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照顾 Machado-Joseph 病:当前的认识和如何帮助患者。

Caring for Machado-Joseph disease: current understanding and how to help patients.

机构信息

Department of Neurology, University of Campinas (UNICAMP), Campinas, São Paulo, Brazil.

出版信息

Parkinsonism Relat Disord. 2010 Jan;16(1):2-7. doi: 10.1016/j.parkreldis.2009.08.012. Epub 2009 Oct 6.

Abstract

Machado-Joseph disease or spinocerebellar ataxia 3 (MJD/SCA3) is a clinically heterogeneous, neurodegenerative disorder characterized by varying degrees of ataxia, ophthalmoplegia, peripheral neuropathy, pyramidal dysfunction and movement disorder. MJD/SCA3 is caused by a CAG repeat expansion mutation in the protein coding region of the ATXN3 gene located at chromosome 14q32.1. Current hypotheses regarding pathogenesis favor the view that mutated ataxin-3, with its polyglutamine expansion, is prone to adopt an abnormal conformation, engage in altered protein-protein interactions and aggregate. Expanded CAG repeat length correlates with the range and severity of the clinical manifestations and inversely correlates with age of disease onset. Though MJD/SCA3 is classically described as affecting the cerebellum, brainstem and basal ganglia, recent neuropathology and neuroimaging series demonstrate involvement of other areas such as the thalamus and cerebral cortex. Clinically, much emphasis has been placed in the description and recognition of the non-motor symptoms observed in these patients, such as pain, cramps, fatigue and depression. Currently, no disease modifying treatment exists for MJD/SCA3. Standard of care includes genetic counseling, exercise/physical therapy programs, and speech and swallow evaluation. Symptomatic treatment for clinical findings such as depression, sleep disorders, parkinsonism, dystonia, cramps, and pain is important to improve the quality of life for those with MJD/SCA3.

摘要

马查多-约瑟夫病或脊髓小脑共济失调 3 型(MJD/SCA3)是一种临床表现多样的神经退行性疾病,其特征为不同程度的共济失调、眼肌麻痹、周围神经病、锥体束功能障碍和运动障碍。MJD/SCA3 是由位于 14q32.1 染色体上的 ATXN3 基因蛋白编码区的 CAG 重复扩展突变引起的。目前关于发病机制的假说倾向于认为,突变的共济失调蛋白-3 及其多聚谷氨酰胺扩展后容易形成异常构象,参与改变蛋白-蛋白相互作用并聚集。扩展的 CAG 重复长度与临床表现的范围和严重程度相关,与发病年龄呈负相关。尽管 MJD/SCA3 经典地被描述为影响小脑、脑干和基底节,但最近的神经病理学和神经影像学系列研究表明,其他区域如丘脑和大脑皮层也有受累。临床上,人们非常重视描述和认识这些患者中观察到的非运动症状,如疼痛、痉挛、疲劳和抑郁。目前,MJD/SCA3 没有可改变疾病进程的治疗方法。标准治疗包括遗传咨询、运动/物理治疗计划以及言语和吞咽评估。对抑郁、睡眠障碍、帕金森病、肌张力障碍、痉挛和疼痛等临床发现进行对症治疗,对于提高 MJD/SCA3 患者的生活质量非常重要。

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Polyglutamine neurodegeneration: protein misfolding revisited.多聚谷氨酰胺神经变性:重新审视蛋白质错误折叠
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Sleep symptoms and their clinical correlates in Machado-Joseph disease.马查多-约瑟夫病的睡眠症状及其临床关联
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Axonal dysfunction in the deep white matter in Machado-Joseph disease.马查多-约瑟夫病中深部白质的轴突功能障碍。
J Neuroimaging. 2009 Jan;19(1):9-12. doi: 10.1111/j.1552-6569.2008.00260.x. Epub 2008 May 8.

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