Literature DB >> 22752493

Clinical features of Friedreich ataxia.

Martin B Delatycki1, Louise A Corben.   

Abstract

Friedreich ataxia, the most common hereditary ataxia, affects approximately 1 per 29,000 white individuals. In about 98% of these individuals, it is due to homozygosity for a GAA trinucleotide repeat expansion in intron 1 of FXN; in the other 2%, it is due to compound heterozygosity for a GAA expansion and point mutation or deletion. The condition affects multiple sites in the central and peripheral nervous system as well as a number of other organ systems, resulting in multiple signs and symptoms. Onset of this autosomal recessive condition is usually in the first 2 decades of life. Major clinical features include progressive ataxia, absent lower limb reflexes, upgoing plantar responses, and peripheral sensory neuropathy. The main nonneurological sites of morbidity are the heart, resulting in cardiomyopathy, and the pancreas, resulting in diabetes mellitus. In this review, we provide an overview of the clinical features of Friedreich ataxia and discuss differential diagnoses.

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Year:  2012        PMID: 22752493      PMCID: PMC3674491          DOI: 10.1177/0883073812448230

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  41 in total

1.  Analysis of echocardiograms in a large heterogeneous cohort of patients with friedreich ataxia.

Authors:  Sean R Regner; Sarah J Lagedrost; Ted Plappert; Erin K Paulsen; Lisa S Friedman; Madeline L Snyder; Susan L Perlman; Katherine D Mathews; George R Wilmot; Kimberly A Schadt; Martin St John Sutton; David R Lynch
Journal:  Am J Cardiol       Date:  2011-11-10       Impact factor: 2.778

2.  Utilisation of advance motor information is impaired in Friedreich ataxia.

Authors:  Louise A Corben; Martin B Delatycki; John L Bradshaw; Andrew J Churchyard; Nellie Georgiou-Karistianis
Journal:  Cerebellum       Date:  2011-12       Impact factor: 3.847

3.  Cardiomyopathy of Friedreich's ataxia: use of mouse models to understand human disease and guide therapeutic development.

Authors:  R Mark Payne; P Melanie Pride; Clifford M Babbey
Journal:  Pediatr Cardiol       Date:  2011-03-01       Impact factor: 1.655

4.  Early changes in left ventricular long-axis function in Friedreich ataxia: relation with the FXN gene mutation and cardiac structural change.

Authors:  Philip M Mottram; Martin B Delatycki; Lesley Donelan; John S Gelman; Louise Corben; Roger E Peverill
Journal:  J Am Soc Echocardiogr       Date:  2011-05-12       Impact factor: 5.251

5.  G130V, a common FRDA point mutation, appears to have arisen from a common founder.

Authors:  M B Delatycki; M Knight; M Koenig; M Cossée; R Williamson; S M Forrest
Journal:  Hum Genet       Date:  1999-10       Impact factor: 4.132

6.  Clinical and genetic study of Friedreich ataxia in an Australian population.

Authors:  M B Delatycki; D B Paris; R J Gardner; G A Nicholson; N Nassif; E Storey; J C MacMillan; V Collins; R Williamson; S M Forrest
Journal:  Am J Med Genet       Date:  1999-11-19

7.  The Fitts task reveals impairments in planning and online control of movement in Friedreich ataxia: reduced cerebellar-cortico connectivity?

Authors:  L A Corben; N Georgiou-Karistianis; J L Bradshaw; D R Hocking; A J Churchyard; M B Delatycki
Journal:  Neuroscience       Date:  2011-06-25       Impact factor: 3.590

8.  Mortality in Friedreich ataxia.

Authors:  Amy Y Tsou; Erin K Paulsen; Sarah J Lagedrost; Susan L Perlman; Katherine D Mathews; George R Wilmot; Bernard Ravina; Arnulf H Koeppen; David R Lynch
Journal:  J Neurol Sci       Date:  2011-06-08       Impact factor: 3.181

9.  Successful treatment of auditory perceptual disorder in individuals with Friedreich ataxia.

Authors:  G Rance; L A Corben; E Du Bourg; A King; M B Delatycki
Journal:  Neuroscience       Date:  2010-09-17       Impact factor: 3.590

10.  The test of everyday attention reveals significant sustained volitional attention and working memory deficits in friedreich ataxia.

