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对一个患有智力残疾、早发性痉挛和小脑萎缩的家庭进行外显子组测序,检测到EXOSC3基因中的一个新突变。

Exome sequencing in a family with intellectual disability, early onset spasticity, and cerebellar atrophy detects a novel mutation in EXOSC3.

作者信息

Zanni Ginevra, Scotton Chiara, Passarelli Chiara, Fang Mingyan, Barresi Sabina, Dallapiccola Bruno, Wu Bin, Gualandi Francesca, Ferlini Alessandra, Bertini E, Wei Wang

机构信息

Department of Neurosciences, Unit of Molecular Medicine for Neuromuscular and Neurodegenerative disorders, Rome, Italy.

出版信息

Neurogenetics. 2013 Nov;14(3-4):247-50. doi: 10.1007/s10048-013-0371-z. Epub 2013 Aug 24.

DOI:10.1007/s10048-013-0371-z
PMID:23975261
Abstract

Whole exome sequencing in two-generational kindred from Bangladesh with early onset spasticity, mild intellectual disability, distal amyotrophy, and cerebellar atrophy transmitted as an autosomal recessive trait identified the following two missense mutations in the EXOSC3 gene: a novel p.V80F mutation and a known p.D132A change previously associated with mild variants of pontocerebellar hypoplasia type 1. This study confirms the involvement of RNA processing proteins in disorders with motor neuron and cerebellar degeneration overlapping with spinocerebellar ataxia 36 and rare forms of hereditary spastic paraplegia with cerebellar features.

摘要

在来自孟加拉国的一个两代家系中进行全外显子组测序,该家系具有早发性痉挛、轻度智力残疾、远端肌萎缩和小脑萎缩,呈常染色体隐性遗传特征,在EXOSC3基因中鉴定出以下两个错义突变:一个新的p.V80F突变和一个已知的p.D132A变化,该变化先前与1型桥脑小脑发育不全的轻度变异有关。这项研究证实了RNA加工蛋白参与了与脊髓小脑共济失调36重叠的运动神经元和小脑变性疾病以及具有小脑特征的罕见遗传性痉挛性截瘫形式。

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Exome sequencing in a family with intellectual disability, early onset spasticity, and cerebellar atrophy detects a novel mutation in EXOSC3.对一个患有智力残疾、早发性痉挛和小脑萎缩的家庭进行外显子组测序,检测到EXOSC3基因中的一个新突变。
Neurogenetics. 2013 Nov;14(3-4):247-50. doi: 10.1007/s10048-013-0371-z. Epub 2013 Aug 24.
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Identification of a novel homozygous SPG7 mutation in a Japanese patient with spastic ataxia: making an efficient diagnosis using exome sequencing for autosomal recessive cerebellar ataxia and spastic paraplegia.一名日本痉挛性共济失调患者中新型纯合SPG7突变的鉴定:利用外显子组测序对常染色体隐性小脑共济失调和痉挛性截瘫进行高效诊断
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CLP1 links tRNA metabolism to progressive motor-neuron loss.CLP1 将 tRNA 代谢与进行性运动神经元丧失联系起来。
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Two siblings with a novel variant of EXOSC3 extended phenotypic spectrum of pontocerebellar hypoplasia 1B to an exceptionally mild form.两兄妹携带 EXOSC3 的新型变异,其桥脑小脑发育不全 1B 的表型谱扩展到非常轻微的形式。
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A Drosophila model of Pontocerebellar Hypoplasia reveals a critical role for the RNA exosome in neurons.一个小脑桥脑发育不全的果蝇模型揭示了 RNA 外切酶在神经元中的关键作用。
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Biallelic variants in the RNA exosome gene EXOSC5 are associated with developmental delays, short stature, cerebellar hypoplasia and motor weakness.RNA 外切体基因 EXOSC5 的双等位基因变异与发育迟缓、身材矮小、小脑发育不良和运动无力有关。
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