Literature DB >> 10102904

Defect in dimethylglycine dehydrogenase, a new inborn error of metabolism: NMR spectroscopy study.

S H Moolenaar1, J Poggi-Bach, U F Engelke, J M Corstiaensen, A Heerschap, J G de Jong, B A Binzak, J Vockley, R A Wevers.   

Abstract

BACKGROUND: A38-year-old man presented with a history of fish odor (since age 5) and unusual muscle fatigue with increased serum creatine kinase. Our aim was to identify the metabolic error in this new condition.
METHODS: We used 1H NMR spectroscopy to study serum and urine from the patient.
RESULTS: The concentration of N, N-dimethylglycine (DMG) was increased approximately 100-fold in the serum and approximately 20-fold in the urine. The presence of DMG as a storage product was confirmed by use of 13C NMR spectroscopy and gas chromatography-mass spectrometry. The high concentration of DMG was caused by a deficiency of the enzyme dimethylglycine dehydrogenase (DMGDH). A homozygous missense mutation was found in the DMGDH gene of the patient.
CONCLUSIONS: DMGDH deficiency must be added to the differential diagnosis of patients complaining of a fish odor. This deficiency is the first inborn error of metabolism discovered by use of in vitro 1H NMR spectroscopy of body fluids. Copyright 1999 American Association for Clinical Chemistry

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Year:  1999        PMID: 10102904

Source DB:  PubMed          Journal:  Clin Chem        ISSN: 0009-9147            Impact factor:   8.327


  15 in total

1.  A method for comprehensive analysis of urinary acylglycines by using ultra-performance liquid chromatography quadrupole linear ion trap mass spectrometry.

Authors:  Avalyn E Lewis-Stanislaus; Liang Li
Journal:  J Am Soc Mass Spectrom       Date:  2010-09-18       Impact factor: 3.109

2.  Riboflavin-responsive trimethylaminuria in a patient with homocystinuria on betaine therapy.

Authors:  Nigel J Manning; Elizabeth K Allen; Richard J Kirk; Mark J Sharrard; Edwin J Smith
Journal:  JIMD Rep       Date:  2011-11-20

3.  Cloning of dimethylglycine dehydrogenase and a new human inborn error of metabolism, dimethylglycine dehydrogenase deficiency.

Authors:  B A Binzak; R A Wevers; S H Moolenaar; Y M Lee; W L Hwu; J Poggi-Bach; U F Engelke; H M Hoard; J G Vockley; J Vockley
Journal:  Am J Hum Genet       Date:  2001-02-28       Impact factor: 11.025

Review 4.  MR spectroscopy: a powerful tool for investigating brain function and neurological diseases.

Authors:  A P Burlina; T Aureli; F Bracco; F Conti; L Battistin
Journal:  Neurochem Res       Date:  2000-10       Impact factor: 3.996

5.  Hyperhomocysteinemia and cardiovascular disease: The nutritional perspectives.

Authors:  R Pandey; S Gupta; H Lal; H C Mehta; S K Aggarwal
Journal:  Indian J Clin Biochem       Date:  2000-08

6.  Molecular basis of dimethylglycine dehydrogenase deficiency associated with pathogenic variant H109R.

Authors:  R P McAndrew; J Vockley; J-J P Kim
Journal:  J Inherit Metab Dis       Date:  2008-10-21       Impact factor: 4.982

7.  Class selection of amino acid metabolites in body fluids using chemical derivatization and their enhanced 13C NMR.

Authors:  Narasimhamurthy Shanaiah; M Aruni Desilva; G A Nagana Gowda; Michael A Raftery; Bryan E Hainline; Daniel Raftery
Journal:  Proc Natl Acad Sci U S A       Date:  2007-07-02       Impact factor: 11.205

8.  Whole-exome sequencing identifies homozygous AFG3L2 mutations in a spastic ataxia-neuropathy syndrome linked to mitochondrial m-AAA proteases.

Authors:  Tyler Mark Pierson; David Adams; Florian Bonn; Paola Martinelli; Praveen F Cherukuri; Jamie K Teer; Nancy F Hansen; Pedro Cruz; James C Mullikin For The Nisc Comparative Sequencing Program; Robert W Blakesley; Gretchen Golas; Justin Kwan; Anthony Sandler; Karin Fuentes Fajardo; Thomas Markello; Cynthia Tifft; Craig Blackstone; Elena I Rugarli; Thomas Langer; William A Gahl; Camilo Toro
Journal:  PLoS Genet       Date:  2011-10-13       Impact factor: 5.917

9.  Dimethylglycine Deficiency and the Development of Diabetes.

Authors:  Martin Magnusson; Thomas J Wang; Clary Clish; Gunnar Engström; Peter Nilsson; Robert E Gerszten; Olle Melander
Journal:  Diabetes       Date:  2015-03-20       Impact factor: 9.461

Review 10.  The human flavoproteome.

Authors:  Wolf-Dieter Lienhart; Venugopal Gudipati; Peter Macheroux
Journal:  Arch Biochem Biophys       Date:  2013-03-15       Impact factor: 4.013

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