Literature DB >> 3239952

Ataxia-ocular motor apraxia: a syndrome mimicking ataxia-telangiectasia.

J Aicardi1, C Barbosa, E Andermann, F Andermann, R Morcos, Q Ghanem, Y Fukuyama, Y Awaya, P Moe.   

Abstract

We report 14 patients with a slowly progressive syndrome featuring ataxia, choreoathetosis, and ocular motor apraxia in both the horizontal and vertical planes. Although the neurological signs were indistinguishable from those of ataxia-telangiectasia, the onset tended to be later and none of the patients had evidence of multisystemic involvement. Specifically, there was no tendency to frequent infections, and immunoglobulins, alpha-fetoprotein, T- and B-lymphocyte markers, and chromosomes 7 and 14 were normal in all tested patients. The simultaneous absence of telangiectasias and of other nonneurological manifestations made ataxia-telangiectasia an unlikely diagnosis. We suggest that these patients suffer from an unusual type of spinocerebellar degeneration. This syndrome has been observed in different populations from three continents, with a genetic pattern suggesting recessive autosomal inheritance.

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Year:  1988        PMID: 3239952     DOI: 10.1002/ana.410240404

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  27 in total

1.  Hint, Fhit, and GalT: function, structure, evolution, and mechanism of three branches of the histidine triad superfamily of nucleotide hydrolases and transferases.

Authors:  Charles Brenner
Journal:  Biochemistry       Date:  2002-07-23       Impact factor: 3.162

Review 2.  Mitochondrial DNA damage and its consequences for mitochondrial gene expression.

Authors:  Susan D Cline
Journal:  Biochim Biophys Acta       Date:  2012-06-19

Review 3.  Molecular underpinnings of Aprataxin RNA/DNA deadenylase function and dysfunction in neurological disease.

Authors:  Matthew J Schellenberg; Percy P Tumbale; R Scott Williams
Journal:  Prog Biophys Mol Biol       Date:  2015-01-29       Impact factor: 3.667

4.  Crystal structures of aprataxin ortholog Hnt3 reveal the mechanism for reversal of 5'-adenylated DNA.

Authors:  Yong Gong; Deyu Zhu; Jingjin Ding; Chuan-Na Dou; Xiaoming Ren; Lichuan Gu; Tao Jiang; Da-Cheng Wang
Journal:  Nat Struct Mol Biol       Date:  2011-10-09       Impact factor: 15.369

Review 5.  Molecular pathology of ataxia telangiectasia.

Authors:  A M R Taylor; P J Byrd
Journal:  J Clin Pathol       Date:  2005-10       Impact factor: 3.411

Review 6.  DNA repair deficiency and neurological disease.

Authors:  Peter J McKinnon
Journal:  Nat Rev Neurosci       Date:  2009-01-15       Impact factor: 34.870

7.  Intermittent horizontal saccade failure ('ocular motor apraxia') in children.

Authors:  C M Harris; F Shawkat; I Russell-Eggitt; J Wilson; D Taylor
Journal:  Br J Ophthalmol       Date:  1996-02       Impact factor: 4.638

8.  Autosomal recessive spinocerebellar ataxia and peripheral neuropathy with raised alpha-fetoprotein.

Authors:  Louise Izatt; Andrea H Németh; Anjela Meesaq; Kerry R Mills; A Malcolm R Taylor; Christopher E Shaw
Journal:  J Neurol       Date:  2004-07       Impact factor: 4.849

9.  Ataxia-ocular motor apraxia syndrome: an investigation of cellular radiosensitivity of patients and their families.

Authors:  M A Hannan; D Sigut; M Waghray; G G Gascon
Journal:  J Med Genet       Date:  1994-12       Impact factor: 6.318

10.  CK2 phosphorylation-dependent interaction between aprataxin and MDC1 in the DNA damage response.

Authors:  Olivier J Becherel; Burkhard Jakob; Amy L Cherry; Nuri Gueven; Markus Fusser; Amanda W Kijas; Cheng Peng; Sachin Katyal; Peter J McKinnon; Junjie Chen; Bernd Epe; Stephen J Smerdon; Gisela Taucher-Scholz; Martin F Lavin
Journal:  Nucleic Acids Res       Date:  2009-12-14       Impact factor: 16.971

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