Cortical Features in Child and Adolescent Carriers of Mutant Huntingtin (mHTT).

IF 2.1 Q3 NEUROSCIENCES Journal of Huntington's disease Pub Date : 2022-01-01 DOI:10.3233/JHD-210512

Erin E Reasoner, Ellen van der Plas, Douglas R Langbehn, Amy L Conrad, Timothy R Koscik, Eric A Epping, Vincent A Magnotta, Peggy C Nopoulos

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引用次数: 1

Abstract

Background: Molecular studies provide evidence that mutant huntingtin (mHTT) affects early cortical development; however, cortical development has not been evaluated in child and adolescent carriers of mHTT.

Objective: To evaluate the impact of mHTT on the developmental trajectories of cortical thickness and surface area.

Methods: Children and adolescents (6-18 years) participated in the KidsHD study. mHTT carrier status was determined for research purposes only to classify participants as gene expanded (GE) and gene non-expanded (GNE). Cortical features were extracted from 3T neuroimaging using FreeSurfer. Nonlinear mixed effects models were conducted to determine if age, group, and CAG repeat were associated with cortical morphometry.

Results: Age-related changes in cortical morphometry were similar across groups. Expanded CAG repeat was not significantly associated with cortical features.

Conclusion: While striatal development is markedly different in GE and GNE, developmental change of the cortex appears grossly normal among child and adolescent carrier of mHTT.

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儿童和青少年突变亨廷顿蛋白(mHTT)携带者的皮质特征。

背景:分子研究证明突变型亨廷顿蛋白(mHTT)影响早期皮层发育;然而，尚未评估儿童和青少年mHTT携带者的皮质发育情况。目的:探讨mHTT对大鼠皮质厚度和表面积发育轨迹的影响。方法:儿童和青少年(6-18岁)参与KidsHD研究。确定mHTT携带者状态仅用于研究目的，将参与者分为基因扩增(GE)和基因非扩增(GNE)。使用FreeSurfer从3T神经成像中提取皮层特征。采用非线性混合效应模型来确定年龄、组别和CAG重复数是否与皮质形态测定有关。结果:不同年龄组的皮层形态变化相似。CAG重复扩增与皮质特征无显著相关性。结论:儿童和青少年mHTT携带者的纹状体发育在GE和GNE中有显著差异，而皮层的发育变化基本正常。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Journal of Huntington's disease NEUROSCIENCES-

CiteScore

4.80

自引率

9.70%

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