Literature DB >> 23430795

NDUFS8-related Complex I Deficiency Extends Phenotype from "PEO Plus" to Leigh Syndrome.

Adela Della Marina1, Ulrike Schara, Angela Pyle, Claudia Möller-Hartmann, Elke Holinski-Feder, Angela Abicht, Birgit Czermin, Hanns Lochmüller, Helen Griffin, Mauro Santibanez-Koref, Patrick F Chinnery, Rita Horvath.   

Abstract

With over 1,000 nuclear genes that could potentially cause a mitochondrial disorder, the current diagnostic approach requires targeted molecular analysis, guided by a combination of clinical and biochemical features. However, the expanding molecular and clinical spectrum means that this approach does not always yield a result. Here we report the unusual clinical presentation of "Progressive External Ophthalmoplegia (PEO) plus" Leigh syndrome in three children from a consanguineous family where exome sequencing identified mutations in NDUFS8. NDUFS8 is a nuclear-encoded structural core protein of complex I, and mutations are expected to cause infantile onset and severe disease. Our patients had a later onset, milder and a clinically distinct phenotype, and this gene would not normally be considered in this context. Being untargeted to specific genes, whole exome analysis has the potential to re-write the phenotype and reveal an unexpected molecular aetiology, as illustrated by this family.

Entities:  

Year:  2012        PMID: 23430795      PMCID: PMC3755572          DOI: 10.1007/8904_2012_195

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  12 in total

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Authors:  Hélène Pagniez-Mammeri; Sandrine Loublier; Alain Legrand; Paule Bénit; Pierre Rustin; Abdelhamid Slama
Journal:  Mol Genet Metab       Date:  2011-11-18       Impact factor: 4.797

2.  SURF1 missense mutations promote a mild Leigh phenotype.

Authors:  D Piekutowska-Abramczuk; M Magner; E Popowska; M Pronicki; E Karczmarewicz; J Sykut-Cegielska; T Kmiec; E Jurkiewicz; T Szymanska-Debinska; L Bielecka; M Krajewska-Walasek; K Vesela; J Zeman; E Pronicka
Journal:  Clin Genet       Date:  2009-08       Impact factor: 4.438

Review 3.  Assembly factors as a new class of disease genes for mitochondrial complex I deficiency: cause, pathology and treatment options.

Authors:  Jessica Nouws; Leo G J Nijtmans; Jan A Smeitink; Rutger O Vogel
Journal:  Brain       Date:  2011-10-27       Impact factor: 13.501

4.  NDUFA4 is a subunit of complex IV of the mammalian electron transport chain.

Authors:  Eduardo Balsa; Ricardo Marco; Ester Perales-Clemente; Radek Szklarczyk; Enrique Calvo; Manuel O Landázuri; José Antonio Enríquez
Journal:  Cell Metab       Date:  2012-08-16       Impact factor: 27.287

5.  High-throughput, pooled sequencing identifies mutations in NUBPL and FOXRED1 in human complex I deficiency.

Authors:  Sarah E Calvo; Elena J Tucker; Alison G Compton; Denise M Kirby; Gabriel Crawford; Noel P Burtt; Manuel Rivas; Candace Guiducci; Damien L Bruno; Olga A Goldberger; Michelle C Redman; Esko Wiltshire; Callum J Wilson; David Altshuler; Stacey B Gabriel; Mark J Daly; David R Thorburn; Vamsi K Mootha
Journal:  Nat Genet       Date:  2010-09-05       Impact factor: 38.330

6.  The p.M292T NDUFS2 mutation causes complex I-deficient Leigh syndrome in multiple families.

Authors:  Helen A L Tuppen; Vanessa E Hogan; Langping He; Emma L Blakely; Lisa Worgan; Mazhor Al-Dosary; Gabriele Saretzki; Charlotte L Alston; Andrew A Morris; Michael Clarke; Simon Jones; Anita M Devlin; Sahar Mansour; Zofia M A Chrzanowska-Lightowlers; David R Thorburn; Robert McFarland; Robert W Taylor
Journal:  Brain       Date:  2010-09-06       Impact factor: 13.501

Review 7.  Late-onset Leigh syndrome in a patient with mitochondrial complex I NDUFS8 mutations.