Authors:  Felicity Klopper; Martin B Delatycki; Louise A Corben; John L Bradshaw; Gary Rance; Nellie Georgiou-Karistianis
Journal:  J Int Neuropsychol Soc       Date:  2010-11-17       Impact factor: 2.892

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  58 in total

Review 1.  Synthesis, delivery and regulation of eukaryotic heme and Fe-S cluster cofactors.

Authors:  Dulmini P Barupala; Stephen P Dzul; Pamela Jo Riggs-Gelasco; Timothy L Stemmler
Journal:  Arch Biochem Biophys       Date:  2016-01-16       Impact factor: 4.013

Review 2.  Milestones in Friedreich ataxia: more than a century and still learning.

Authors:  Agessandro Abrahão; José Luiz Pedroso; Pedro Braga-Neto; Edson Bor-Seng-Shu; Patricia de Carvalho Aguiar; Orlando Graziani Povoas Barsottini
Journal:  Neurogenetics       Date:  2015-02-08       Impact factor: 2.660

3.  Can Latent Class Analysis Be Used to Improve the Diagnostic Process in Pediatric Patients with Chronic Ataxia?

Authors:  Samantha Klassen; Brenden Dufault; Michael S Salman
Journal:  Cerebellum       Date:  2017-04       Impact factor: 3.847

4.  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

5.  Health-related quality of life and depressive symptoms in Friedreich ataxia.

Authors:  Javier Pérez-Flores; Atteneri Hernández-Torres; Fernando Montón; Antonieta Nieto
Journal:  Qual Life Res       Date:  2019-09-28       Impact factor: 4.147

6.  Application of Quantitative Motor Assessments in Friedreich Ataxia and Evaluation of Their Relation to Clinical Measures.

Authors:  Christian Hohenfeld; Imis Dogan; Robin Schubert; Claire Didszun; Ludger Schöls; Matthis Synofzik; Ilaria A Giordano; Thomas Klockgether; Jörg B Schulz; Ralf Reilmann; Kathrin Reetz
Journal:  Cerebellum       Date:  2019-10       Impact factor: 3.847

7.  Excision of Expanded GAA Repeats Alleviates the Molecular Phenotype of Friedreich's Ataxia.

Authors:  Yanjie Li; Urszula Polak; Angela D Bhalla; Natalia Rozwadowska; Jill Sergesketter Butler; David R Lynch; Sharon Y R Dent; Marek Napierala
Journal:  Mol Ther       Date:  2015-03-11       Impact factor: 11.454

8.  Hypoxia Rescues Frataxin Loss by Restoring Iron Sulfur Cluster Biogenesis.

Authors:  Tslil Ast; Joshua D Meisel; Shachin Patra; Hong Wang; Robert M H Grange; Sharon H Kim; Sarah E Calvo; Lauren L Orefice; Fumiaki Nagashima; Fumito Ichinose; Warren M Zapol; Gary Ruvkun; David P Barondeau; Vamsi K Mootha
Journal:  Cell       Date:  2019-04-25       Impact factor: 41.582

9.  Dysphagia in Friedreich Ataxia.

Authors:  Megan J Keage; Martin B Delatycki; Isabelle Gupta; Louise A Corben; Adam P Vogel
Journal:  Dysphagia       Date:  2017-05-04       Impact factor: 3.438

10.  Friedreich Ataxia in Classical Galactosaemia.

Authors:  Siobhán Neville; Siobhan O'Sullivan; Bronagh Sweeney; Bryan Lynch; Donncha Hanrahan; Ina Knerr; Sally Ann Lynch; Ellen Crushell
Journal:  JIMD Rep       Date:  2015-07-29
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