Literature DB >> 24265579

A novel POLG gene mutation in a patient with SANDO.

Bulent Kurt1, Ali B Naini, William C Copeland, Jiesheng Lu, Salvatore Dimauro, Michio Hirano.   

Abstract

The human mitochondrial genome is replicated by DNA polymerase γ, which is encoded by polymerase γ gene (POLG1) on chromosome 15q25. Patients with POLG1 mutations usually present as Alpers' syndrome or progressive external ophthalmoplegia. Our patient was a 48-year old woman with sensory ataxic neuropathy, dysarthria, ophthalmoplegia, and dysphagia. Sequence analysis revealed that she has two heterozygous missense mutations in the POLG1, a c.1774C>T substitution in exon 10, which results in a p.L591F amino acid change; and a c.3286C>T substitution in exon 21, which results in a p.R1096C amino acid change. The 1774C>T substitution is a novel mutation. Previously described adult patients with one mutation in exon 10 and the other in exon 21 of POLG1 had presented with progressive external ophthalmoplegia. We now describe a patient with mutations in the same exons but suffering from the more complex clinical syndrome of sensory ataxic neuropathy, dysarthria, ophthalmoplegia.

Entities:  

Keywords:  Novel mutation; PEO; POLG1; SANDO

Year:  2012        PMID: 24265579      PMCID: PMC3832984          DOI: 10.5455/jeim.200312.cr.001

Source DB:  PubMed          Journal:  J Exp Integr Med        ISSN: 1309-4572


  8 in total

Review 1.  DNA polymerase gamma, the mitochondrial replicase.

Authors:  Laurie S Kaguni
Journal:  Annu Rev Biochem       Date:  2004       Impact factor: 23.643

2.  Phenotypic spectrum associated with mutations of the mitochondrial polymerase gamma gene.

Authors:  Rita Horvath; Gavin Hudson; Gianfrancesco Ferrari; Nancy Fütterer; Sofia Ahola; Eleonora Lamantea; Holger Prokisch; Hanns Lochmüller; Robert McFarland; V Ramesh; Thomas Klopstock; Peter Freisinger; Fabrizio Salvi; Johannes A Mayr; Rene Santer; Marketa Tesarova; Jiri Zeman; Bjarne Udd; Robert W Taylor; Douglass Turnbull; Michael Hanna; Doreen Fialho; Anu Suomalainen; Massimo Zeviani; Patrick F Chinnery
Journal:  Brain       Date:  2006-04-18       Impact factor: 13.501

3.  Mutation of POLG is associated with progressive external ophthalmoplegia characterized by mtDNA deletions.

Authors:  G Van Goethem; B Dermaut; A Löfgren; J J Martin; C Van Broeckhoven
Journal:  Nat Genet       Date:  2001-07       Impact factor: 38.330

Review 4.  Replication of animal mitochondrial DNA.

Authors:  D A Clayton
Journal:  Cell       Date:  1982-04       Impact factor: 41.582

5.  Mutations of ANT1, Twinkle, and POLG1 in sporadic progressive external ophthalmoplegia (PEO).

Authors:  A Agostino; L Valletta; P F Chinnery; G Ferrari; F Carrara; R W Taylor; A M Schaefer; D M Turnbull; V Tiranti; M Zeviani
Journal:  Neurology       Date:  2003-04-22       Impact factor: 9.910

6.  Molecular and clinical genetics of mitochondrial diseases due to POLG mutations.

Authors:  Lee-Jun C Wong; Robert K Naviaux; Nicola Brunetti-Pierri; Qing Zhang; Eric S Schmitt; Cavatina Truong; Margherita Milone; Bruce H Cohen; Beverly Wical; Jaya Ganesh; Alice A Basinger; Barbara K Burton; Kathryn Swoboda; Donald L Gilbert; Adeline Vanderver; Russell P Saneto; Bruno Maranda; Georgianne Arnold; Jose E Abdenur; Paula J Waters; William C Copeland
Journal:  Hum Mutat       Date:  2008-09       Impact factor: 4.878

7.  Analysis of mutant DNA polymerase gamma in patients with mitochondrial DNA depletion.

Authors:  Jan-Willem Taanman; Shamima Rahman; Alistair T Pagnamenta; Andrew A M Morris; Maria Bitner-Glindzicz; Nicole I Wolf; James V Leonard; Peter T Clayton; Anthony H V Schapira
Journal:  Hum Mutat       Date:  2009-02       Impact factor: 4.878

8.  Depletion of mitochondrial DNA in fibroblast cultures from patients with POLG1 mutations is a consequence of catalytic mutations.

Authors:  Neil Ashley; Anthony O'Rourke; Conrad Smith; Susan Adams; Vasantha Gowda; Massimo Zeviani; Garry K Brown; Carl Fratter; Joanna Poulton
Journal:  Hum Mol Genet       Date:  2008-05-16       Impact factor: 6.150

  8 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.