Literature DB >> 25126051

An overview of current mouse models recapitulating coenzyme q10 deficiency syndrome.

Floriana Licitra1, Hélène Puccio1.   

Abstract

Coenzyme Q (CoQ), also known as ubiquinone, is an essential lipophilic molecule present in all cellular membranes and involved in a variety of cellular functions, in particular as an electron carrier in the mitochondrial respiratory chain and as a potent antioxidant. CoQ is synthesized endogenously through a complex metabolic pathway involving over 10 different components. Primary CoQ10 deficiency in humans, due to mutations in genes involved in CoQ biosynthesis, is a heterogeneous group of rare disorders presenting severe and complex clinical symptoms. The generation of mouse models deficient in CoQ is important to further clarify the cellular function of CoQ and to unravel the complexity in the pathophysiological consequences of CoQ deficiency. This review summarizes the current knowledge on mouse models of primary CoQ deficiency.

Entities:  

Keywords:  Cerebellar ataxia; Coenzyme Q; Encephalopathy; Knockout; Mouse models; Nephrotic syndrome

Year:  2014        PMID: 25126051      PMCID: PMC4112528          DOI: 10.1159/000362942

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  36 in total

Review 1.  Metabolism and function of coenzyme Q.

Authors:  Mikael Turunen; Jerker Olsson; Gustav Dallner
Journal:  Biochim Biophys Acta       Date:  2004-01-28

2.  Mouse homologue of coq7/clk-1, longevity gene in Caenorhabditis elegans, is essential for coenzyme Q synthesis, maintenance of mitochondrial integrity, and neurogenesis.

Authors:  D Nakai; S Yuasa; M Takahashi; T Shimizu; S Asaumi; K Isono; T Takao; Y Suzuki; H Kuroyanagi; K Hirokawa; H Koseki; T Shirsawa
Journal:  Biochem Biophys Res Commun       Date:  2001-11-30       Impact factor: 3.575

3.  Glomerular and tubular epithelial defects in kd/kd mice lead to progressive renal failure.

Authors:  Michael P Madaio; Rexford S Ahima; Ray Meade; Daniel J Rader; Alberto Mendoza; Min Peng; John E Tomaszewski; Wayne W Hancock; David L Gasser
Journal:  Am J Nephrol       Date:  2005-11-09       Impact factor: 3.754

4.  Leigh syndrome with nephropathy and CoQ10 deficiency due to decaprenyl diphosphate synthase subunit 2 (PDSS2) mutations.

Authors:  Luis Carlos López; Markus Schuelke; Catarina M Quinzii; Tomotake Kanki; Richard J T Rodenburg; Ali Naini; Salvatore Dimauro; Michio Hirano
Journal:  Am J Hum Genet       Date:  2006-10-27       Impact factor: 11.025

5.  Evolutionary conservation of the clk-1-dependent mechanism of longevity: loss of mclk1 increases cellular fitness and lifespan in mice.

Authors:  Xingxing Liu; Ning Jiang; Bryan Hughes; Eve Bigras; Eric Shoubridge; Siegfried Hekimi
Journal:  Genes Dev       Date:  2005-09-29       Impact factor: 11.361

6.  Prenyldiphosphate synthase, subunit 1 (PDSS1) and OH-benzoate polyprenyltransferase (COQ2) mutations in ubiquinone deficiency and oxidative phosphorylation disorders.

Authors:  Julie Mollet; Irina Giurgea; Dimitri Schlemmer; Gustav Dallner; Dominique Chretien; Agnès Delahodde; Delphine Bacq; Pascale de Lonlay; Arnold Munnich; Agnès Rötig
Journal:  J Clin Invest       Date:  2007-03       Impact factor: 14.808

Review 7.  Endogenous synthesis of coenzyme Q in eukaryotes.

Authors:  UyenPhuong C Tran; Catherine F Clarke
Journal:  Mitochondrion       Date:  2007-03-30       Impact factor: 4.160

8.  COQ2 nephropathy: a newly described inherited mitochondriopathy with primary renal involvement.

Authors:  Francesca Diomedi-Camassei; Silvia Di Giandomenico; Filippo M Santorelli; Gianluca Caridi; Fiorella Piemonte; Giovanni Montini; Gian Marco Ghiggeri; Luisa Murer; Laura Barisoni; Anna Pastore; Andrea Onetti Muda; Maria Luisa Valente; Enrico Bertini; Francesco Emma
Journal:  J Am Soc Nephrol       Date:  2007-09-12       Impact factor: 10.121

9.  Muscle coenzyme Q deficiency in familial mitochondrial encephalomyopathy.

Authors:  S Ogasahara; A G Engel; D Frens; D Mack
Journal:  Proc Natl Acad Sci U S A       Date:  1989-04       Impact factor: 11.205

10.  CABC1 gene mutations cause ubiquinone deficiency with cerebellar ataxia and seizures.

Authors:  Julie Mollet; Agnès Delahodde; Valérie Serre; Dominique Chretien; Dimitri Schlemmer; Anne Lombes; Nathalie Boddaert; Isabelle Desguerre; Pascale de Lonlay; Hélène Ogier de Baulny; Arnold Munnich; Agnès Rötig
Journal:  Am J Hum Genet       Date:  2008-03       Impact factor: 11.025

