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两个患有2A型遗传性运动感觉神经病的西班牙家庭:临床、电生理及分子学研究结果

Two Spanish families with Charcot-Marie-Tooth type 2A: clinical, electrophysiological and molecular findings.

作者信息

Banchs I, Casasnovas C, Montero J, Martínez-Matos J A, Volpini V

机构信息

Molecular Diagnosis Center of Inherited Diseases, Institut d'Investigacions Biomèdiques de Bellvitge (IDIBELL), L'Hospitalet de Llobregat, Barcelona, Spain.

出版信息

Neuromuscul Disord. 2008 Dec;18(12):974-8. doi: 10.1016/j.nmd.2008.09.006. Epub 2008 Nov 8.

Abstract

Mutations in the Mitofusin 2 (MFN2) gene have been related to the axonal type of Charcot-Marie-Tooth type 2 (CMT 2A). We report the first two Spanish families with CMT 2 and mutations in MFN2 gene. Molecular studies of one family with late onset revealed the novel mutation Arg364Gln. The affected family members presented mild clinical and electrophysiological worsening after 14 years of follow-up. The other family presented an early onset and optic atrophy. Molecular studies revealed the Arg94Gln mutation. This is the first report of a family in which this mutation is related to optic atrophy. Molecular analysis aimed at detecting mutations of MFN2 could be extremely useful in mild axonal neuropathies with slow evolution and indispensable in cases of dominant inheritance or optic atrophy. Population studies of mutations in MFN2 should be undertaken to discover the real frequencies in the Mediterranean area.

摘要

线粒体融合蛋白2(MFN2)基因的突变与2型夏科-马里-图思病(CMT 2A)的轴索性型相关。我们报告了首例两个患有CMT 2且携带MFN2基因突变的西班牙家族。对一个晚发型家族的分子研究发现了新的突变Arg364Gln。经过14年的随访,受影响的家族成员出现了轻度的临床和电生理恶化。另一个家族表现为早发型和视神经萎缩。分子研究发现了Arg94Gln突变。这是首次报道该突变与视神经萎缩相关的家族。旨在检测MFN2基因突变的分子分析对于进展缓慢的轻度轴索性神经病可能极为有用,而在显性遗传或视神经萎缩的病例中则必不可少。应开展MFN2基因突变的群体研究,以发现地中海地区的实际发生率。

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