Authors:  Vincent Procaccio; Douglas C Wallace
Journal:  Neurology       Date:  2004-05-25       Impact factor: 9.910

Review 8.  Mitochondrial complex I deficiency: from organelle dysfunction to clinical disease.

Authors:  Felix Distelmaier; Werner J H Koopman; Lambertus P van den Heuvel; Richard J Rodenburg; Ertan Mayatepek; Peter H G M Willems; Jan A M Smeitink
Journal:  Brain       Date:  2009-03-31       Impact factor: 13.501

9.  Genomic structure of the human NDUFS8 gene coding for the iron-sulfur TYKY subunit of the mitochondrial NADH:ubiquinone oxidoreductase.

Authors:  R de Sury; P Martinez; V Procaccio; J Lunardi; J P Issartel
Journal:  Gene       Date:  1998-07-17       Impact factor: 3.688

10.  A new phenotype of brain iron accumulation with dystonia, optic atrophy, and peripheral neuropathy.

Authors:  Rita Horvath; Elke Holinski-Feder; Vivienne C M Neeve; Angela Pyle; Helen Griffin; Deephthi Ashok; Charlotte Foley; Gavin Hudson; Bernd Rautenstrauss; Gudrun Nürnberg; Peter Nürnberg; Jörg Kortler; Birgit Neitzel; Ingelore Bässmann; Thahira Rahman; Bernard Keavney; John Loughlin; Sophie Hambleton; Benedikt Schoser; Hanns Lochmüller; Mauro Santibanez-Koref; Patrick F Chinnery
Journal:  Mov Disord       Date:  2012-04-16       Impact factor: 10.338

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  7 in total

1.  Mito-Nuclear Interactions Affecting Lifespan and Neurodegeneration in a Drosophila Model of Leigh Syndrome.

Authors:  Carin A Loewen; Barry Ganetzky
Journal:  Genetics       Date:  2018-03-01       Impact factor: 4.562

Review 2.  Next-generation sequencing for mitochondrial disorders.

Authors:  C J Carroll; V Brilhante; A Suomalainen
Journal:  Br J Pharmacol       Date:  2014-04       Impact factor: 8.739

3.  Accurate mitochondrial DNA sequencing using off-target reads provides a single test to identify pathogenic point mutations.

Authors:  Helen R Griffin; Angela Pyle; Emma L Blakely; Charlotte L Alston; Jennifer Duff; Gavin Hudson; Rita Horvath; Ian J Wilson; Mauro Santibanez-Koref; Robert W Taylor; Patrick F Chinnery
Journal:  Genet Med       Date:  2014-06-05       Impact factor: 8.822

Review 4.  The genetics of Leigh syndrome and its implications for clinical practice and risk management.

Authors:  Ilene S Ruhoy; Russell P Saneto
Journal:  Appl Clin Genet       Date:  2014-11-13

5.  Heroin Abuse Results in Shifted RNA Expression to Neurodegenerative Diseases and Attenuation of TNFα Signaling Pathway.

Authors:  Mei Zhu; Yu Xu; Huawei Wang; Zongwen Shen; Zhenrong Xie; Fengrong Chen; Yunhong Gao; Xin Chen; Ying Zhang; Qiang Wu; Xuejun Li; Juehua Yu; Huayou Luo; Kunhua Wang
Journal:  Sci Rep       Date:  2018-06-18       Impact factor: 4.379

6.  Expansion of the clinical and neuroimaging spectrum associated with NDUFS8-related disorder.

Authors:  Milena M Andzelm; Shanti Balasubramaniam; Edward Yang; Alison G Compton; Kate Millington; Jia Zhu; Irina Anselm; Lance H Rodan; David R Thorburn; John Christodoulou; Siddharth Srivastava
Journal:  JIMD Rep       Date:  2022-08-23

Review 7.  Blackout in the powerhouse: clinical phenotypes associated with defects in the assembly of OXPHOS complexes and the mitoribosome.

Authors:  Daniella H Hock; David R L Robinson; David A Stroud
Journal:  Biochem J       Date:  2020-11-13       Impact factor: 3.857

  7 in total

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