Ross Christopher A
Johns Hopkins University School of Medicine, Department of Psychiatry, Division of Neurobiology, Baltimore, MD 21205, USA.
Neuron. 2002 Aug 29;35(5):819-22. doi: 10.1016/s0896-6273(02)00872-3.
The mechanisms of neurodegeneration in the CAG repeat polyglutamine diseases, including Spinal and Bulbar Muscular Atrophy (SBMA), Huntington's disease (HD), DentatoRubral and PallidoLuysian Atrophy (DRPLA), and Spino-Cerebellar Ataxia (SCA), have been controversial. Issues have included the role of polyglutamine aggregation and possible amyloid formation, localization in the cell nucleus, and possible proteolytic processing. Proposed mechanisms have included activation of caspases or other triggers of apoptosis, mitochondrial or metabolic toxicity, and interference with gene transcription. Recent studies using transgenic mouse and Drosophila models have helped resolve some of these issues and raise hopes for development of therapeutic targets.
CAG重复多聚谷氨酰胺疾病(包括脊髓延髓肌萎缩症(SBMA)、亨廷顿舞蹈症(HD)、齿状核红核苍白球萎缩症(DRPLA)以及脊髓小脑共济失调症(SCA))中的神经退行性变机制一直存在争议。争议点包括多聚谷氨酰胺聚集和可能的淀粉样蛋白形成的作用、在细胞核中的定位以及可能的蛋白水解过程。提出的机制包括半胱天冬酶的激活或其他凋亡触发因素、线粒体或代谢毒性以及对基因转录的干扰。最近使用转基因小鼠和果蝇模型的研究有助于解决其中一些问题,并为治疗靶点的开发带来了希望。