DiFiglia M, Sapp E, Chase K O, Davies S W, Bates G P, Vonsattel J P, Aronin N
Department of Neurology, Massachusetts General Hospital, Boston, MA 02114, USA.
Science. 1997 Sep 26;277(5334):1990-3. doi: 10.1126/science.277.5334.1990.
The cause of neurodegeneration in Huntington's disease (HD) is unknown. Patients with HD have an expanded NH2-terminal polyglutamine region in huntingtin. An NH2-terminal fragment of mutant huntingtin was localized to neuronal intranuclear inclusions (NIIs) and dystrophic neurites (DNs) in the HD cortex and striatum, which are affected in HD, and polyglutamine length influenced the extent of huntingtin accumulation in these structures. Ubiquitin was also found in NIIs and DNs, which suggests that abnormal huntingtin is targeted for proteolysis but is resistant to removal. The aggregation of mutant huntingtin may be part of the pathogenic mechanism in HD.
亨廷顿舞蹈病(HD)中神经退行性变的病因尚不清楚。HD患者的亨廷素氨基端多聚谷氨酰胺区域发生了扩展。突变型亨廷素的氨基端片段定位于HD中受影响的大脑皮质和纹状体的神经元核内包涵体(NIIs)和营养不良性神经突(DNs),并且多聚谷氨酰胺的长度影响亨廷素在这些结构中的积聚程度。在NIIs和DNs中也发现了泛素,这表明异常的亨廷素是蛋白酶解的靶点,但难以被清除。突变型亨廷素的聚集可能是HD致病机制的一部分。