Striatal volume contributes to the prediction of onset of Huntington disease in incident cases.
Striatal volume contributes to the prediction of onset of Huntington disease in incident cases.
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DOI：
10.1016/j.biopsych.2011.07.030
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发表时间：
2012-05-01
影响因子：
10.6
通讯作者：
Paulsen, Jane S.
中科院分区：
文献类型：
--
作者：
Aylward, Elizabeth H.;Liu, Dawei;Nopoulos, Peggy C.;Ross, Christopher A.;Pierson, Ronald K.;Mills, James A.;Long, Jeffrey D.;Paulsen, Jane S.
先前的神经影像学研究表明，亨廷顿病（HD）的脑萎缩在运动异常严重到足以诊断之前许多年就开始了。大多数计划用于HD诊断前阶段个体的临床试验建议使用疾病发病延迟作为主要结局指标。虽然基于年龄和CAG重复长度已经开发出公式来预测HD运动何时发生，但有额外的措施可以提高疾病发病预测的准确性。目前的研究检查了85个个体的纹状体和白质体积的MRI测量，从HD前期到可诊断的运动发病（“事件病例”），85个个体在CAG重复长度、性别和年龄上与事件病例相匹配，在研究过程中未被诊断为HD。在最初的MRI扫描后1到4年被诊断为hd的人，纹状体和白质的体积明显小于那些仍处于hd前期的人。壳核体积是区分两组的最佳指标。结果表明，MRI体积测量可能有助于在HD前期临床试验中选择个体，其中HD运动发作是主要的结果测量。在计划hd前期的多地点临床试验时，研究人员可能还想考虑使用更客观的测量，如MRI体积，除了可诊断的运动障碍的发病，作为主要的结果测量。
Previous neuroimaging research indicates that brain atrophy in Huntington disease (HD) begins many years before movement abnormalities become severe enough to warrant diagnosis. Most clinical trials being planned for individuals in the prediagnostic stage of HD propose to use delay of disease onset as the primary outcome measure. Although formulae have been developed, based on age and CAG repeat length, to predict when HD motor onset will occur, it would be useful to have additional measures that can improve the accuracy of prediction of disease onset. The current study examined MRI measures of striatum and white matter volume in 85 individuals prospectively followed from pre-HD stage through diagnosable motor onset (“incident cases”) and 85 individuals individually-matched with incident cases on CAG repeat length, sex, and age, who were not diagnosed with HD during the course of the study. Volumes of striatum and white matter were significantly smaller in individuals who would be diagnosed 1 to 4 years following the initial MRI scan, compared to those who would remain in the pre-HD stage. Putamen volume was the measure that best distinguished between the two groups. Results suggest that MRI volumetric measures may be helpful in selecting individuals for future clinical trials in pre-HD where HD motor onset is the primary outcome measure. In planning for multisite clinical trials in pre-HD, investigators may also want to consider using more objective measures, such as MRI volumes, in addition to onset of diagnosable movement disorder, as major outcome measures.
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影响因子：
--
作者：
Aylward, EH;Codori, AM;Brandt, J
通讯作者：
Brandt, J
影响因子：
11
作者：
STARKSTEIN, SE;BRANDT, J;FOLSTEIN, M
通讯作者：
FOLSTEIN, M
影响因子：
3.5
作者：
Langbehn, DR;Brinkman, RR;Hayden, MR
通讯作者：
Hayden, MR
影响因子：
5.7
作者：
Magnotta, VA;Harris, G;Heckel, D
通讯作者：
Heckel, D
影响因子：
3.8
作者：
Paulsen JS;Nopoulos PC;Aylward E;Ross CA;Johnson H;Magnotta VA;Juhl A;Pierson RK;Mills J;Langbehn D;Nance M;PREDICT-HD Investigators and Coordinators of the Huntington's Study Group (HSG)
通讯作者：
PREDICT-HD Investigators and Coordinators of the Huntington's Study Group (HSG)