Literature DB >> 18195149

Juvenile Alpers disease.

Esko Wiltshire1, Guido Davidzon, Salvatore DiMauro, Hasan O Akman, Lynette Sadleir, Lindsay Haas, Jane Zuccollo, Alison McEwen, David R Thorburn.   

Abstract

BACKGROUND: Alpers disease is commonly associated with polymerase gamma deficiency and usually affects infants or young children.
OBJECTIVE: To report a juvenile case of Alpers disease due to mutations in the polymerase gamma gene (POLG1).
DESIGN: Clinical, pathologic, biochemical, and molecular analysis.
SETTING: Tertiary care university hospital and academic institutions. PATIENT: A 17-year-old adolescent girl with intractable epilepsy and liver disease. MAIN OUTCOME MEASURES: Clinical course and pathologic, biochemical, and molecular features.
RESULTS: Biochemical and pathologic evidence suggested a respiratory chain defect, which was confirmed by enzyme analysis of the liver. Mutational analysis of POLG1 showed 2 novel mutations: T851A and R1047W.
CONCLUSION: The POLG1 mutations can cause juvenile and childhood Alpers disease.

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Year:  2008        PMID: 18195149     DOI: 10.1001/archneurol.2007.14

Source DB:  PubMed          Journal:  Arch Neurol        ISSN: 0003-9942


  12 in total

Review 1.  Mitochondrial DNA replication and disease: insights from DNA polymerase γ mutations.

Authors:  Jeffrey D Stumpf; William C Copeland
Journal:  Cell Mol Life Sci       Date:  2010-10-08       Impact factor: 9.261

2.  Disease mutations in the human mitochondrial DNA polymerase thumb subdomain impart severe defects in mitochondrial DNA replication.

Authors:  Rajesh Kasiviswanathan; Matthew J Longley; Sherine S L Chan; William C Copeland
Journal:  J Biol Chem       Date:  2009-05-28       Impact factor: 5.157

Review 3.  Alpers-Huttenlocher syndrome.

Authors:  Russell P Saneto; Bruce H Cohen; William C Copeland; Robert K Naviaux
Journal:  Pediatr Neurol       Date:  2013-03       Impact factor: 3.372

Review 4.  Mitochondrial genome maintenance in health and disease.

Authors:  William C Copeland; Matthew J Longley
Journal:  DNA Repair (Amst)       Date:  2014-04-26

5.  mip1 containing mutations associated with mitochondrial disease causes mutagenesis and depletion of mtDNA in Saccharomyces cerevisiae.

Authors:  Jeffrey D Stumpf; Christopher M Bailey; Diana Spell; Matthew Stillwagon; Karen S Anderson; William C Copeland
Journal:  Hum Mol Genet       Date:  2010-02-25       Impact factor: 6.150

6.  POLG DNA testing as an emerging standard of care before instituting valproic acid therapy for pediatric seizure disorders.

Authors:  Russell P Saneto; Inn-Chi Lee; Mary Kay Koenig; Xinhua Bao; Shao-Wen Weng; Robert K Naviaux; Lee-Jun C Wong
Journal:  Seizure       Date:  2010-02-06       Impact factor: 3.184

7.  Yeast model analysis of novel polymerase gamma variants found in patients with autosomal recessive mitochondrial disease.

Authors:  Magdalena Kaliszewska; Jakub Kruszewski; Biruta Kierdaszuk; Anna Kostera-Pruszczyk; Monika Nojszewska; Anna Łusakowska; Joel Vizueta; Dorota Sabat; Dorota Lutyk; Michał Lower; Dorota Piekutowska-Abramczuk; Aneta Kaniak-Golik; Ewa Pronicka; Anna Kamińska; Ewa Bartnik; Paweł Golik; Katarzyna Tońska
Journal:  Hum Genet       Date:  2015-06-16       Impact factor: 4.132

Review 8.  Alpers-Huttenlocher syndrome: the role of a multidisciplinary health care team.

Authors:  Russell P Saneto
Journal:  J Multidiscip Healthc       Date:  2016-07-26

Review 9.  POLG-related disorders and their neurological manifestations.

Authors:  Shamima Rahman; William C Copeland
Journal:  Nat Rev Neurol       Date:  2019-01       Impact factor: 42.937

10.  Dissecting the neuronal vulnerability underpinning Alpers' syndrome: a clinical and neuropathological study.

Authors:  Hannah Hayhurst; Maria-Eleni Anagnostou; Helen J Bogle; John P Grady; Robert W Taylor; Laurence A Bindoff; Robert McFarland; Doug M Turnbull; Nichola Z Lax
Journal:  Brain Pathol       Date:  2018-10-09       Impact factor: 6.508

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