نتایج جستجو برای: hereditary ataxia

تعداد نتایج: 100227  

2015
YANMIN SONG YUNHAI LIU NING ZHANG LILI LONG

The aim of the present study was to conduct a familial investigation and provide a genetic diagnosis to a family presenting with spastic paraplegia and clinically diagnosed with hereditary spastic paraplegia (HSP). Blood samples were obtained from the family, and mutations in the gene causing spinocerebellar ataxia type 3 (SCA3)/Machado-Joseph disease (MJD), known as MJD1, were analyzed using t...

2016
Katherine L. Helbig Ulrike B.S. Hedrich Deepali N. Shinde Ilona Krey Anne‐Christin Teichmann Julia Hentschel Julian Schubert Adam C. Chamberlin Robert Huether Hsiao‐Mei Lu Wendy A. Alcaraz Sha Tang Chelsy Jungbluth Sarah L. Dugan Leena Vainionpää Kathrin N. Karle Matthis Synofzik Ludger Schöls Rebecca Schüle Anna‐Elina Lehesjoki Ingo Helbig Holger Lerche Johannes R. Lemke

The hereditary spastic paraplegias (HSPs) are heterogeneous neurodegenerative disorders with over 50 known causative genes. We identified a recurrent mutation in KCNA2 (c.881G>A, p.R294H), encoding the voltage-gated K(+) -channel, KV 1.2, in two unrelated families with HSP, intellectual disability (ID), and ataxia. Follow-up analysis of > 2,000 patients with various neurological phenotypes iden...

Journal: :Journal of medical genetics 2006
S Türkmen O Demirhan K Hoffmann A Diers C Zimmer K Sperling S Mundlos

BACKGROUND Congenital hereditary non-progressive hypoplasia of the cerebellum is a rare condition, frequently associated with other neuropathology such as lissencephaly. Clinically, the condition is associated with variable degrees of mental retardation, microcephaly, seizures, and movement disorders due to ataxia. In severe cases, patients are unable to ambulate independently, but nevertheless...

Journal: :Redox report : communications in free radical research 2003
Dianne J Watters

Ataxia telangiectasia is one of a group of recessive hereditary genomic instability disorders and is characterized by progressive neurodegeneration, immunodeficiency and cancer susceptibility. Heterozygotes for the mutated gene are more susceptible to cancer and to ischaemic heart disease. The affected gene, ATM (ataxia telangiectasia mutated), has been cloned and codes for a protein kinase (AT...

2017
Yasmin Fardghassemi Arnaud Tauffenberger Sarah Gosselin Alex Parker

Background: Polyglutamine expansion diseases are a group of hereditary neurodegenerative disorders that develop when a CAG repeat in the causative genes are unstably expanded above a certain threshold. The expansion of trinucleotide CAG repeats cause hereditary adult-onset neurodegenerative disorders such as Huntington’s disease, dentatorubral-pallidoluysian atrophy, spinobulbar muscular atroph...

2013
Charalampos Tzoulis Stefan Johansson Bjørn Ivar Haukanes Helge Boman Per Morten Knappskog Laurence A. Bindoff

We employed whole exome sequencing to investigate three Norwegian siblings with an autosomal recessive spastic ataxia and epilepsy. All patients were compound heterozygous (c.13352T>C, p.Leu4451Pro; c.6890T>G, p.Leu2297Trp) for mutations in the SACS gene establishing the diagnosis of autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS). The clinical features shown by our patients ...

Journal: :Clinical genetics 2016
S Zeng J Zeng M He X Zeng Y Zhou Z Liu K Xia Q Pan H Jiang L Shen X Yan B Tang J Wang

Spinocerebellar ataxia type 36 (SCA36) is a new SCA subtype recently reported in Japanese and Spanish pedigrees. To assess the frequency and clinical characteristics of SCA36 in patients from Mainland China, we combined the repeat-primed polymerase chain reaction method and Southern blot analysis to detect the GGCCTG hexanucleotide repeats of NOP56 in 364 probands with SCA, 126 probands with he...

2016
Boglarka Bansagi David Lewis-Smith Endre Pal Jennifer Duff Helen Griffin Angela Pyle Juliane S. Müller Gabor Rudas Zsuzsanna Aranyi Hanns Lochmüller Patrick F. Chinnery Rita Horvath

Menkes disease is an X-linked multisystem disorder with epilepsy, kinky hair, and neurodegeneration caused by mutations in the copper transporter ATP7A. Other ATP7A mutations have been linked to juvenile occipital horn syndrome and adult-onset hereditary motor neuropathy.1,2 About 5%-10% of the patients present with "atypical Menkes disease" characterized by longer survival, cerebellar ataxia, ...

2012
Barbara Tomassini Gaetano Arcuri Silvia Fortuni Chiranjeevi Sandi Vahid Ezzatizadeh Carlo Casali Ivano Condò Florence Malisan Sahar Al-Mahdawi Mark Pook Roberto Testi

Friedreich's ataxia (FRDA) is the most common hereditary ataxia, affecting ∼3 in 100 000 individuals in Caucasian populations. It is caused by intronic GAA repeat expansions that hinder the expression of the FXN gene, resulting in defective levels of the mitochondrial protein frataxin. Sensory neurons in dorsal root ganglia (DRG) are particularly damaged by frataxin deficiency. There is no spec...

Journal: :American journal of human genetics 2015
Wolfgang M Schmidt S Lane Rutledge Rebecca Schüle Benjamin Mayerhofer Stephan Züchner Eugen Boltshauser Reginald E Bittner

Hereditary ataxias comprise a group of genetically heterogeneous disorders characterized by clinically variable cerebellar dysfunction and accompanied by involvement of other organ systems. The molecular underpinnings for many of these diseases are widely unknown. Previously, we discovered the disruption of Scyl1 as the molecular basis of the mouse mutant mdf, which is affected by neurogenic mu...

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