop Med, Dept Med Stat, London WC1E 7HT, England
[3] UCL, UCL Inst Neurol, London, England
[4] Leiden Univ, Med Ctr, Dept Neurol, NL-2300 RA Leiden, Netherlands
[5] Queller Consulting, Dunedin, FL USA
[6] Hop La Pitie Salpetriere, APHP, Dept Genet & Cytogenet, Paris, France
[7] Hop La Pitie Salpetriere, APHP, INSERM, UMR S679, Paris, France
[8] Univ British Columbia, Dept Med Genet, Vancouver, BC, Canada
[9] Univ Iowa, Dept Psychiat, Iowa City, IA 52242 USA
[10] Univ Iowa, Dept Biostat Secondary, Iowa City, IA USA
关键词
CARRIERS; DECLINE; HD;
D O I
10.1136/jnnp-2011-301940
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Background Deterioration of cognitive functioning is a debilitating symptom in many neurodegenerative diseases, such as Huntington's disease (HD). To date, there are no effective treatments for the cognitive problems associated with HD. Cognitive assessment outcomes will have a central role in the efforts to develop treatments to delay onset or slow the progression of the disease. The TRACK-HD study was designed to build a rational basis for the selection of cognitive outcomes for HD clinical trials. Methods There were a total of 349 participants, including controls (n=116), premanifest HD (n=117) and early HD (n=116). A standardised cognitive assessment battery (including nine cognitive tests comprising 12 outcome measures) was administered at baseline, and at 12 and 24 months, and consisted of a combination of paper and pencil and computerised tasks selected to be sensitive to cortical-striatal damage or HD. Each cognitive outcome was analysed separately using a generalised least squares regression model. Results are expressed as effect sizes to permit comparisons between tasks. Results 10 of the 12 cognitive outcomes showed evidence of deterioration in the early HD group, relative to controls, over 24 months, with greatest sensitivity in Symbol Digit, Circle Tracing direct and indirect, and Stroop word reading. In contrast, there was very little evidence of deterioration in the premanifest HD group relative to controls. Conclusions The findings describe tests that are sensitive to longitudinal cognitive change in HD and elucidate important considerations for selecting cognitive outcomes for clinical trials of compounds aimed at ameliorating cognitive decline in HD.
引用
收藏
页码:687 / 694
页数:8
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