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17号染色体与遗传性痴呆:对三个非阿尔茨海默病家族及晚发性家族性阿尔茨海默病亲属的连锁研究

Chromosome 17 and hereditary dementia: linkage studies in three non-Alzheimer families and kindreds with late-onset FAD.

作者信息

Bird T D, Wijsman E M, Nochlin D, Leehey M, Sumi S M, Payami H, Poorkaj P, Nemens E, Rafkind M, Schellenberg G D

机构信息

VA Puget Sound Health Care System, Seattle, WA 98108, USA.

出版信息

Neurology. 1997 Apr;48(4):949-54. doi: 10.1212/wnl.48.4.949.

DOI:10.1212/wnl.48.4.949
PMID:9109883
Abstract

Several previous families with differing clinical and pathologic characteristics have demonstrated linkage to the 17q21-22 region. We have performed a linkage analysis with chromosome 17 markers on three families showing autosomal dominant inheritance of non-Alzheimer dementia and 60 kindreds with late-onset familial Alzheimer's disease (FAD). Family A shows unequivocal evidence of linkage with a maximum lod score of 5.0 for marker D17S934 (theta = 0.001). This family has an unusual syndrome of a schizophrenia-like psychosis beginning in the fifth or sixth decade followed by severe dementia with an average disease duration of 13.8 years. Neuropathology from five autopsies in this family has shown marked neurofibrillary tangle formation (NFT), degeneration of the amygdala, and no amyloid plaques. This confirms the presence of a gene associated with dementia on 17q and extends the related phenotype to include schizophrenia-like symptoms and classic NFT pathology. A second family with early aphasia progressing to dementia and cortical-basal ganglion-like degeneration also has suggestive evidence for linkage to 17q. A third family with very early-onset dementia (mean, 31 years) and nonspecific pathology can be excluded from the 17q region and emphasizes additional genetic heterogeneity in non-Alzheimer hereditary dementia. Finally, we also present evidence against linkage to D17S579 in the set of 60 families with late-onset FAD, providing further evidence that the chromosome 17 gene is unlikely to be involved in the pathogenesis of typical AD.

摘要

之前有几个临床和病理特征各异的家族已证明与17q21 - 22区域存在连锁关系。我们对三个显示非阿尔茨海默病痴呆常染色体显性遗传的家族以及60个晚发性家族性阿尔茨海默病(FAD)家族进行了17号染色体标记的连锁分析。家族A显示出与标记D17S934明确的连锁证据,最大lod值为5.0(θ = 0.001)。这个家族有一种不寻常的综合征,在第五或第六个十年开始出现类似精神分裂症的精神病,随后是严重痴呆,平均病程为13.8年。该家族五例尸检的神经病理学显示有明显的神经原纤维缠结形成(NFT)、杏仁核退化且无淀粉样斑块。这证实了17q上存在与痴呆相关的基因,并将相关表型扩展至包括类似精神分裂症的症状和典型的NFT病理学。第二个家族有早期失语并进展为痴呆以及皮质 - 基底神经节样变性,也有与17q连锁的提示性证据。第三个家族有非常早发的痴呆(平均31岁)且病理表现不特异,可排除与17q区域的连锁关系,这强调了非阿尔茨海默遗传性痴呆中存在额外的基因异质性。最后,我们还提供了在60个晚发性FAD家族中与D17S579无连锁关系的证据,进一步证明17号染色体基因不太可能参与典型AD的发病机制。

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Chromosome 17 and hereditary dementia: linkage studies in three non-Alzheimer families and kindreds with late-onset FAD.17号染色体与遗传性痴呆:对三个非阿尔茨海默病家族及晚发性家族性阿尔茨海默病亲属的连锁研究
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