Alper Gulay, Narayanan Vinodh
Division of Child Neurology, Department of Pediatrics, Children's Hospital of Pittsburgh, 3705 Fifth Avenue, Pittsburgh, PA 15213, USA.
Pediatr Neurol. 2003 May;28(5):335-41. doi: 10.1016/s0887-8994(03)00004-3.
Friedreich's ataxia, the most common hereditary ataxia, is caused by expansion of a GAA triplet located within the first intron of the frataxin gene on chromosome 9q13. There is a clear correlation between size of the expanded repeat and severity of the phenotype. Frataxin is a mitochondrial protein that plays a role in iron homeostasis. Deficiency of frataxin results in mitochondrial iron accumulation, defects in specific mitochondrial enzymes, enhanced sensitivity to oxidative stress, and eventually free-radical mediated cell death. Friedreich's ataxia is considered a nuclear encoded mitochondrial disease. This review discusses the major and rapid progress made in Friedreich's ataxia from gene mapping and identification of the gene to pathogenesis and encouraging therapeutic implications.
弗里德赖希共济失调是最常见的遗传性共济失调,由位于9号染色体q13区域的铁调素基因第一内含子内的GAA三联体扩增引起。扩增重复序列的大小与表型严重程度之间存在明显的相关性。铁调素是一种在线粒体铁稳态中起作用的线粒体蛋白。铁调素缺乏会导致线粒体铁积累、特定线粒体酶缺陷、对氧化应激的敏感性增强,并最终导致自由基介导的细胞死亡。弗里德赖希共济失调被认为是一种核编码的线粒体疾病。本综述讨论了弗里德赖希共济失调从基因定位、基因鉴定到发病机制以及令人鼓舞的治疗意义等方面取得的重大且快速的进展。