Literature DB >> 19736010

Progressive myofiber loss with extensive fibro-fatty replacement in a child with mitochondrial DNA depletion syndrome and novel thymidine kinase 2 gene mutations.

James Collins1, Kevin E Bove, David Dimmock, Paula Morehart, Lee-Jun Wong, Brenda Wong.   

Abstract

The mitochondrial DNA depletion syndromes (MDS) are autosomal recessive disorders with a decreased mitochondrial DNA copy number. Mutations in thymidine kinase 2 (TK2) have been responsible for the myopathic form of MDS. We describe a child with congenital muscle weakness who had a progressive mitochondrial myopathy associated with extensive fibro-fatty replacement of myofibers resembling muscular dystrophy. MDS was suspected based upon findings in the initial muscle biopsy. Sequence analysis of the TK2 gene revealed two novel heterozygous mutations: the frame shift mutation, c.255_c.258delAGAA, and the heterozygous missense mutation, c.515G>A, (p.R172Q). This report extends the phenotype and genotype of TK2 defects.

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Year:  2009        PMID: 19736010     DOI: 10.1016/j.nmd.2009.08.002

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  6 in total

1.  Potentially diagnostic electron paramagnetic resonance spectra elucidate the underlying mechanism of mitochondrial dysfunction in the deoxyguanosine kinase deficient rat model of a genetic mitochondrial DNA depletion syndrome.

Authors:  Brian Bennett; Daniel Helbling; Hui Meng; Jason Jarzembowski; Aron M Geurts; Marisa W Friederich; Johan L K Van Hove; Michael W Lawlor; David P Dimmock
Journal:  Free Radic Biol Med       Date:  2016-01-08       Impact factor: 7.376

Review 2.  Defects in mitochondrial DNA replication and human disease.

Authors:  William C Copeland
Journal:  Crit Rev Biochem Mol Biol       Date:  2012 Jan-Feb       Impact factor: 8.250

Review 3.  Mitochondrial DNA depletion syndromes: review and updates of genetic basis, manifestations, and therapeutic options.

Authors:  Ayman W El-Hattab; Fernando Scaglia
Journal:  Neurotherapeutics       Date:  2013-04       Impact factor: 7.620

4.  Case Report: Thymidine Kinase 2 (TK2) Deficiency: A Novel Mutation Associated With Childhood-Onset Mitochondrial Myopathy and Atypical Progression.

Authors:  Arianna Manini; Megi Meneri; Carmelo Rodolico; Stefania Corti; Antonio Toscano; Giacomo Pietro Comi; Olimpia Musumeci; Dario Ronchi
Journal:  Front Neurol       Date:  2022-02-25       Impact factor: 4.003

5.  Pathological Features in Paediatric Patients with TK2 Deficiency.

Authors:  Cristina Jou; Andres Nascimento; Anna Codina; Julio Montoya; Ester López-Gallardo; Sonia Emperador; Eduardo Ruiz-Pesini; Raquel Montero; Daniel Natera-de Benito; Carlos I Ortez; Jesus Marquez; Maria V Zelaya; Alfonso Gutierrez-Mata; Carmen Badosa; Laura Carrera-García; Jesica Expósito-Escudero; Monica Roldán; Yolanda Camara; Ramon Marti; Isidre Ferrer; Cecilia Jimenez-Mallebrera; Rafael Artuch
Journal:  Int J Mol Sci       Date:  2022-09-20       Impact factor: 6.208

6.  Retrospective natural history of thymidine kinase 2 deficiency.

Authors:  Caterina Garone; Robert W Taylor; Andrés Nascimento; Joanna Poulton; Carl Fratter; Cristina Domínguez-González; Julie C Evans; Mariana Loos; Pirjo Isohanni; Anu Suomalainen; Dipak Ram; M Imelda Hughes; Robert McFarland; Emanuele Barca; Carlos Lopez Gomez; Sandeep Jayawant; Neil D Thomas; Adnan Y Manzur; Karin Kleinsteuber; Miguel A Martin; Timothy Kerr; Grainne S Gorman; Ewen W Sommerville; Patrick F Chinnery; Monika Hofer; Christoph Karch; Jeffrey Ralph; Yolanda Cámara; Marcos Madruga-Garrido; Jana Domínguez-Carral; Carlos Ortez; Sonia Emperador; Julio Montoya; Anupam Chakrapani; Joshua F Kriger; Robert Schoenaker; Bruce Levin; John L P Thompson; Yuelin Long; Shamima Rahman; Maria Alice Donati; Salvatore DiMauro; Michio Hirano
Journal:  J Med Genet       Date:  2018-03-30       Impact factor: 6.318

  6 in total

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