Molecular Aging & Development Laboratory, Department of Surgery, Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts, United States of America.
PLoS One. 2010 May 20;5(5):e10659. doi: 10.1371/journal.pone.0010659.
Down syndrome (DS, trisomy 21) is the most common chromosomal disorder and the leading genetic cause of intellectual disability in humans. In DS, triplication of chromosome 21 invariably includes the APP gene (21q21) encoding the Alzheimer's disease (AD) amyloid precursor protein (APP). Triplication of the APP gene accelerates APP expression leading to cerebral accumulation of APP-derived amyloid-beta peptides (Abeta), early-onset AD neuropathology, and age-dependent cognitive sequelae. The DS phenotype complex also includes distinctive early-onset cerulean cataracts of unknown etiology. Previously, we reported increased Abeta accumulation, co-localizing amyloid pathology, and disease-linked supranuclear cataracts in the ocular lenses of subjects with AD. Here, we investigate the hypothesis that related AD-linked Abeta pathology underlies the distinctive lens phenotype associated with DS. Ophthalmological examinations of DS subjects were correlated with phenotypic, histochemical, and biochemical analyses of lenses obtained from DS, AD, and normal control subjects. Evaluation of DS lenses revealed a characteristic pattern of supranuclear opacification accompanied by accelerated supranuclear Abeta accumulation, co-localizing amyloid pathology, and fiber cell cytoplasmic Abeta aggregates (approximately 5 to 50 nm) identical to the lens pathology identified in AD. Peptide sequencing, immunoblot analysis, and ELISA confirmed the identity and increased accumulation of Abeta in DS lenses. Incubation of synthetic Abeta with human lens protein promoted protein aggregation, amyloid formation, and light scattering that recapitulated the molecular pathology and clinical features observed in DS lenses. These results establish the genetic etiology of the distinctive lens phenotype in DS and identify the molecular origin and pathogenic mechanism by which lens pathology is expressed in this common chromosomal disorder. Moreover, these findings confirm increased Abeta accumulation as a key pathogenic determinant linking lens and brain pathology in both DS and AD.
唐氏综合征(DS,21 三体)是最常见的染色体疾病,也是人类智力残疾的主要遗传原因。在 DS 中,21 号染色体的三倍体总是包括 APP 基因(21q21),该基因编码阿尔茨海默病(AD)淀粉样前体蛋白(APP)。APP 基因的三倍体加速了 APP 的表达,导致大脑中 APP 衍生的淀粉样-β肽(Abeta)的积累、早发性 AD 神经病理学和与年龄相关的认知后果。DS 表型复杂还包括病因不明的独特早发性天蓝色白内障。以前,我们报道了 AD 患者的眼晶状体中 Abeta 积累增加、淀粉样病理共定位和与疾病相关的核上白内障。在这里,我们假设与 AD 相关的 Abeta 病理学是与 DS 相关的独特晶状体表型的基础。对 DS 患者的眼科检查与从 DS、AD 和正常对照组获得的晶状体的表型、组织化学和生化分析相关联。对 DS 晶状体的评估显示出核上混浊的特征性模式,伴随着核上 Abeta 积累的加速、淀粉样病理共定位和纤维细胞细胞质 Abeta 聚集物(约 5 至 50nm),与在 AD 中鉴定的晶状体病理学相同。肽测序、免疫印迹分析和 ELISA 证实了 DS 晶状体中 Abeta 的身份和增加的积累。合成 Abeta 与人晶状体蛋白孵育促进了蛋白质聚集、淀粉样形成和光散射,再现了在 DS 晶状体中观察到的分子病理学和临床特征。这些结果确立了 DS 中独特晶状体表型的遗传病因,并确定了晶状体病理学在这种常见染色体疾病中表达的分子起源和发病机制。此外,这些发现证实了 Abeta 积累的增加是将 DS 和 AD 中的晶状体和脑病理学联系起来的关键致病决定因素。