Literature DB >> 20135231

An overview of a cohort of South African patients with mitochondrial disorders.

Izelle Smuts1, Roan Louw, Hanli du Toit, Brenda Klopper, Lodewyk J Mienie, Francois H van der Westhuizen.   

Abstract

Mitochondrial disorders are frequently encountered inherited diseases characterized by unexplained multisystem involvement with a chronic, intermittent, or progressive nature. The objective of this paper is to describe the profile of patients with mitochondrial disorders in South Africa. Patients with possible mitochondrial disorders were accessed over 10 years. Analyses for respiratory chain and pyruvate dehydrogenase complex enzymes were performed on muscle. A diagnosis of a mitochondrial disorder was accepted only if an enzyme activity was deficient. Sixty-three patients were diagnosed with a mitochondrial disorder, including 40 African, 20 Caucasian, one mixed ancestry, and two Indian patients. The most important findings were the difference between African patients and other ethnicities: respiratory chain enzyme complexes CI+III or CII+III deficiencies were found in 52.5% of African patients, being of statistical significance (p value = 0.0061). They also presented predominantly with myopathy (p value = 0.0018); the male:female ratio was 1:1.2. Twenty-five (62.5%) African patients presented with varying degrees of a myopathy accompanied by a myopathic face, high palate, and scoliosis. Fourteen of these 25 also had ptosis and/or progressive external ophthalmoplegia. No patients of other ethnicities presented with this specific myopathic phenotype. Caucasian patients (16/20) presented predominantly with central nervous system involvement. Of the South African pediatric neurology patients, Africans are more likely to present with myopathy and CII+III deficiency, and Caucasian patients are more likely to present with encephalopathy or encephalomyopathy.

Entities:  

Mesh:

Substances:

Year:  2010        PMID: 20135231     DOI: 10.1007/s10545-009-9031-8

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  21 in total

1.  An evolutionary perspective on pathogenic mtDNA mutations: haplogroup associations of clinical disorders.

Authors:  Corinna Herrnstadt; Neil Howell
Journal:  Mitochondrion       Date:  2004-10-01       Impact factor: 4.160

2.  Multiple presentation of mitochondrial disorders.

Authors:  A Nissenkorn; A Zeharia; D Lev; A Fatal-Valevski; V Barash; A Gutman; S Harel; T Lerman-Sagie
Journal:  Arch Dis Child       Date:  1999-09       Impact factor: 3.791

Review 3.  Clinical presentation of mitochondrial disorders in childhood.

Authors:  A Munnich; A Rötig; D Chretien; V Cormier; T Bourgeron; J P Bonnefont; J M Saudubray; P Rustin
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

4.  Mitochondrial disorders: a proposal for consensus diagnostic criteria in infants and children.

Authors:  Nicole I Wolf; Jan A M Smeitink
Journal:  Neurology       Date:  2002-11-12       Impact factor: 9.910

5.  Amino-acid depletion induced by abnormal amino-acid conjugation and protein restriction in isovaleric acidemia.

Authors:  D T Loots; L J Mienie; E Erasmus
Journal:  Eur J Clin Nutr       Date:  2007-02-07       Impact factor: 4.016

6.  The role of mtDNA background in disease expression: a new primary LHON mutation associated with Western Eurasian haplogroup J.

Authors:  Michael D Brown; Elena Starikovskaya; Olga Derbeneva; Seyed Hosseini; Jon C Allen; Irina E Mikhailovskaya; Rem I Sukernik; Douglas C Wallace
Journal:  Hum Genet       Date:  2002-01-24       Impact factor: 4.132

Review 7.  Mitochondrial disorders: clinical presentation and diagnostic dilemmas.

Authors:  J A M Smeitink
Journal:  J Inherit Metab Dis       Date:  2003       Impact factor: 4.982

8.  Clinical spectrum, morbidity, and mortality in 113 pediatric patients with mitochondrial disease.

Authors:  Fernando Scaglia; Jeffrey A Towbin; William J Craigen; John W Belmont; E O'Brian Smith; Stephen R Neish; Stephanie M Ware; Jill V Hunter; Susan D Fernbach; Georgirene D Vladutiu; Lee-Jun C Wong; Hannes Vogel
Journal:  Pediatrics       Date:  2004-10       Impact factor: 7.124

9.  Urinary excretion of homocitric acid and methylhomocitric acid in propionic acidaemia: minor metabolic products of the citrate synthase aldol condensation reaction.

Authors:  J P van Rooyen; L J Mienie; E Erasmus; W J de Wet; M Duran; S K Wadman
Journal:  Clin Chim Acta       Date:  1994-10-14       Impact factor: 3.786

10.  Leigh syndrome: clinical features and biochemical and DNA abnormalities.

Authors:  S Rahman; R B Blok; H H Dahl; D M Danks; D M Kirby; C W Chow; J Christodoulou; D R Thorburn
Journal:  Ann Neurol       Date:  1996-03       Impact factor: 10.422

View more
  7 in total

Review 1.  Maternally inherited mitochondrial respiratory disorders: from pathogenetic principles to therapeutic implications.

Authors:  Martine Uittenbogaard; Anne Chiaramello
Journal:  Mol Genet Metab       Date:  2020-06-27       Impact factor: 4.797

2.  Knowledge and awareness of mitochondrial diseases among physicians in the tertiary hospitals in Ghana.

Authors:  Eric A Mensah; Bismark Sarfo; Alfred E Yawson; Joshua Arthur; Augustine Ocloo
Journal:  PLoS One       Date:  2022-10-20       Impact factor: 3.752

3.  Toward a mtDNA locus-specific mutation database using the LOVD platform.

Authors:  Joanna L Elson; Mary G Sweeney; Vincent Procaccio; John W Yarham; Antonio Salas; Qing-Peng Kong; Francois H van der Westhuizen; Robert D S Pitceathly; David R Thorburn; Marie T Lott; Douglas C Wallace; Robert W Taylor; Robert McFarland
Journal:  Hum Mutat       Date:  2012-07-02       Impact factor: 4.878

4.  Characterization of mtDNA variation in a cohort of South African paediatric patients with mitochondrial disease.

Authors:  Elizna M van der Walt; Izelle Smuts; Robert W Taylor; Joanna L Elson; Douglass M Turnbull; Roan Louw; Francois H van der Westhuizen
Journal:  Eur J Hum Genet       Date:  2012-01-18       Impact factor: 4.246

5.  Mitochondrial DNA sequence context in the penetrance of mitochondrial t-RNA mutations: A study across multiple lineages with diagnostic implications.

Authors:  Rachel A Queen; Jannetta S Steyn; Phillip Lord; Joanna L Elson
Journal:  PLoS One       Date:  2017-11-21       Impact factor: 3.240

6.  What can a comparative genomics approach tell us about the pathogenicity of mtDNA mutations in human populations?

Authors:  Hannah O'Keefe; Rachel Queen; Phillip Lord; Joanna L Elson
Journal:  Evol Appl       Date:  2019-08-27       Impact factor: 5.183

7.  A broad comparative genomics approach to understanding the pathogenicity of Complex I mutations.

Authors:  Galya V Klink; Hannah O'Keefe; Amrita Gogna; Georgii A Bazykin; Joanna L Elson
Journal:  Sci Rep       Date:  2021-10-01       Impact factor: 4.379

  7 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.