Literature DB >> 25681081

Pathologic Variants of the Mitochondrial Phosphate Carrier SLC25A3: Two New Patients and Expansion of the Cardiomyopathy/Skeletal Myopathy Phenotype With and Without Lactic Acidosis.

E J Bhoj1, M Li, R Ahrens-Nicklas, L C Pyle, J Wang, V W Zhang, C Clarke, L J Wong, N Sondheimer, C Ficicioglu, M Yudkoff.   

Abstract

Variants in the SLC25A3 gene, which codes for the mitochondrial phosphate transporter (PiC), lead to a failure of inorganic phosphate (Pi) transport across the mitochondrial membrane, which is required in the final step of oxidative phosphorylation. The literature described two affected sibships with variants in SLC25A3; all cases had skeletal myopathy and cardiomyopathy (OMIM 610773). We report here two new patients who had neonatal cardiomyopathy; one of whom did not have skeletal myopathy nor elevated lactate. Patient 1 had a homozygous splice site variant, c.158-9A>G, which has been previously reported in a Turkish family. Patient 2 was found to be a compound heterozygote for two novel variants, c.599T>G (p.Leu200Trp) and c. 886_898delGGTAGCAGTGCTTinsCAGATAC (p.Gly296_Ser300delinsGlnIlePro). Protein structure analysis indicated that both variants are likely to be pathogenic. Sequencing of SLC25A3 should be considered in patients with isolated cardiomyopathy, even those without generalized skeletal myopathy or lactic acidosis.

Entities:  

Year:  2015        PMID: 25681081      PMCID: PMC4501241          DOI: 10.1007/8904_2014_364

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


  9 in total

1.  Mitochondrial phosphate-carrier deficiency: a novel disorder of oxidative phosphorylation.

Authors:  Johannes A Mayr; Olaf Merkel; Sepp D Kohlwein; Boris R Gebhardt; Hansjosef Böhles; Ulrike Fötschl; Johannes Koch; Michaela Jaksch; Hanns Lochmüller; Rita Horváth; Peter Freisinger; Wolfgang Sperl
Journal:  Am J Hum Genet       Date:  2007-01-10       Impact factor: 11.025

2.  Expression in Escherichia coli, functional characterization, and tissue distribution of isoforms A and B of the phosphate carrier from bovine mitochondria.

Authors:  G Fiermonte; V Dolce; F Palmieri
Journal:  J Biol Chem       Date:  1998-08-28       Impact factor: 5.157

3.  Screening for nuclear genetic defects in the ATP synthase-associated genes TMEM70, ATP12 and ATP5E in patients with 3-methylglutaconic aciduria.

Authors:  F Tort; M Del Toro; W Lissens; J Montoya; M Fernàndez-Burriel; A Font; N Buján; A Navarro-Sastre; E López-Gallardo; J A Arranz; E Riudor; P Briones; A Ribes
Journal:  Clin Genet       Date:  2011-09       Impact factor: 4.438

4.  Tissue-specific expression of the two isoforms of the mitochondrial phosphate carrier in bovine tissues.

Authors:  V Dolce; G Fiermonte; F Palmieri
Journal:  FEBS Lett       Date:  1996-12-09       Impact factor: 4.124

5.  Deficiency of the mitochondrial phosphate carrier presenting as myopathy and cardiomyopathy in a family with three affected children.

Authors:  Johannes A Mayr; Franz A Zimmermann; Rita Horváth; Hans-Christian Schneider; Benedikt Schoser; Elke Holinski-Feder; Birgit Czermin; Peter Freisinger; Wolfgang Sperl
Journal:  Neuromuscul Disord       Date:  2011-07-16       Impact factor: 4.296

6.  Kinetic mechanism of phosphate/phosphate and phosphate/OH- antiports catalyzed by reconstituted phosphate carrier from beef heart mitochondria.

Authors:  R Stappen; R Krämer
Journal:  J Biol Chem       Date:  1994-04-15       Impact factor: 5.157

7.  Mitochondrial ATP synthase deficiency due to a mutation in the ATP5E gene for the F1 epsilon subunit.

Authors:  Johannes A Mayr; Vendula Havlícková; Franz Zimmermann; Iris Magler; Vilma Kaplanová; Pavel Jesina; Alena Pecinová; Hana Nusková; Johannes Koch; Wolfgang Sperl; Josef Houstek
Journal:  Hum Mol Genet       Date:  2010-06-21       Impact factor: 6.150

8.  TMEM70 mutations cause isolated ATP synthase deficiency and neonatal mitochondrial encephalocardiomyopathy.

Authors:  Alena Cízková; Viktor Stránecký; Johannes A Mayr; Markéta Tesarová; Vendula Havlícková; Jan Paul; Robert Ivánek; Andreas W Kuss; Hana Hansíková; Vilma Kaplanová; Marek Vrbacký; Hana Hartmannová; Lenka Nosková; Tomás Honzík; Zdenek Drahota; Martin Magner; Katerina Hejzlarová; Wolfgang Sperl; Jirí Zeman; Josef Houstek; Stanislav Kmoch
Journal:  Nat Genet       Date:  2008-10-26       Impact factor: 38.330

