Suppr超能文献

常染色体显性遗传性脊髓小脑共济失调:新出现的机制主题提示浦肯野细胞普遍存在易损性。

The autosomal dominant spinocerebellar ataxias: emerging mechanistic themes suggest pervasive Purkinje cell vulnerability.

作者信息

Hekman Katherine E, Gomez Christopher M

机构信息

Department of Vascular Surgery, McGaw Medical Center of Northwestern University, Chicago, Illinois, USA.

Department of Neurology, The University of Chicago, Chicago, Illinois, USA.

出版信息

J Neurol Neurosurg Psychiatry. 2015 May;86(5):554-61. doi: 10.1136/jnnp-2014-308421. Epub 2014 Aug 18.

Abstract

The spinocerebellar ataxias are a genetically heterogeneous group of disorders with clinically overlapping phenotypes arising from Purkinje cell degeneration, cerebellar atrophy and varying degrees of degeneration of other grey matter regions. For 22 of the 32 subtypes, a genetic cause has been identified. While recurring themes are emerging, there is no clear correlation between the clinical phenotype or penetrance, the type of genetic defect or the category of the disease mechanism, or the neuronal types involved beyond Purkinje cells. These phenomena suggest that cerebellar Purkinje cells may be a uniquely vulnerable neuronal cell type, more susceptible to a wider variety of genetic/cellular insults than most other neuron types.

摘要

脊髓小脑共济失调是一组基因异质性疾病,其临床表型相互重叠,由浦肯野细胞变性、小脑萎缩以及其他灰质区域不同程度的变性引起。在32个亚型中,已确定22个亚型的遗传病因。虽然一些反复出现的主题正在显现,但临床表型或外显率、遗传缺陷类型或疾病机制类别,以及除浦肯野细胞外所涉及的神经元类型之间,并无明确的相关性。这些现象表明,小脑浦肯野细胞可能是一种特别易损的神经元细胞类型,比大多数其他神经元类型更容易受到更广泛的各种遗传/细胞损伤。

相似文献

1
The autosomal dominant spinocerebellar ataxias: emerging mechanistic themes suggest pervasive Purkinje cell vulnerability.
J Neurol Neurosurg Psychiatry. 2015 May;86(5):554-61. doi: 10.1136/jnnp-2014-308421. Epub 2014 Aug 18.
2
Why do so many genetic insults lead to Purkinje Cell degeneration and spinocerebellar ataxia?
Neurosci Lett. 2019 Jan 1;688:49-57. doi: 10.1016/j.neulet.2018.02.004. Epub 2018 Feb 5.
3
Clinical, genetic and neuropathological characterization of spinocerebellar ataxia type 37.
Brain. 2018 Jul 1;141(7):1981-1997. doi: 10.1093/brain/awy137.
4
Purkinje cell loss is the major brain pathology of spinocerebellar ataxia type 10.
J Neurol Neurosurg Psychiatry. 2013 Dec;84(12):1409-11. doi: 10.1136/jnnp-2013-305080. Epub 2013 Jun 29.
9
Senseless makes sense for spinocerebellar ataxia-1.
Nat Neurosci. 2005 Nov;8(11):1422-4. doi: 10.1038/nn1105-1422.

引用本文的文献

4
Neuroglobin overexpression in cerebellar neurons of Harlequin mice improves mitochondrial homeostasis and reduces ataxic behavior.
Mol Ther. 2024 Jul 3;32(7):2150-2175. doi: 10.1016/j.ymthe.2024.05.030. Epub 2024 May 24.
6
Cognitive, Emotional, and Other Non-motor Symptoms of Spinocerebellar Ataxias.
Curr Neurol Neurosci Rep. 2024 Mar;24(3):47-54. doi: 10.1007/s11910-024-01331-4. Epub 2024 Jan 25.
8
Cognitive Decline and Mood Alterations in the Mouse Model of Spinocerebellar Ataxia Type 2.
Cerebellum. 2024 Feb;23(1):145-161. doi: 10.1007/s12311-023-01520-w. Epub 2023 Jan 21.
9
Pre-ataxic loss of intrinsic plasticity and motor learning in a mouse model of SCA1.
Brain. 2023 Jun 1;146(6):2332-2345. doi: 10.1093/brain/awac422.
10
The extra-cerebellar effects of spinocerebellar ataxia type 1 (SCA1): looking beyond the cerebellum.
Cell Mol Life Sci. 2022 Jul 8;79(8):404. doi: 10.1007/s00018-022-04419-7.

本文引用的文献

1
Consensus paper: pathological mechanisms underlying neurodegeneration in spinocerebellar ataxias.
Cerebellum. 2014 Apr;13(2):269-302. doi: 10.1007/s12311-013-0539-y.
2
Repeat-associated non-ATG (RAN) translation in neurological disease.
Hum Mol Genet. 2013 Oct 15;22(R1):R45-51. doi: 10.1093/hmg/ddt371. Epub 2013 Aug 4.
4
New subtype of spinocerebellar ataxia with altered vertical eye movements mapping to chromosome 1p32.
JAMA Neurol. 2013 Jun;70(6):764-71. doi: 10.1001/jamaneurol.2013.2311.
5
Hereditary ataxia and spastic paraplegia in Portugal: a population-based prevalence study.
JAMA Neurol. 2013 Jun;70(6):746-55. doi: 10.1001/jamaneurol.2013.1707.
7
Mutations in potassium channel kcnd3 cause spinocerebellar ataxia type 19.
Ann Neurol. 2012 Dec;72(6):870-80. doi: 10.1002/ana.23700.
8
Mutations in KCND3 cause spinocerebellar ataxia type 22.
Ann Neurol. 2012 Dec;72(6):859-69. doi: 10.1002/ana.23701.
10

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验