Literature DB >> 23183622

A novel mutation in the aprataxin (APTX) gene in an Iranian individual suffering early-onset ataxia with oculomotor apraxia type 1(AOA1) disease.

Nayereh Nouri1, Narges Nouri2, Omid Aryani3, Behnam Kamalidehghan4, Maryam Sedghi1, Massoud Houshmand5.   

Abstract

BACKGROUND: Ataxia with oculomotor apraxia type 1 (AOA1) shows early onset with autosomal recessive inheritance and is caused by a mutation in the aprataxin (APTX) gene encoding for the APTX protein.
METHODS: In this study, a 7-year-old girl born of a first-cousin consanguineous marriage was described with early-onset progressive ataxia and AOA, with increased cholesterol concentration and decreased albumin concentration in serum. PCR and direct DNA sequencing was performed after DNA extraction.
RESULTS: Sequencing analysis revealed a novel homozygous deletion in c.643 and A>T single nucleotide polymorphism in c.641 in exon 6 of the APTX gene [ENST00000379825].
CONCLUSION: It seems that this region of exon 6 is probably a hot spot; however, no deletions have been reported in exon 6 yet.

Entities:  

Keywords:  Ataxia oculomotor apraxia 1; aprataxin; Iranian

Mesh:

Substances:

Year:  2012        PMID: 23183622      PMCID: PMC3600965          DOI: 10.6091/ibj.1077.2012

Source DB:  PubMed          Journal:  Iran Biomed J        ISSN: 1028-852X


  6 in total

1.  The gene mutated in ataxia-ocular apraxia 1 encodes the new HIT/Zn-finger protein aprataxin.

Authors:  M C Moreira; C Barbot; N Tachi; N Kozuka; E Uchida; T Gibson; P Mendonça; M Costa; J Barros; T Yanagisawa; M Watanabe; Y Ikeda; M Aoki; T Nagata; P Coutinho; J Sequeiros; M Koenig
Journal:  Nat Genet       Date:  2001-10       Impact factor: 38.330

2.  Early-onset ataxia with ocular motor apraxia and hypoalbuminemia is caused by mutations in a new HIT superfamily gene.

Authors:  H Date; O Onodera; H Tanaka; K Iwabuchi; K Uekawa; S Igarashi; R Koike; T Hiroi; T Yuasa; Y Awaya; T Sakai; T Takahashi; H Nagatomo; Y Sekijima; I Kawachi; Y Takiyama; M Nishizawa; N Fukuhara; K Saito; S Sugano; S Tsuji
Journal:  Nat Genet       Date:  2001-10       Impact factor: 38.330

3.  Recessive ataxia with ocular apraxia: review of 22 Portuguese patients.

Authors:  C Barbot; P Coutinho; R Chorão; C Ferreira; J Barros; I Fineza; K Dias; J Monteiro; A Guimarães; P Mendonça; M do Céu Moreira; J Sequeiros
Journal:  Arch Neurol       Date:  2001-02

4.  Cerebellar ataxia with oculomotor apraxia type 1: clinical and genetic studies.

Authors:  Isabelle Le Ber; Maria-Ceù Moreira; Sophie Rivaud-Péchoux; Céline Chamayou; François Ochsner; Thierry Kuntzer; Marc Tardieu; Gérard Saïd; Marie-Odile Habert; Geneviève Demarquay; Christian Tannier; Jean-Marie Beis; Alexis Brice; Michel Koenig; Alexandra Dürr
Journal:  Brain       Date:  2003-09-23       Impact factor: 13.501

5.  Early-onset ataxia with ocular motor apraxia and hypoalbuminemia: the aprataxin gene mutations.

Authors:  H Shimazaki; Y Takiyama; K Sakoe; K Ikeguchi; K Niijima; J Kaneko; M Namekawa; T Ogawa; H Date; S Tsuji; I Nakano; M Nishizawa
Journal:  Neurology       Date:  2002-08-27       Impact factor: 9.910

6.  Aprataxin mutations are a rare cause of early onset ataxia in Germany.

Authors:  Matthias Habeck; Christine Zühlke; Karl H P Bentele; Stephan Unkelbach; Wolfram Kress; Katrin Bürk; Eberhard Schwinger; Yorck Hellenbroich
Journal:  J Neurol       Date:  2004-05       Impact factor: 4.849

  6 in total
  4 in total

1.  Novel APTX Mutation in a Hispanic Subject Affected by Ataxia with Oculomotor Apraxia Type 1.

Authors:  Martin Paucar; Isabel Alonso; Mats Eriksson; Stanislav Beniaminov; Paula Coutinho; Per Svenningsson
Journal:  Mov Disord Clin Pract       Date:  2014-12-10

Review 2.  Chronic oxidative damage together with genome repair deficiency in the neurons is a double whammy for neurodegeneration: Is damage response signaling a potential therapeutic target?

Authors:  Haibo Wang; Prakash Dharmalingam; Velmarini Vasquez; Joy Mitra; Istvan Boldogh; K S Rao; Thomas A Kent; Sankar Mitra; Muralidhar L Hegde
Journal:  Mech Ageing Dev       Date:  2016-09-20       Impact factor: 5.432

3.  Genetic variants in diseases of the extrapyramidal system.

Authors:  Anna Oczkowska; Wojciech Kozubski; Margarita Lianeri; Jolanta Dorszewska
Journal:  Curr Genomics       Date:  2014-02       Impact factor: 2.236

4.  Ataxia Oculomotor Apraxia Type 1 in the Siblings of a Family: A Novel Mutation.

Authors:  Parvaneh Karimzadeh; Simin Khayatzadeh Kakhki; Shaghayegh Sadat Esmail Nejad; Masood Houshmand; Mohammad Ghofrani
Journal:  Iran J Child Neurol       Date:  2017
  4 in total

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