نتایج جستجو برای: aoa1

تعداد نتایج: 38  

Journal: :Orphanet Journal of Rare Diseases 2006
Francesc Palau Carmen Espinós

Autosomal recessive cerebellar ataxias (ARCA) are a heterogeneous group of rare neurological disorders involving both central and peripheral nervous system, and in some case other systems and organs, and characterized by degeneration or abnormal development of cerebellum and spinal cord, autosomal recessive inheritance and, in most cases, early onset occurring before the age of 20 years. This g...

2015
Mansour Akbari Peter Sykora Vilhelm A. Bohr

Aborted DNA ligation events in eukaryotic cells can generate 5'-adenylated (5'-AMP) DNA termini that can be removed from DNA by aprataxin (APTX). Mutations in APTX cause an inherited human disease syndrome characterized by early-onset progressive ataxia with ocular motor apraxia (AOA1). APTX is found in the nuclei and mitochondria of eukaryotic cells. Depletion of APTX causes mitochondrial dysf...

Journal: :Journal of neurology, neurosurgery, and psychiatry 2018
Emer O'Connor Jana Vandrovcova Enrico Bugiardini Viorica Chelban Andreea Manole Indran Davagnanam Sarah Wiethoff Alan Pittman David S Lynch Stephanie Efthymiou Silvia Marino Adnan Y Manzur Mark Roberts Michael G Hanna Henry Houlden Emma Matthews Nicholas W Wood

Letter Mutations in genes involved in singlestrand break repair (SSBR) have been linked to hereditary cerebellar ataxias. Notably, Ataxia-oculomotor apraxia 1 (AOA1 (MIM: 2 08 920)), Spinocerebellar ataxia with axonal neuropathy 1 (SCAN1 (MIM: 6 07 250)) and Ataxia-oculomotor apraxia 4 (AOA4 (MIM: 616267616267616267)) are associated with mutations in APTX (MIM: 606350), TDP1 (MIM: 607198) and P...

2013
Morten Scheibye-Knudsen Karsten Scheibye-Alsing Chandrika Canugovi Deborah L. Croteau Vilhelm A. Bohr

The inherent complex and pleiotropic phenotype of mitochondrial diseases poses a significant diagnostic challenge for clinicians as well as an analytical barrier for scientists. To overcome these obstacles we compiled a novel database, www.mitodb.com, containing the clinical features of primary mitochondrial diseases. Based on this we developed a number of qualitative and quantitative measures,...

2012
Fabio Coppedè

Oxidative DNA damage is one of the earliest detectable events in several neurodegenerative diseases, often preceding the onset of the clinical symptoms. Moreover, neurons in the adult human brain can re-enter the cell division cycle, likely allowing DNA repair. Impairments of DNA repair pathways are reported in neurons of patients suffering from one of several neurodegenative diseases and might...

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...

2004
G J Breedveld B van Wetten 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...

Journal: :Journal of Biological Chemistry 2021

DNA ligase I (LIG1) completes the base excision repair (BER) pathway at last nick-sealing step after polymerase (pol) ? gap-filling synthesis. However, mechanism by which LIG1 fidelity mediates faithful substrate–product channeling and ligation of intermediates final steps BER remains unclear. We previously reported that pol 8-oxo-2'-deoxyribonucleoside 5'-triphosphate insertion confounds LIG1,...

نمودار تعداد نتایج جستجو در هر سال

با کلیک روی نمودار نتایج را به سال انتشار فیلتر کنید