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行为变异额颞叶痴呆(bvFTD)中 pTDP-43 病理学的连续分布。

Sequential distribution of pTDP-43 pathology in behavioral variant frontotemporal dementia (bvFTD).

机构信息

Center for Neurodegenerative Disease research (CNDR), Perelman School of Medicine at the University of Pennsylvania, 3rd Floor Maloney Building, 3600 Spruce Street, Philadelphia, PA 19104, USA.

Clinical Neuroanatomy Section, Department of Neurology, Center for Biomedical research, University of Ulm, Helmholtzstrasse 8/1, 89081 Ulm, Germany.

出版信息

Acta Neuropathol. 2014 Mar;127(3):423-439. doi: 10.1007/s00401-013-1238-y. Epub 2014 Jan 10.

Abstract

We examined regional distribution patterns of phosphorylated 43-kDa TAR DNA-binding protein (pTDP-43) intraneuronal inclusions in frontotemporal lobar degeneration (FTLD). Immunohistochemistry was performed on 70 μm sections from FTLD-TDP autopsy cases (n = 39) presenting with behavioral variant frontotemporal dementia. Two main types of cortical pTDP-43 pathology emerged, characterized by either predominantly perikaryal pTDP-43 inclusions (cytoplasmic type, cFTLD) or long aggregates in dendrites (neuritic type, nFTLD). Cortical involvement in nFTLD was extensive and frequently reached occipital areas, whereas cases with cFTLD often involved bulbar somatomotor neurons and the spinal cord. We observed four patterns indicative of potentially sequential dissemination of pTDP-43: cases with the lowest burden of pathology (pattern I) were characterized by widespread pTDP-43 lesions in the orbital gyri, gyrus rectus, and amygdala. With increasing burden of pathology (pattern II) pTDP-43 lesions emerged in the middle frontal and anterior cingulate gyrus as well as in anteromedial temporal lobe areas, the superior and medial temporal gyri, striatum, red nucleus, thalamus, and precerebellar nuclei. More advanced cases showed a third pattern (III) with involvement of the motor cortex, bulbar somatomotor neurons, and the spinal cord anterior horn, whereas cases with the highest burden of pathology (pattern IV) were characterized by pTDP-43 lesions in the visual cortex. We interpret the four neuropathological patterns in bvFTD to be consistent with the hypothesis that pTDP-43 pathology can spread sequentially and may propagate along axonal pathways.

摘要

我们研究了额颞叶变性(FTLD)中磷酸化 43kDa TAR DNA 结合蛋白(pTDP-43)神经元内包涵体的区域性分布模式。对 39 例行为变异型额颞叶痴呆(bvFTD)尸检 FTLD-TDP 病例的 70μm 切片进行免疫组织化学染色。出现两种主要类型的皮质 pTDP-43 病理学,其特征分别为胞浆内 pTDP-43 包涵体(细胞质型,cFTLD)为主或树突中长聚集物(神经丝型,nFTLD)为主。nFTLD 的皮质受累广泛,常累及枕叶区域,而 cFTLD 病例常累及延髓躯体运动神经元和脊髓。我们观察到四种可能与 pTDP-43 潜在传播顺序相关的模式:负担最低的病例(模式 I)表现为眶回、直回和杏仁核广泛的 pTDP-43 病变。随着病理学负担的增加(模式 II),pTDP-43 病变出现在额中回和前扣带回,以及前内侧颞叶区域、颞上回和内侧面、纹状体、红核、丘脑和小脑前核。更晚期的病例表现为第三模式(III),累及运动皮质、延髓躯体运动神经元和脊髓前角,而负担最重的病例(模式 IV)表现为视觉皮质的 pTDP-43 病变。我们将 bvFTD 中的四个神经病理学模式解释为与 pTDP-43 病理学可以顺序传播并可能沿轴突途径传播的假设一致。

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