Literature DB >> 25724872

Parkinsonism, cognitive deficit and behavioural disturbance caused by a novel mutation in the polymerase gamma gene.

Manuel Delgado-Alvarado1, Patricia de la Riva2, Haritz Jiménez-Urbieta3, Belén Gago1, Alazne Gabilondo2, Belén Bornstein4, María Cruz Rodríguez-Oroz5.   

Abstract

Polymerase γ (POLG) is the enzyme responsible for the replication and maintenance of mitochondrial DNA (mtDNA). Mutations in the POLG1 gene can lead to mitochondrial dysfunction, producing a wide range of neurological and non-neurological phenotypes. Neurological manifestations include ataxia, muscular weakness, epilepsy, progressive external ophthalmoplegia (PEO), ptosis, neuropathy, psychiatric disorders and, more rarely, parkinsonism. We present the case of an 80-year old female patient with a history of PEO, ptosis, childish behaviour, obsessive disorder, cognitive decline, and parkinsonism. A comprehensive study showed striatal dopamine deficiency on DaT Scan and ragged red fibres as evidenced by Gomori staining in a biopsy of the biceps brachii. Multiple deletions of mtDNA were detected, and sequencing of the POLG1 gene identified a novel substitution, 2834A>T, in exon 18, changing the p.His945Leu amino acid. In silico analysis using PolyPhen-2 (http://genetics.bwh.hardvard.edu/pph2/) predicted that this change is probably damaging, with a score of 1.0 (0-1).
Copyright © 2015 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Cognitive impairment; Obsessive disorder; POLG; Parkinsonism; Progressive external ophthalmoplegia; Ptosis

Mesh:

Substances:

Year:  2015        PMID: 25724872     DOI: 10.1016/j.jns.2015.02.011

Source DB:  PubMed          Journal:  J Neurol Sci        ISSN: 0022-510X            Impact factor:   3.181


  6 in total

Review 1.  Role of the mitochondrial DNA replication machinery in mitochondrial DNA mutagenesis, aging and age-related diseases.

Authors:  Karen L DeBalsi; Kirsten E Hoff; William C Copeland
Journal:  Ageing Res Rev       Date:  2016-04-30       Impact factor: 10.895

Review 2.  Genes and Pathways Involved in Adult Onset Disorders Featuring Muscle Mitochondrial DNA Instability.

Authors:  Naghia Ahmed; Dario Ronchi; Giacomo Pietro Comi
Journal:  Int J Mol Sci       Date:  2015-08-05       Impact factor: 5.923

3.  Pathogenicity in POLG syndromes: DNA polymerase gamma pathogenicity prediction server and database.

Authors:  Anssi Nurminen; Gregory A Farnum; Laurie S Kaguni
Journal:  BBA Clin       Date:  2017-04-18

Review 4.  Environmental and Genetic Variables Influencing Mitochondrial Health and Parkinson's Disease Penetrance.

Authors:  Alessandra Zanon; Peter P Pramstaller; Andrew A Hicks; Irene Pichler
Journal:  Parkinsons Dis       Date:  2018-03-07

5.  POLG R964C and GBA L444P mutations in familial Parkinson's disease: Case report and literature review.

Authors:  Pei-Chen Hsieh; Chun-Chieh Wang; Chia-Lung Tsai; Yuan-Ming Yeh; Yun Shien Lee; Yih-Ru Wu
Journal:  Brain Behav       Date:  2019-04-02       Impact factor: 2.708

6.  A case of drug-induced parkinsonism and tardive akathisia with e1143g polymerase γ mutation-innocent bystander or a culprit?

Authors:  Pretty Sara Idiculla; Syed Taimour Hussain; Junaid Habib Siddiqui
Journal:  J Clin Transl Res       Date:  2021-05-14
  6 in total

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