Friedreich ataxia: an overview

被引:236
作者
Delatycki, MB [1 ]
Williamson, R [1 ]
Forrest, SM [1 ]
机构
[1] Royal Childrens Hosp, Murdoch Inst, Parkville, Vic 3052, Australia
关键词
Friedreich ataxia; FRDA gene;
D O I
10.1136/jmg.37.1.1
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
Friedreich ataxia, an autosomal recessive neurodegenerative disease, is the most common of the inherited ataxias. The recent discovery of the gene that is mutated in this condition, FRDA, has led to rapid advances in the understanding of the pathogenesis of Friedreich ataxia. About 98% of mutant alleles have an expansion of a GAA trinucleotide repeat in intron 1 of the gene. This leads to reduced levels of the protein, frataxin. There is mounting evidence to suggest that Friedreich ataxia is the result of accumulation of iron in mitochondria leading to excess production of free radicals, which then results in cellular damage and death. Currently there is no known treatment that alters the natural course of the disease. The discovery of the FRDA gene and its possible function has raised hope that rational therapeutic strategies will be developed.
引用
收藏
页码:1 / 8
页数:8
相关论文
共 103 条
  • [1] ANDERMANN E, 1976, Canadian Journal of Neurological Sciences, V3, P287
  • [2] Iron and copper transport in yeast and its relevance to human disease
    Askwith, C
    Kaplan, J
    [J]. TRENDS IN BIOCHEMICAL SCIENCES, 1998, 23 (04) : 135 - 138
  • [3] Regulation of mitochondrial iron accumulation by Yfh1p, a putative homolog of frataxin
    Babcock, M
    deSilva, D
    Oaks, R
    DavisKaplan, S
    Jiralerspong, S
    Montermini, L
    Pandolfo, M
    Kaplan, J
    [J]. SCIENCE, 1997, 276 (5319) : 1709 - 1712
  • [4] Bell J, 1939, TREASURY HUMAN INH 3, V4, P141
  • [5] Intronic GAA triplet repeat expansion in Friedreich's ataxia presenting with pure sensory ataxia
    Berciano, J
    Combarros, O
    DeCastro, M
    Palau, F
    [J]. JOURNAL OF NEUROLOGY, 1997, 244 (06) : 390 - 391
  • [6] The GAA triplet-repeat expansion in Friedreich ataxia interferes with transcription and may be associated with an unusual DNA structure
    Bidichandani, SI
    Ashizawa, T
    Patel, PI
    [J]. AMERICAN JOURNAL OF HUMAN GENETICS, 1998, 62 (01) : 111 - 121
  • [7] Bidichandani SI, 1997, AM J HUM GENET, V60, P1251
  • [8] Mitochondrial intermediate peptidase and the yeast frataxin homolog together maintain mitochondrial iron homeostasis in Saccharomyces cerevisiae
    Branda, SS
    Yang, ZY
    Chew, A
    Isaya, G
    [J]. HUMAN MOLECULAR GENETICS, 1999, 8 (06) : 1099 - 1110
  • [9] Friedreich's ataxia: Autosomal recessive disease caused by an intronic GAA triplet repeat expansion
    Campuzano, V
    Montermini, L
    Molto, MD
    Pianese, L
    Cossee, M
    Cavalcanti, F
    Monros, E
    Rodius, F
    Duclos, F
    Monticelli, A
    Zara, F
    Canizares, J
    Koutnikova, H
    Bidichandani, SI
    Gellera, C
    Brice, A
    Trouillas, P
    DeMichele, G
    Filla, A
    DeFrutos, R
    Palau, F
    Patel, PI
    DiDonato, S
    Mandel, JL
    Cocozza, S
    Koenig, M
    Pandolfo, M
    [J]. SCIENCE, 1996, 271 (5254) : 1423 - 1427
  • [10] Frataxin is reduced in Friedreich ataxia patients and is associated with mitochondrial membranes
    Campuzano, V
    Montermini, L
    Lutz, Y
    Cova, L
    Hindelang, C
    Jiralerspong, S
    Trottier, Y
    Kish, SJ
    Faucheux, B
    Trouillas, P
    Authier, FJ
    Durr, A
    Mandel, JL
    Vescovi, A
    Pandolfo, M
    Koenig, M
    [J]. HUMAN MOLECULAR GENETICS, 1997, 6 (11) : 1771 - 1780