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伴有E184D早老素-1突变的家族性阿尔茨海默病的NACP/α-突触核蛋白、NAC和β-淀粉样蛋白病理学:两例尸检病例的临床病理学研究

NACP/alpha-synuclein, NAC, and beta-amyloid pathology of familial Alzheimer's disease with the E184D presenilin-1 mutation: a clinicopathological study of two autopsy cases.

作者信息

Yokota Osamu, Terada Seishi, Ishizu Hideki, Ujike Hiroshi, Ishihara Takeshi, Nakashima Hanae, Yasuda Minoru, Kitamura Yoshihiro, Uéda Kenji, Checler Frédéric, Kuroda Shigetoshi

机构信息

Department of Neuropsychiatry, Okayama University Graduate School of Medicine and Dentistry, 2-5-1 Shikata-cho, Okayama 700-8558, Japan.

出版信息

Acta Neuropathol. 2002 Dec;104(6):637-48. doi: 10.1007/s00401-002-0596-7. Epub 2002 Aug 14.

DOI:10.1007/s00401-002-0596-7
PMID:12410385
Abstract

Approximately 60% of familial and sporadic Alzheimer's disease (AD) cases manifest Lewy bodies (LBs), of which a major component is alpha-synuclein. Although the pathogenic role of alpha-synuclein in AD remains unclear, LB formation might be associated with pathological beta-amyloid (Abeta) overproduction. Here, we present the clinical and pathological characteristics of two affected family members from a pedigree with the E184D mutation of presenilin-1. One case presented with typical clinical features of AD, but the other case also developed clinical characteristics of dementia with Lewy bodies (DLB), including visual hallucinations, delusions, and parkinsonism. In both cases, neuropathological examination revealed numerous neurofibrillary tangles and severe Abeta deposition in senile plaques and amyloid angiopathy, in which Abeta42 rather than Abeta40 was predominant. Furthermore, remarkable alpha-synuclein pathology, including LBs and the accumulation of the non-Abeta component of AD amyloid (NAC) in plaques and astrocytes, was detected only in the case that presented with the symptoms of DLB. These findings suggest that (1) LB pathology can influence the clinical features of familial AD, (2) the E184D mutation of presenilin-1 may be associated with the LB formation through Abeta overproduction, although the process of LB formation is strongly affected by other unknown mechanisms, (3) in neurodegenerative disorders with LBs, there is a common pathophysiological background inducing NAC accumulation in neuritic plaques and astrocytes, and (4) the NAC accumulation in neuritic plaques is modulated by the abnormally aggregated tau protein.

摘要

约60%的家族性和散发性阿尔茨海默病(AD)病例出现路易小体(LB),其主要成分是α-突触核蛋白。尽管α-突触核蛋白在AD中的致病作用尚不清楚,但LB的形成可能与病理性β-淀粉样蛋白(Aβ)的过度产生有关。在此,我们展示了来自一个早老素-1基因E184D突变家系的两名患病家庭成员的临床和病理特征。其中一例表现出典型的AD临床特征,但另一例还出现了路易体痴呆(DLB)的临床特征,包括视幻觉、妄想和帕金森综合征。在这两例中,神经病理学检查均发现大量神经原纤维缠结,以及老年斑和淀粉样血管病中严重的Aβ沉积,其中Aβ42而非Aβ40占主导。此外,仅在表现出DLB症状的病例中检测到显著的α-突触核蛋白病理学改变,包括LB以及斑块和星形胶质细胞中AD淀粉样蛋白非Aβ成分(NAC)的积累。这些发现表明:(1)LB病理学可影响家族性AD的临床特征;(2)早老素-1基因的E184D突变可能通过Aβ过度产生与LB形成相关,尽管LB形成过程受其他未知机制的强烈影响;(3)在伴有LB的神经退行性疾病中,存在一个共同的病理生理背景,可诱导NAC在神经炎性斑块和星形胶质细胞中积累;(4)神经炎性斑块中的NAC积累受异常聚集的tau蛋白调节。

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