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在1型脑铁沉积神经变性病(NBIA-1,既往称Hallervorden-Spatz综合征)中α-突触核蛋白聚集,伴有广泛的皮质和脑干型路易小体。

Alpha-synuclein accumulation in a case of neurodegeneration with brain iron accumulation type 1 (NBIA-1, formerly Hallervorden-Spatz syndrome) with widespread cortical and brainstem-type Lewy bodies.

作者信息

Neumann M, Adler S, Schlüter O, Kremmer E, Benecke R, Kretzschmar H A

机构信息

Institut für Neuropathologie, Universität München, Germany.

出版信息

Acta Neuropathol. 2000 Nov;100(5):568-74. doi: 10.1007/s004010000224.

Abstract

We studied a 27-year-old woman who died after a 6-year history of progressive dementia, dystonia, ataxia, apraxia, spasticity, choreoathetosis, visual and auditory hallucinations, and optic atrophy. Magnetic resonance imaging showed decreased intensity in the globus pallidus, substantia nigra, and dentate nuclei in T2-weighted images, supporting the clinical diagnosis of neurodegeneration with brain iron accumulation type 1 (NBIA-1; formerly known as Hallervorden-Spatz syndrome). At autopsy the brain showed mild frontotemporal atrophy and discoloration of the globus pallidus and the substantia nigra pars reticularis. Histologically, features typical of NBIA-1 were found including widespread axonal spheroids and large deposits of iron pigment in the discolored regions. Additionally, excessive numbers of Lewy bodies (LBs) were found throughout all examined brain stem and cortical regions. LBs of both types, as well as Lewy neurites in this case of NBIA-1, were strongly labeled by antibodies against alpha-synuclein. These findings give further evidence that accumulation of alpha-synuclein is generally associated with LB formation, i.e., in Parkinson's disease, dementia with Lewy bodies and NBIA-1. The case presented here is particularly notable for its high number of LBs in all areas of the cerebral cortex.

摘要

我们研究了一名27岁女性,她在经历了6年的进行性痴呆、肌张力障碍、共济失调、失用症、痉挛、舞蹈手足徐动症、视幻觉和听幻觉以及视神经萎缩后死亡。磁共振成像显示,在T2加权图像中,苍白球、黑质和齿状核的信号强度降低,支持脑铁沉积1型神经退行性变(NBIA-1;以前称为Hallervorden-Spatz综合征)的临床诊断。尸检时,大脑显示轻度额颞叶萎缩以及苍白球和黑质网状部变色。组织学检查发现了NBIA-1的典型特征,包括广泛的轴突球状体以及在变色区域有大量铁色素沉积。此外,在所有检查的脑干和皮质区域均发现了过多的路易小体(LB)。在该NBIA-1病例中,两种类型的路易小体以及路易神经突均被抗α-突触核蛋白抗体强烈标记。这些发现进一步证明,α-突触核蛋白的积累通常与路易小体的形成有关,即在帕金森病、路易体痴呆和NBIA-1中。此处呈现的病例特别值得注意的是,其大脑皮质所有区域的路易小体数量都很多。

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