Literature DB >> 18403580

Ataxia with oculomotor apraxia type 1 (AOA1): clinical and neuropsychological features in 2 new patients and differential diagnosis.

Stefano D'Arrigo1, Daria Riva, Sara Bulgheroni, Luisa Chiapparini, Barbara Castellotti, Cinzia Gellera, Chiara Pantaleoni.   

Abstract

Ataxia with oculomotor apraxia type 1 (AOA1) is an autosomal recessive disease characterized by early-onset and slowly progressive cerebellar ataxia, areflexia, and peripheral neuropathy. Ocular apraxia is most prominent in the early stage of the disease, by contrast, hypoalbuminemia, hypercholesterolemia, and cognitive impairment are present in the adult stage. AOA1 is caused by a mutation in the APTX gene (9p13.3) encoding a nuclear protein named aprataxin, which is involved in the mechanism of DNA repair. We report here the clinical features of 2 patients with mutations in the APTX gene, and we discuss the differential diagnosis with other forms of hereditary ataxia.

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Year:  2008        PMID: 18403580     DOI: 10.1177/0883073808314959

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


  9 in total

1.  Lack of aprataxin impairs mitochondrial functions via downregulation of the APE1/NRF1/NRF2 pathway.

Authors:  Beatriz Garcia-Diaz; Emanuele Barca; Andrea Balreira; Luis C Lopez; Saba Tadesse; Sindhu Krishna; Ali Naini; Caterina Mariotti; Barbara Castellotti; Catarina M Quinzii
Journal:  Hum Mol Genet       Date:  2015-05-14       Impact factor: 6.150

2.  Clinical presentations of coenzyme q10 deficiency syndrome.

Authors:  Catarina M Quinzii; Valentina Emmanuele; Michio Hirano
Journal:  Mol Syndromol       Date:  2014-07

3.  Cerebellar Ataxia and CoQ10 Deficiency.

Authors:  Catarina M Quinzii; Michio Hirano; Ali Naini
Journal:  J Neurol Disord Stroke       Date:  2013

4.  Ataxia with oculomotor apraxia type1 (AOA1): novel and recurrent aprataxin mutations, coenzyme Q10 analyses, and clinical findings in Italian patients.

Authors:  Barbara Castellotti; Caterina Mariotti; Marco Rimoldi; Roberto Fancellu; Massimo Plumari; Sara Caimi; Graziella Uziel; Nardo Nardocci; Isabella Moroni; Giovanna Zorzi; Davide Pareyson; Daniela Di Bella; Stefano Di Donato; Franco Taroni; Cinzia Gellera
Journal:  Neurogenetics       Date:  2011-04-05       Impact factor: 2.660

5.  Aprataxin localizes to mitochondria and preserves mitochondrial function.

Authors:  Peter Sykora; Deborah L Croteau; Vilhelm A Bohr; David M Wilson
Journal:  Proc Natl Acad Sci U S A       Date:  2011-04-18       Impact factor: 11.205

Review 6.  Heterogeneity of coenzyme Q10 deficiency: patient study and literature review.

Authors:  Valentina Emmanuele; Luis C López; Luis López; Andres Berardo; Ali Naini; Saba Tadesse; Bing Wen; Erin D'Agostino; Martha Solomon; Salvatore DiMauro; Catarina Quinzii; Michio Hirano
Journal:  Arch Neurol       Date:  2012-08

Review 7.  DNA repair abnormalities leading to ataxia: shared neurological phenotypes and risk factors.

Authors:  Edward C Gilmore
Journal:  Neurogenetics       Date:  2014-07-20       Impact factor: 2.660

Review 8.  Primary coenzyme Q10 (CoQ 10) deficiencies and related nephropathies.

Authors:  Fatih Ozaltin
Journal:  Pediatr Nephrol       Date:  2013-06-05       Impact factor: 3.714

9.  SETX mutations are a frequent genetic cause of juvenile and adult onset cerebellar ataxia with neuropathy and elevated serum alpha-fetoprotein.

Authors:  Lorenzo Nanetti; Simona Cavalieri; Viviana Pensato; Alessandra Erbetta; Davide Pareyson; Marta Panzeri; Giovanna Zorzi; Carlo Antozzi; Isabella Moroni; Cinzia Gellera; Alfredo Brusco; Caterina Mariotti
Journal:  Orphanet J Rare Dis       Date:  2013-08-14       Impact factor: 4.123

  9 in total

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