Literature DB >> 14605509

Silent and symptomatic primary carnitine deficiency within the same family due to identical mutations in the organic cation/carnitine transporter OCTN2.

U Spiekerkoetter1, G Huener, T Baykal, M Demirkol, M Duran, R Wanders, J Nezu, E Mayatepek.   

Abstract

A family of Turkish origin with primary systemic carnitine deficiency in the father and his two sons is described. In all three individuals, the same homozygous mutation in the OCTN2 gene (R471H) was present and carnitine uptake in fibroblasts was deficient. Whereas one boy became symptomatic with a Reye-syndrome-like picture of hepatopathy and encephalopathy in infancy, the other affected family members remained asymptomatic up to their current ages of 28 and 5 years, respectively.

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Year:  2003        PMID: 14605509     DOI: 10.1023/a:1025968502527

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


  6 in total

1.  Genetic epidemiology of the carnitine transporter OCTN2 gene in a Japanese population and phenotypic characterization in Japanese pedigrees with primary systemic carnitine deficiency.

Authors:  A Koizumi; J Nozaki; T Ohura; T Kayo; Y Wada; J Nezu; R Ohashi; I Tamai; Y Shoji; G Takada; S Kibira; T Matsuishi; A Tsuji
Journal:  Hum Mol Genet       Date:  1999-11       Impact factor: 6.150

2.  Selectivity of the polyspecific cation transporter rOCT1 is changed by mutation of aspartate 475 to glutamate.

Authors:  V Gorboulev; C Volk; P Arndt; A Akhoundova; H Koepsell
Journal:  Mol Pharmacol       Date:  1999-12       Impact factor: 4.436

3.  Functional analysis of mutations in the OCTN2 transporter causing primary carnitine deficiency: lack of genotype-phenotype correlation.

Authors:  Y Wang; F Taroni; B Garavaglia; N Longo
Journal:  Hum Mutat       Date:  2000-11       Impact factor: 4.878

4.  Arginine 454 and lysine 370 are essential for the anion specificity of the organic anion transporter, rOAT3.

Authors:  B Feng; M J Dresser; Y Shu; S J Johns; K M Giacomini
Journal:  Biochemistry       Date:  2001-05-08       Impact factor: 3.162

5.  Novel OCTN2 mutations: no genotype-phenotype correlations: early carnitine therapy prevents cardiomyopathy.

Authors:  Anne-Marie Lamhonwah; Simon E Olpin; Rodney J Pollitt; Christine Vianey-Saban; Priscille Divry; Nathalie Guffon; Guy T N Besley; Russell Onizuka; Linda J De Meirleir; Ljerka Cvitanovic-Sojat; Ivo Baric; Carlo Dionisi-Vici; Ksenija Fumic; Miljenka Maradin; Ingrid Tein
Journal:  Am J Med Genet       Date:  2002-08-15

6.  Primary systemic carnitine deficiency is caused by mutations in a gene encoding sodium ion-dependent carnitine transporter.

Authors:  J Nezu; I Tamai; A Oku; R Ohashi; H Yabuuchi; N Hashimoto; H Nikaido; Y Sai; A Koizumi; Y Shoji; G Takada; T Matsuishi; M Yoshino; H Kato; T Ohura; G Tsujimoto; J Hayakawa; M Shimane; A Tsuji
Journal:  Nat Genet       Date:  1999-01       Impact factor: 38.330

  6 in total
  24 in total

1.  Spontaneous development of intestinal and colonic atrophy and inflammation in the carnitine-deficient jvs (OCTN2(-/-)) mice.

Authors:  Prem S Shekhawat; Sonne R Srinivas; Dietrich Matern; Michael J Bennett; Richard Boriack; Varghese George; Hongyan Xu; Puttur D Prasad; Penny Roon; Vadivel Ganapathy
Journal:  Mol Genet Metab       Date:  2007-09-19       Impact factor: 4.797

2.  Lethal Undiagnosed Very Long-Chain Acyl-CoA Dehydrogenase Deficiency with Mild C14-Acylcarnitine Abnormalities on Newborn Screening.

Authors:  U Spiekerkoetter; M Mueller; M Sturm; M Hofmann; D T Schneider
Journal:  JIMD Rep       Date:  2012-02-26

3.  Primary Carnitine Deficiency Presents Atypically with Long QT Syndrome: A Case Report.

Authors:  Irene De Biase; Neena Lorenzana Champaigne; Richard Schroer; Laura Malinda Pollard; Nicola Longo; Tim Wood
Journal:  JIMD Rep       Date:  2011-09-06

4.  Identification of mutations and evaluation of cardiomyopathy in Turkish patients with primary carnitine deficiency.

Authors:  M Kilic; R K Ozgül; T Coşkun; D Yücel; M Karaca; H S Sivri; A Tokatli; M Sahin; T Karagöz; A Dursun
Journal:  JIMD Rep       Date:  2011-09-22

5.  Enzymes involved in L-carnitine biosynthesis are expressed by small intestinal enterocytes in mice: implications for gut health.

Authors:  Prem S Shekhawat; Srinivas Sonne; A Lee Carter; Dietrich Matern; Vadivel Ganapathy
Journal:  J Crohns Colitis       Date:  2012-09-21       Impact factor: 9.071

Review 6.  Mitochondrial fatty acid oxidation disorders: clinical presentation of long-chain fatty acid oxidation defects before and after newborn screening.

Authors:  Ute Spiekerkoetter
Journal:  J Inherit Metab Dis       Date:  2010-05-07       Impact factor: 4.982

7.  Functional and molecular studies in primary carnitine deficiency.

Authors:  Marta Frigeni; Bijina Balakrishnan; Xue Yin; Fernanda R O Calderon; Rong Mao; Marzia Pasquali; Nicola Longo
Journal:  Hum Mutat       Date:  2017-09-14       Impact factor: 4.878

Review 8.  Inborn errors of energy metabolism associated with myopathies.

Authors:  Anibh M Das; Ulrike Steuerwald; Sabine Illsinger
Journal:  J Biomed Biotechnol       Date:  2010-05-26

9.  Role of carnitine in disease.

Authors:  Judith L Flanagan; Peter A Simmons; Joseph Vehige; Mark Dp Willcox; Qian Garrett
Journal:  Nutr Metab (Lond)       Date:  2010-04-16       Impact factor: 4.169

10.  Primary Carnitine deficiency in the Faroe Islands: health and cardiac status in 76 adult patients diagnosed by screening.

Authors:  Jan Rasmussen; Lars Køber; Allan M Lund; Olav W Nielsen
Journal:  J Inherit Metab Dis       Date:  2013-08-21       Impact factor: 4.982

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