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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

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.

作者信息

Doi Hiroshi, Ohba Chihiro, Tsurusaki Yoshinori, Miyatake Satoko, Miyake Noriko, Saitsu Hirotomo, Kawamoto Yuko, Yoshida Tamaki, Koyano Shigeru, Suzuki Yume, Kuroiwa Yoshiyuki, Tanaka Fumiaki, Matsumoto Naomichi

机构信息

Department of Human Genetics, Yokohama City University, Japan.

出版信息

Intern Med. 2013;52(14):1629-33. doi: 10.2169/internalmedicine.52.0252. Epub 2013 Jul 15.

Abstract

Autosomal recessive cerebellar ataxias and autosomal recessive hereditary spastic paraplegias are clinically and genetically heterogeneous disorders with diverse neurological and non-neurological features. We herein describe a Japanese patient with a slowly progressive form of ataxia and spastic paraplegia. Using whole exome sequencing, we identified a novel homozygous frameshift mutation in SPG7, encoding paraplegin, in this patient. This is the first report of an SPG7 mutation in the Japanese population. For disorders previously undetected in a particular population, or unrecognized/atypical phenotypes, exome sequencing may facilitate molecular diagnosis.

摘要

常染色体隐性遗传性小脑共济失调和常染色体隐性遗传性痉挛性截瘫是临床和遗传上异质性的疾病,具有多样的神经学和非神经学特征。我们在此描述一名患有缓慢进展型共济失调和痉挛性截瘫的日本患者。通过全外显子组测序,我们在该患者中鉴定出一种编码 paraplegin 的 SPG7 基因中的新型纯合移码突变。这是日本人群中 SPG7 突变的首次报道。对于特定人群中先前未检测到的疾病,或未被识别/非典型的表型,外显子组测序可能有助于分子诊断。

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