Literature DB >> 19349200

Two new mutations in the MT-TW gene leading to the disruption of the secondary structure of the tRNA(Trp) in patients with Leigh syndrome.

Emna Mkaouar-Rebai1, Imen Chamkha, Fatma Kammoun, Thouraya Kammoun, Hajer Aloulou, Mongia Hachicha, Chahnez Triki, Faiza Fakhfakh.   

Abstract

Leigh syndrome is a progressive neurodegenerative disorder occurring in infancy and childhood characterized in most cases by a psychomotor retardation, optic atrophy, ataxia, dystonia, failure to thrive, seizures and respiratory failure. In this study, we performed a systematic sequence analysis of mitochondrial genes associated with LS in Tunisian patients. We sequenced the encoded complex I units: ND2, ND3, ND4, ND5 and ND6 genes and the mitochondrial ATPase 6, tRNA(Val), tRNA(Leu(UUR)), tRNA(Trp) and tRNA(Lys) genes in 10 unrelated patients with Leigh syndrome. We revealed the presence of 34 reported polymorphisms, nine novel nucleotide variants and two new mutations (T5523G and A5559G) in the tested patients. These two mutations were localized in two conserved regions of the tRNA(Trp) and affect, respectively, the D-stem and the T-stem of the mitochondrial tRNA leading to a disruption of the secondary structure of this tRNA. SSP-PCR analysis showed that the T5523G and A5559G mutations were present with respective heteroplasmic rates of 66% and 43 %. We report here the first mutational screening of mitochondrial mutations in Tunisian patients with Leigh syndrome which described two novel mutations associated with this disorder.

Entities:  

Mesh:

Substances:

Year:  2009        PMID: 19349200     DOI: 10.1016/j.ymgme.2009.03.003

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  8 in total

1.  Mitochondrial myopathy associated with a novel 5522G>A mutation in the mitochondrial tRNA(Trp) gene.

Authors:  Ivo Barić; Ksenija Fumić; Danijela Petković Ramadža; Wolfgang Sperl; Franz A Zimmermann; Diana Muačević-Katanec; Zoran Mitrović; Leo Pažanin; Ljerka Cvitanović Šojat; Tihomir Kekez; Zeljko Reiner; Johannes A Mayr
Journal:  Eur J Hum Genet       Date:  2012-12-12       Impact factor: 4.246

2.  Molecular pathomechanisms and cell-type-specific disease phenotypes of MELAS caused by mutant mitochondrial tRNA(Trp).

Authors:  Hideyuki Hatakeyama; Ayako Katayama; Hirofumi Komaki; Ichizo Nishino; Yu-Ichi Goto
Journal:  Acta Neuropathol Commun       Date:  2015-08-22       Impact factor: 7.801

Review 3.  The genetics of Leigh syndrome and its implications for clinical practice and risk management.

Authors:  Ilene S Ruhoy; Russell P Saneto
Journal:  Appl Clin Genet       Date:  2014-11-13

4.  Management of Leigh syndrome due to NDUFAF6 variants.

Authors:  Josef Finsterer; Fulvio A Scorza
Journal:  Mol Genet Metab Rep       Date:  2019-01-30

Review 5.  Clinical and genetic spectrums of 413 North African families with inherited retinal dystrophies and optic neuropathies.

Authors:  Aymane Bouzidi; Hicham Charoute; Majida Charif; Ghita Amalou; Mostafa Kandil; Abdelhamid Barakat; Guy Lenaers
Journal:  Orphanet J Rare Dis       Date:  2022-05-12       Impact factor: 4.303

6.  Next-generation sequencing of Tunisian Leigh syndrome patients reveals novel variations: impact for diagnosis and treatment.

Authors:  Meriem Hechmi; Majida Charif; Ichraf Kraoua; Meriem Fassatoui; Hamza Dallali; Valerie Desquiret-Dumas; Céline Bris; David Goudenège; Cyrine Drissi; Saïd Galaï; Slah Ouerhani; Vincent Procaccio; Patrizia Amati-Bonneau; Sonia Abdelhak; Ilhem Ben Youssef-Turki; Guy Lenaers; Rym Kefi
Journal:  Biosci Rep       Date:  2022-09-30       Impact factor: 3.976

7.  Molecular investigation of pediatric portuguese patients with sensorineural hearing loss.

Authors:  Célia Nogueira; Miguel Coutinho; Cristina Pereira; Alessandra Tessa; Filippo M Santorelli; Laura Vilarinho
Journal:  Genet Res Int       Date:  2011-09-25

8.  Early Onset and Severe Clinical Course Associated with the m.5540G>A Mutation in MT-TW.

Authors:  Jorge L Granadillo; Timothy Moss; Richard A Lewis; Elise G Austin; Howard Kelfer; Jing Wang; Lee-Jun C Wong; Fernando Scaglia
Journal:  Mol Genet Metab Rep       Date:  2014
  8 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.