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  8 in total

1.  Cerebellar Ataxia and Coenzyme Q Deficiency through Loss of Unorthodox Kinase Activity.

Authors:  Jonathan A Stefely; Floriana Licitra; Leila Laredj; Andrew G Reidenbach; Zachary A Kemmerer; Anais Grangeray; Tiphaine Jaeg-Ehret; Catherine E Minogue; Arne Ulbrich; Paul D Hutchins; Emily M Wilkerson; Zheng Ruan; Deniz Aydin; Alexander S Hebert; Xiao Guo; Elyse C Freiberger; Laurence Reutenauer; Adam Jochem; Maya Chergova; Isabel E Johnson; Danielle C Lohman; Matthew J P Rush; Nicholas W Kwiecien; Pankaj K Singh; Anna I Schlagowski; Brendan J Floyd; Ulrika Forsman; Pavel J Sindelar; Michael S Westphall; Fabien Pierrel; Joffrey Zoll; Matteo Dal Peraro; Natarajan Kannan; Craig A Bingman; Joshua J Coon; Philippe Isope; Hélène Puccio; David J Pagliarini
Journal:  Mol Cell       Date:  2016-08-04       Impact factor: 17.970

2.  Targeting a Braf/Mapk pathway rescues podocyte lipid peroxidation in CoQ-deficiency kidney disease.

Authors:  Eriene-Heidi Sidhom; Choah Kim; Maria Kost-Alimova; May Theng Ting; Keith Keller; Julian Avila-Pacheco; Andrew Jb Watts; Katherine A Vernon; Jamie L Marshall; Estefanía Reyes-Bricio; Matthew Racette; Nicolas Wieder; Giulio Kleiner; Elizabeth J Grinkevich; Fei Chen; Astrid Weins; Clary B Clish; Jillian L Shaw; Catarina M Quinzii; Anna Greka
Journal:  J Clin Invest       Date:  2021-03-01       Impact factor: 14.808

3.  Inefficient thermogenic mitochondrial respiration due to futile proton leak in a mouse model of fragile X syndrome.

Authors:  Keren K Griffiths; Aili Wang; Lifei Wang; Matthew Tracey; Giulio Kleiner; Catarina M Quinzii; Linlin Sun; Guang Yang; Jose F Perez-Zoghbi; Pawel Licznerski; Mu Yang; Elizabeth A Jonas; Richard J Levy
Journal:  FASEB J       Date:  2020-04-20       Impact factor: 5.191

4.  New pathogenic variants in COQ4 cause ataxia and neurodevelopmental disorder without detectable CoQ10 deficiency in muscle or skin fibroblasts.

Authors:  Serena Mero; Leonardo Salviati; Vincenzo Leuzzi; Anna Rubegni; Cristina Calderan; Francesca Nardecchia; Daniele Galatolo; Maria Andrea Desbats; Valentina Naef; Federica Gemignani; Maria Novelli; Alessandra Tessa; Roberta Battini; Filippo M Santorelli; Maria Marchese
Journal:  J Neurol       Date:  2021-03-11       Impact factor: 4.849

5.  CLD1 Reverses the Ubiquinone Insufficiency of Mutant cat5/coq7 in a Saccharomyces cerevisiae Model System.

Authors:  Adwitiya Kar; Haley Beam; Megan B Borror; Michael Luckow; Xiaoli Gao; Shane L Rea
Journal:  PLoS One       Date:  2016-09-07       Impact factor: 3.240

6.  Coenzyme Q deficiency causes impairment of the sulfide oxidation pathway.

Authors:  Marcello Ziosi; Ivano Di Meo; Giulio Kleiner; Xing-Huang Gao; Emanuele Barca; Maria J Sanchez-Quintero; Saba Tadesse; Hongfeng Jiang; Changhong Qiao; Richard J Rodenburg; Emmanuel Scalais; Markus Schuelke; Belinda Willard; Maria Hatzoglou; Valeria Tiranti; Catarina M Quinzii
Journal:  EMBO Mol Med       Date:  2017-01       Impact factor: 12.137

Review 7.  Coenzyme Q10 and Immune Function: An Overview.

Authors:  David Mantle; Robert A Heaton; Iain P Hargreaves
Journal:  Antioxidants (Basel)       Date:  2021-05-11

8.  N-Acetyl cysteine ameliorates hyperglycemia-induced cardiomyocyte toxicity by improving mitochondrial energetics and enhancing endogenous Coenzyme Q9/10 levels.

Authors:  Phiwayinkosi V Dludla; Patrick Orlando; Sonia Silvestri; Sithandiwe E Mazibuko-Mbeje; Rabia Johnson; Fabio Marcheggiani; Ilenia Cirilli; Christo J F Muller; Johan Louw; Nnini Obonye; Thembeka Nyawo; Bongani B Nkambule; Luca Tiano
Journal:  Toxicol Rep       Date:  2019-11-05
  8 in total

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