9.  Genetic dysfunction of MT-ATP6 causes axonal Charcot-Marie-Tooth disease.

Authors:  Robert D S Pitceathly; Sinéad M Murphy; Ellen Cottenie; Annapurna Chalasani; Mary G Sweeney; Cathy Woodward; Ese E Mudanohwo; Iain Hargreaves; Simon Heales; John Land; Janice L Holton; Henry Houlden; Julian Blake; Michael Champion; Frances Flinter; Stephanie A Robb; Rupert Page; Michael Rose; Jacqueline Palace; Carol Crowe; Cheryl Longman; Michael P Lunn; Shamima Rahman; Mary M Reilly; Michael G Hanna
Journal:  Neurology       Date:  2012-08-29       Impact factor: 9.910

  9 in total
  12 in total

Review 1.  Molecular regulation of MCU: Implications in physiology and disease.

Authors:  Neeharika Nemani; Santhanam Shanmughapriya; Muniswamy Madesh
Journal:  Cell Calcium       Date:  2018-06-27       Impact factor: 6.817

Review 2.  The mitochondrial phosphate carrier: Role in oxidative metabolism, calcium handling and mitochondrial disease.

Authors:  Erin L Seifert; Erzsébet Ligeti; Johannes A Mayr; Neal Sondheimer; György Hajnóczky
Journal:  Biochem Biophys Res Commun       Date:  2015-06-16       Impact factor: 3.575

3.  Natural and Induced Mitochondrial Phosphate Carrier Loss: DIFFERENTIAL DEPENDENCE OF MITOCHONDRIAL METABOLISM AND DYNAMICS AND CELL SURVIVAL ON THE EXTENT OF DEPLETION.

Authors:  Erin L Seifert; Aniko Gál; Michelle G Acoba; Qipei Li; Lauren Anderson-Pullinger; Tunde Golenár; Cynthia Moffat; Neal Sondheimer; Steven M Claypool; György Hajnóczky
Journal:  J Biol Chem       Date:  2016-10-25       Impact factor: 5.157

Review 4.  Diseases Caused by Mutations in Mitochondrial Carrier Genes SLC25: A Review.

Authors:  Ferdinando Palmieri; Pasquale Scarcia; Magnus Monné
Journal:  Biomolecules       Date:  2020-04-23

5.  Mitochondrial copper and phosphate transporter specificity was defined early in the evolution of eukaryotes.

Authors:  Xinyu Zhu; Aren Boulet; Katherine M Buckley; Casey B Phillips; Micah G Gammon; Laura E Oldfather; Stanley A Moore; Scot C Leary; Paul A Cobine
Journal:  Elife       Date:  2021-02-16       Impact factor: 8.140

6.  The mitochondrial carrier SFXN1 is critical for complex III integrity and cellular metabolism.

Authors:  Michelle Grace Acoba; Ebru S Selen Alpergin; Santosh Renuse; Lucía Fernández-Del-Río; Ya-Wen Lu; Oleh Khalimonchuk; Catherine F Clarke; Akhilesh Pandey; Michael J Wolfgang; Steven M Claypool
Journal:  Cell Rep       Date:  2021-03-16       Impact factor: 9.423

7.  Association of Genetic Variant at Chromosome 12q23.1 With Neuropathic Pain Susceptibility.

Authors:  Abirami Veluchamy; Harry L Hébert; Natalie R van Zuydam; Ewan R Pearson; Archie Campbell; Caroline Hayward; Weihua Meng; Mark I McCarthy; David L H Bennett; Colin N A Palmer; Blair H Smith
Journal:  JAMA Netw Open       Date:  2021-12-01

8.  PacBio Iso-Seq Improves the Rainbow Trout Genome Annotation and Identifies Alternative Splicing Associated With Economically Important Phenotypes.

Authors:  Ali Ali; Gary H Thorgaard; Mohamed Salem
Journal:  Front Genet       Date:  2021-07-15       Impact factor: 4.599

Review 9.  Importance of Dietary Phosphorus for Bone Metabolism and Healthy Aging.

Authors:  Juan Serna; Clemens Bergwitz
Journal:  Nutrients       Date:  2020-09-30       Impact factor: 5.717

10.  Slc20a1/Pit1 and Slc20a2/Pit2 are essential for normal skeletal myofiber function and survival.

Authors:  Sampada Chande; Daniel Caballero; Bryan B Ho; Jonathan Fetene; Juan Serna; Dominik Pesta; Ali Nasiri; Michael Jurczak; Nicholas W Chavkin; Nati Hernando; Cecilia M Giachelli; Carsten A Wagner; Caroline Zeiss; Gerald I Shulman; Clemens Bergwitz
Journal:  Sci Rep       Date:  2020-02-20       Impact factor: 4.379

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