Literature DB >> 8967754

Deficiency of complex II of the mitochondrial respiratory chain in late-onset optic atrophy and ataxia.

R W Taylor1, M A Birch-Machin, J Schaefer, L Taylor, R Shakir, B A Ackrell, B Cochran, L A Bindoff, M J Jackson, P Griffiths, D M Turnbull.   

Abstract

Defects of the mitochondrial respiratory chain are increasingly being recognized as an important cause of neurological disease in humans. In many of these patients, the biochemical defect results from an abnormality of the mitochondrial genome. Respiratory chain defects involving complex II, which is entirely encoded by the nuclear genome, are comparatively rare. We report the clinical and biochemical findings in 2 elderly sisters who presented with late-onset neurodegenerative disease. In both patients, a partial deficiency of complex II (approximately 50% of control values) was shown to be present in mitochondria from muscle and platelets. The enzyme defect was not expressed in cultured skin fibroblasts or immortalized lymphocytes. There was an overexpression of the 70-kd flavoprotein subunit in muscle mitochondria from both patients, although we showed that this subunit is present in normal amounts in mitochondrial membranes. Our studies highlight the diversity of the clinical presentation of respiratory chain disease and that complex II deficiency should enter the differential diagnosis of certain patients with late-onset neurodegenerative disease.

Entities:  

Mesh:

Substances:

Year:  1996        PMID: 8967754     DOI: 10.1002/ana.410390212

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  14 in total

Review 1.  Clinical mitochondrial genetics.

Authors:  P F Chinnery; N Howell; R M Andrews; D M Turnbull
Journal:  J Med Genet       Date:  1999-06       Impact factor: 6.318

2.  Succinate dehydrogenase deficiency associated with dilated cardiomyopathy and ventricular noncompaction.

Authors:  Zurab Davili; Sandeep Johar; Colleen Hughes; Daniel Kveselis; Joe Hoo
Journal:  Eur J Pediatr       Date:  2006-11-03       Impact factor: 3.183

3.  Leigh syndrome caused by mutations in the flavoprotein (Fp) subunit of succinate dehydrogenase (SDHA).

Authors:  R Horváth; A Abicht; E Holinski-Feder; A Laner; K Gempel; H Prokisch; H Lochmüller; T Klopstock; M Jaksch
Journal:  J Neurol Neurosurg Psychiatry       Date:  2006-01       Impact factor: 10.154

4.  SDHA mutations causing a multisystem mitochondrial disease: novel mutations and genetic overlap with hereditary tumors.

Authors:  G Herma Renkema; Saskia B Wortmann; Roel J Smeets; Hanka Venselaar; Marion Antoine; Gepke Visser; Tawfeg Ben-Omran; Lambert P van den Heuvel; Henri J L M Timmers; Jan A Smeitink; Richard J T Rodenburg
Journal:  Eur J Hum Genet       Date:  2014-04-30       Impact factor: 4.246

5.  Familial neonatal isolated cardiomyopathy caused by a mutation in the flavoprotein subunit of succinate dehydrogenase.

Authors:  Aviva Levitas; Emad Muhammad; Gali Harel; Ann Saada; Vered Chalifa Caspi; Esther Manor; John C Beck; Val Sheffield; Ruti Parvari
Journal:  Eur J Hum Genet       Date:  2010-06-16       Impact factor: 4.246

Review 6.  Biochemical diagnosis of mitochondrial disorders.

Authors:  Richard J T Rodenburg
Journal:  J Inherit Metab Dis       Date:  2010-05-04       Impact factor: 4.982

7.  Imaging mass cytometry reveals generalised deficiency in OXPHOS complexes in Parkinson's disease.

Authors:  Chun Chen; David McDonald; Alasdair Blain; Ashwin Sachdeva; Laura Bone; Anna L M Smith; Charlotte Warren; Sarah J Pickett; Gavin Hudson; Andrew Filby; Amy E Vincent; Doug M Turnbull; Amy K Reeve
Journal:  NPJ Parkinsons Dis       Date:  2021-05-12

8.  Progressive cerebellar atrophy in a patient with complex II and III deficiency and a novel deleterious variant in SDHA: A Counseling Conundrum.

Authors:  Beattie R H Sturrock; Ellen F Macnamara; Peter McGuire; Shannon Kruk; Ivan Yang; Jennifer Murphy; Cyndi J Tifft; Eliza Gordon-Lipkin
Journal:  Mol Genet Genomic Med       Date:  2021-05-07       Impact factor: 2.183

9.  Leukoencephalopathy with accumulated succinate is indicative of SDHAF1 related complex II deficiency.

Authors:  Andreas Ohlenbusch; Simon Edvardson; Johannes Skorpen; Alf Bjornstad; Ann Saada; Orly Elpeleg; Jutta Gärtner; Knut Brockmann
Journal:  Orphanet J Rare Dis       Date:  2012-09-20       Impact factor: 4.123

10.  Ocular findings in mitochondrial neurogastrointestinal encephalomyopathy: a case report.

Authors:  Piero Barboni; Giacomo Savini; Giuseppe Plazzi; Marzio Bellan; Maria Lucia Valentino; Maurizio Zanini; Pasquale Montagna; Michio Hirano; Valerio Carelli
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2004-03-24       Impact factor: 3.117

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.