نتایج جستجو برای: ataxia oculomotor apraxia 1 aoa1

تعداد نتایج: 2770963  

Journal: : 2021

Objective: This study aims to determine genotype-phenotype characteristics that can help diagnose hereditary ataxia, a rare disease. Methods: The findings of clinical, laboratory, electrophysiological, and magnetic resonance imaging thirteen patients with ataxia in the last five years were reported this study. Phenotypic expressions genetically proved mutation also reviewed. Results: We report ...

2017
Minwoo Lee Nan Young Kim Jin Young Huh Young Eun Kim Yun Joong Kim

This corrects the article on p. 126 in vol. 12, PMID: 26541496.

Journal: :Current Biology 2007
David M. Wilson Mark P. Mattson

Ataxia oculomotor apraxia-1 is a neurological disorder that arises from mutations in the gene encoding the protein aprataxin. A recent study demonstrates that aprataxin is critical for the processing of obstructive DNA termini, suggesting a broader role for DNA single-strand break repair in neurodegenerative disease.

2012
Hok Khim Fam Miraj K. Chowdhury Cornelius F. Boerkoel

Spinocerebellar ataxias (SCAs) are a group of progressive and irreversible neurological diseases affecting gait and movement coordination. Many result from cerebellar degeneration or the impairment of a portion of the neuroaxis that contributes to cerebellar inflow or outflow (Embirucu et al., 2009). In the cerebellum, the dysfunction and death of Purkinje cells, granule cells or interneurons c...

Journal: :The Journal of Cell Biology 2007
Amila Suraweera Olivier J. Becherel Philip Chen Natalie Rundle Rick Woods Jun Nakamura Magtouf Gatei Chiara Criscuolo Alessandro Filla Luciana Chessa Markus Fußer Bernd Epe Nuri Gueven Martin F. Lavin

A defective response to DNA damage is observed in several human autosomal recessive ataxias with oculomotor apraxia, including ataxia-telangiectasia. We report that senataxin, defective in ataxia oculomotor apraxia (AOA) type 2, is a nuclear protein involved in the DNA damage response. AOA2 cells are sensitive to H2O2, camptothecin, and mitomycin C, but not to ionizing radiation, and sensitivit...

Journal: :Brain : a journal of neurology 2009
M Anheim B Monga M Fleury P Charles C Barbot M Salih J P Delaunoy M Fritsch L Arning M Synofzik L Schöls J Sequeiros C Goizet C Marelli I Le Ber J Koht J Gazulla J De Bleecker M Mukhtar N Drouot L Ali-Pacha T Benhassine M Chbicheb A M'Zahem A Hamri B Chabrol J Pouget R Murphy M Watanabe P Coutinho M Tazir A Durr A Brice C Tranchant M Koenig

Ataxia with oculomotor apraxia type 2 (AOA2) is an autosomal recessive disease due to mutations in the senataxin gene, causing progressive cerebellar ataxia with peripheral neuropathy, cerebellar atrophy, occasional oculomotor apraxia and elevated alpha-feto-protein (AFP) serum level. We compiled a series of 67 previously reported and 58 novel ataxic patients who underwent senataxin gene sequen...

Journal: :Journal of medical genetics 2004
G J Breedveld B van Wetten G D te Raa E Brusse J C van Swieten B A Oostra J A Maat-Kievit

T he cerebellar ataxias are a heterogeneous group of neurodegenerative disorders, characterised by symptoms and signs of cerebellar degeneration, pyramidal and extrapyramidal features, and variable polyneuropathy. Prominent clinical features are signs of cerebellar ataxia, such as uncoordinated gait and uncontrolled co-ordination of hand, speech, and eye movements, while (extra) pyramidal signs...

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