Literature DB >> 10633128

Friedreich ataxia: an overview.

M B Delatycki1, R Williamson, S M Forrest.   

Abstract

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.

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Year:  2000        PMID: 10633128      PMCID: PMC1734457          DOI: 10.1136/jmg.37.1.1

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  86 in total

1.  An association between NIDDM and a GAA trinucleotide repeat polymorphism in the X25/frataxin (Friedreich's ataxia) gene.

Authors:  M Ristow; E Giannakidou; J Hebinck; K Busch; M Vorgerd; J Kotzka; B Knebel; J Mueller-Berghaus; C Epplen; A Pfeiffer; C R Kahn; A Doria; W Krone; D Mueller-Wieland
Journal:  Diabetes       Date:  1998-05       Impact factor: 9.461

Review 2.  Iron and copper transport in yeast and its relevance to human disease.

Authors:  C Askwith; J Kaplan
Journal:  Trends Biochem Sci       Date:  1998-04       Impact factor: 13.807

3.  No association between the Friedreich's ataxia gene and NIDDM in the French population.

Authors:  S Dupont; D Dubois; N Vionnet; C Boitard; S Caillat-Zucman; J Timsit; P Froguel
Journal:  Diabetes       Date:  1998-10       Impact factor: 9.461

Review 4.  Prenatal diagnosis of Friedreich ataxia.

Authors:  M Pandolfo; L Montermini
Journal:  Prenat Diagn       Date:  1998-08       Impact factor: 3.050

5.  Inhibitory effects of expanded GAA.TTC triplet repeats from intron I of the Friedreich ataxia gene on transcription and replication in vivo.

Authors:  K Ohshima; L Montermini; R D Wells; M Pandolfo
Journal:  J Biol Chem       Date:  1998-06-05       Impact factor: 5.157

6.  Mutation of the start codon in the FRDA1 gene: linkage analysis of three pedigrees with the ATG to ATT transversion points to a unique common ancestor.

Authors:  C Zühlke; F Laccone; M Cossée; A Kohlschütter; M Koenig; E Schwinger
Journal:  Hum Genet       Date:  1998-07       Impact factor: 4.132

7.  Sperm DNA analysis in a Friedreich ataxia premutation carrier suggests both meiotic and mitotic expansion in the FRDA gene.

Authors:  M B Delatycki; D Paris; R J Gardner; K Forshaw; G A Nicholson; N Nassif; R Williamson; S M Forrest
Journal:  J Med Genet       Date:  1998-09       Impact factor: 6.318

8.  Maturation of wild-type and mutated frataxin by the mitochondrial processing peptidase.

Authors:  H Koutnikova; V Campuzano; M Koenig
Journal:  Hum Mol Genet       Date:  1998-09       Impact factor: 6.150

9.  Mt-Hsp70 homolog, Ssc2p, required for maturation of yeast frataxin and mitochondrial iron homeostasis.

Authors:  S A Knight; N B Sepuri; D Pain; A Dancis
Journal:  J Biol Chem       Date:  1998-07-17       Impact factor: 5.157

10.  Normal serum iron and ferritin concentrations in patients with Friedreich's ataxia.

Authors:  R B Wilson; D R Lynch; K H Fischbeck
Journal:  Ann Neurol       Date:  1998-07       Impact factor: 10.422

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  92 in total

1.  Selective determination of mitochondrial chelatable iron in viable cells with a new fluorescent sensor.

Authors:  Frank Petrat; Daniela Weisheit; Martina Lensen; Herbert de Groot; Reiner Sustmann; Ursula Rauen
Journal:  Biochem J       Date:  2002-02-15       Impact factor: 3.857

2.  Defects in mitochondrial axonal transport and membrane potential without increased reactive oxygen species production in a Drosophila model of Friedreich ataxia.

Authors:  Yujiro Shidara; Peter J Hollenbeck
Journal:  J Neurosci       Date:  2010-08-25       Impact factor: 6.167

3.  A novel deletion-insertion mutation identified in exon 3 of FXN in two siblings with a severe Friedreich ataxia phenotype.

Authors:  Marguerite V Evans-Galea; Louise A Corben; Justin Hasell; Charles A Galea; Michael C Fahey; Desirée du Sart; Martin B Delatycki
Journal:  Neurogenetics       Date:  2011-08-10       Impact factor: 2.660

4.  Stable isotopes and LC-MS for monitoring metabolic disturbances in Friedreich's ataxia platelets.

Authors:  Andrew J Worth; Sankha S Basu; Eric C Deutsch; Wei-Ting Hwang; Nathaniel W Snyder; David R Lynch; Ian A Blair
Journal:  Bioanalysis       Date:  2015       Impact factor: 2.681

5.  Friedreich ataxia is not only a GAA repeats expansion disorder: implications for molecular testing and counselling.

Authors:  Dorota Hoffman-Zacharska; Tomasz Mazurczak; Tomasz Zajkowski; Renata Tataj; Paulina Górka-Skoczylas; Katarzyna Połatyńska; Łukasz Kępczyński; Mariusz Stasiołek; Jerzy Bal
Journal:  J Appl Genet       Date:  2016-02-23       Impact factor: 3.240

6.  Sensitivity of spatiotemporal gait parameters in measuring disease severity in Friedreich ataxia.

Authors:  Sarah C Milne; Darren R Hocking; Nellie Georgiou-Karistianis; Anna Murphy; Martin B Delatycki; Louise A Corben
Journal:  Cerebellum       Date:  2014-12       Impact factor: 3.847

7.  Human frataxin: iron and ferrochelatase binding surface.

Authors:  Krisztina Z Bencze; Taejin Yoon; César Millán-Pacheco; Patrick B Bradley; Nina Pastor; J A Cowan; Timothy L Stemmler
Journal:  Chem Commun (Camb)       Date:  2007-03-28       Impact factor: 6.222

Review 8.  Therapeutic strategies in Friedreich's ataxia.

Authors:  Timothy E Richardson; Heather N Kelly; Amanda E Yu; James W Simpkins
Journal:  Brain Res       Date:  2013-04-13       Impact factor: 3.252

9.  Absence of aprataxin gene mutations in a Greek cohort with sporadic early onset ataxia and normal GAA triplets in frataxin gene.

Authors:  C Daiou; K Christodoulou; G Xiromerisiou; M Panas; E Dardiotis; A Kladi; M Speletas; G Ntaios; A Papadimitriou; A Germenis; Georgios M Hadjigeorgiou
Journal:  Neurol Sci       Date:  2009-12-02       Impact factor: 3.307

10.  Impairment in motor reprogramming in Friedreich ataxia reflecting possible cerebellar dysfunction.

Authors:  Louise A Corben; Martin B Delatycki; John L Bradshaw; Malcolm K Horne; Michael C Fahey; Andrew J Churchyard; Nellie Georgiou-Karistianis
Journal:  J Neurol       Date:  2009-12-03       Impact factor: 4.849

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