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孔图尔西家族帕金森病的临床基因分析

Clinical genetic analysis of Parkinson's disease in the Contursi kindred.

作者信息

Golbe L I, Di Iorio G, Sanges G, Lazzarini A M, La Sala S, Bonavita V, Duvoisin R C

机构信息

Department of Neurology, University of Medicine and Dentistry of New Jersey-Robert Wood Johnson Medical School, New Brunswick 08903, USA.

出版信息

Ann Neurol. 1996 Nov;40(5):767-75. doi: 10.1002/ana.410400513.

DOI:10.1002/ana.410400513
PMID:8957018
Abstract

We performed a clinical genetic analysis of a kindred originating in the town of Contursi in Salerno province, Italy, in which 60 individuals in 5 generations are known to have had Parkinson's disease (PD). Two previously reported autopsy cases showed typical PD with Lewy bodies. The inheritance pattern is apparently autosomally dominant with a segregation ratio of 40.1% for kindred members aged 50 years and older. The mean age at PD onset is 45.6 years (standard deviation, 13.48; range, 20-85) with a mean course to death of 9.2 years (standard deviation, 4.87; range, 2-20). Otherwise, clinical characteristics of PD in the kindred, including variance in onset age and incidence of tremor and levodopa responsiveness, are similar to those of PD in the community. The presence of tremor tended to be concordant in affected parent-child pairs, but there was no parent-child correlation for age at onset or intrasibship clustering of tremor or onset age. A suggestion of anticipation disappeared after adjustment for age-related ascertainment bias. The findings show that a presumably single mutation can produce a heterogeneous PD phenotype, even among siblings. This is consistent with the hypothesis that PD in the community may in fact be caused by such a mutation, but one producing a lower penetrance and older age at onset than those in this kindred.

摘要

我们对源自意大利萨勒诺省孔图尔西镇的一个家族进行了临床基因分析,已知该家族五代中的60人患有帕金森病(PD)。此前报告的两例尸检病例显示为典型的路易体帕金森病。遗传模式显然为常染色体显性遗传,50岁及以上家族成员的分离率为40.1%。帕金森病发病的平均年龄为45.6岁(标准差为13.48;范围为20 - 85岁),平均死亡病程为9.2年(标准差为4.87;范围为2 - 20年)。此外,该家族中帕金森病的临床特征,包括发病年龄差异、震颤发生率和左旋多巴反应性,与社区中帕金森病的特征相似。在受影响的亲子对中,震颤的出现往往具有一致性,但发病年龄不存在亲子相关性,震颤或发病年龄在同胞间也无聚集现象。在调整了与年龄相关的确诊偏倚后,预期现象的迹象消失。研究结果表明,即使在兄弟姐妹中,一个可能的单一突变也能产生异质性的帕金森病表型。这与以下假设一致,即社区中的帕金森病实际上可能由这样一种突变引起,但这种突变的外显率较低且发病年龄比该家族中的情况更大。

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Clinical genetic analysis of Parkinson's disease in the Contursi kindred.孔图尔西家族帕金森病的临床基因分析
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