Friedreich ataxia: an overview

被引:243
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
[21]  
Delatycki MB, 1999, ANN NEUROL, V45, P673, DOI 10.1002/1531-8249(199905)45:5<673::AID-ANA20>3.0.CO
[22]  
2-Q
[23]   Sperm DNA analysis in a Friedreich ataxia premutation carrier suggests both meiotic and mitotic expansion in the FRDA gene [J].
Delatycki, MB ;
Paris, D ;
Gardner, RJM ;
Forshaw, K ;
Nicholson, GA ;
Nassif, N ;
Williamson, R ;
Forrest, SM .
JOURNAL OF MEDICAL GENETICS, 1998, 35 (09) :713-716
[24]   LATE-ONSET FRIEDREICHS-DISEASE - CLINICAL-FEATURES AND MAPPING OF MUTATION TO THE FRDA LOCUS [J].
DEMICHELE, G ;
FILLA, A ;
CAVALCANTI, F ;
DIMAIO, L ;
PIANESE, L ;
CASTALDO, I ;
CALABRESE, O ;
MONTICELLI, A ;
VARRONE, S ;
CAMPANELLA, G ;
LEONE, M ;
PANDOLFO, M ;
COCOZZA, S .
JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY, 1994, 57 (08) :977-979
[25]  
DESILVA R, 1909, J NEUROL NEUROSUR PS, V66, P117
[26]   PYRUVATE-CARBOXYLASE ACTIVITY IS NOT ABNORMAL IN FIBROBLASTS OF PATIENTS WITH FRIEDREICHS ATAXIA [J].
DIJKSTRA, UJ ;
TRIJBELS, JMF ;
RUITENBEEK, W ;
BAKKEREN, JAJM ;
JANSSEN, AJM ;
GABREELS, FJM ;
JOOSTEN, EMG .
ANNALS OF NEUROLOGY, 1984, 16 (02) :262-262
[27]   FRIEDREICH ATAXIA AND LOW PYRUVATE-CARBOXYLASE ACTIVITY IN LIVER AND FIBROBLASTS [J].
DIJKSTRA, UJ ;
WILLEMS, JL ;
JOOSTEN, EMG ;
GABREELS, FJM .
ANNALS OF NEUROLOGY, 1983, 13 (03) :325-328
[28]   No association between the Friedreich's ataxia gene and NIDDM in the French population [J].
Dupont, S ;
Dubois, D ;
Vionnet, N ;
Boitard, C ;
Caillat-Zucman, S ;
Timsit, J ;
Froguel, P .
DIABETES, 1998, 47 (10) :1654-1656
[29]   Clinical and genetic abnormalities in patients with Friedreich's ataxia [J].
Durr, A ;
Cossee, M ;
Agid, Y ;
Campuzano, V ;
Mignard, C ;
Penet, C ;
Mandel, JL ;
Brice, A ;
Koenig, M .
NEW ENGLAND JOURNAL OF MEDICINE, 1996, 335 (16) :1169-1175
[30]   Brain phospholipids and fatty acids in Friedreich's ataxia and spinocerebellar atrophy type-1 [J].
Eder, K ;
Kish, SJ ;
Kirchgessner, M ;
Ross, BM .
MOVEMENT DISORDERS, 1998, 13 (05) :813-819