Literature DB >> 8190311

Clinical and morphologic features of a myopathy associated with a point mutation in the mitochondrial tRNA(Pro) gene.

V V Ionasescu1, M Hart, S DiMauro, C T Moraes.   

Abstract

We studied a 9-year-old girl with progressive weakness of her extremities for two years. Her neurologic evaluation showed weakness of proximal muscles but no ophthalmoparesis. With the exception of elevated serum lactic acid, the general blood screen, EMG, nerve conduction velocity tests, and ECG were normal. Light and electron microscopy of a muscle biopsy showed proliferation of mitochondria containing disorganized cristae. Activities of respiratory chain enzymes containing mitochondrial DNA (mtDNA)-encoded subunits were significantly impaired in muscle homogenates. A G-->A transition at position 15990 previously detected in this patient's muscle was not present in peripheral blood cells of her mother or sister. However, the patient's WBCs appeared to contain a very small percentage of mutant mtDNAs, indicating that the mutation may have originated during early embryogenesis.

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Year:  1994        PMID: 8190311     DOI: 10.1212/wnl.44.5.975

Source DB:  PubMed          Journal:  Neurology        ISSN: 0028-3878            Impact factor:   9.910


  6 in total

1.  A novel mitochondrial transfer RNA proline mutation.

Authors:  S Seneca; C Ceuterik-De Groote; R Van Coster; L De Meirleir
Journal:  J Inherit Metab Dis       Date:  2000-12       Impact factor: 4.982

2.  Comprehensive, rapid and sensitive detection of sequence variants of human mitochondrial tRNA genes.

Authors:  Y Michikawa; G Hofhaus; L S Lerman; G Attardi
Journal:  Nucleic Acids Res       Date:  1997-06-15       Impact factor: 16.971

3.  Clinical and laboratory findings in referrals for mitochondrial DNA analysis.

Authors:  P J Lamont; R Surtees; C E Woodward; J V Leonard; N W Wood; A E Harding
Journal:  Arch Dis Child       Date:  1998-07       Impact factor: 3.791

4.  A novel mutation in the mitochondrial tRNA(Pro) gene associated with late-onset ataxia, retinitis pigmentosa, deafness, leukoencephalopathy and complex I deficiency.

Authors:  Paola Da Pozzo; Elena Cardaioli; Edoardo Malfatti; Gian Nicola Gallus; Alessandro Malandrini; Carmen Gaudiano; Gianna Berti; Federica Invernizzi; Massimo Zeviani; Antonio Federico
Journal:  Eur J Hum Genet       Date:  2009-02-18       Impact factor: 4.246

5.  Exercise intolerance, muscle pain and lactic acidaemia associated with a 7497G>A mutation in the tRNASer(UCN) gene.

Authors:  O Grafakou; F A Hol; K Otfried Schwab; M H Siers; H ter Laak; F Trijbels; R Ensenauer; C Boelen; J Smeitink
Journal:  J Inherit Metab Dis       Date:  2003       Impact factor: 4.982

6.  De novo mtDNA point mutations are common and have a low recurrence risk.

Authors:  Suzanne C E H Sallevelt; Christine E M de Die-Smulders; Alexandra T M Hendrickx; Debby M E I Hellebrekers; Irenaeus F M de Coo; Charlotte L Alston; Charlotte Knowles; Robert W Taylor; Robert McFarland; Hubert J M Smeets
Journal:  J Med Genet       Date:  2016-07-22       Impact factor: 6.318

  6 in total

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