Literature DB >> 9177981

A new case of malonyl coenzyme A decarboxylase deficiency presenting with cardiomyopathy.

S Yano1, L Sweetman, D R Thorburn, S Mofidi, J C Williams.   

Abstract

UNLABELLED: A new case of mitochondrial malonyl coenzyme A decarboxylase deficiency is described. The patient presented with an initial episode of metabolic acidosis, seizures, hypoglycemia, and cardiac failure at 2 months of age which slowly resolved. Subsequent evaluations at 4 years of age for developmental delay revealed a prominent elevation of malonic acid in urine. Malonyl carnitine was also elevated. The activity of Malonyl CoA decarboxylase in cultured fibroblasts was 7% of normal.
CONCLUSION: Malonyl CoA decarboxylase deficiency may result in inhibition of fatty acid oxidation, which may account for the cardiomyopathy.

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Year:  1997        PMID: 9177981     DOI: 10.1007/s004310050619

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  15 in total

1.  Cloning and expression of rat pancreatic beta-cell malonyl-CoA decarboxylase.

Authors:  N Voilley; R Roduit; R Vicaretti; C Bonny; G Waeber; J R Dyck; G D Lopaschuk; M Prentki
Journal:  Biochem J       Date:  1999-05-15       Impact factor: 3.857

2.  Crystallization and preliminary X-ray diffraction analysis of ybfF, a new esterase from Escherichia coli K12.

Authors:  Suk Youl Park; Sang Hak Lee; Jieun Lee; Che Hun Jung; Jeong Sun Kim
Journal:  Acta Crystallogr Sect F Struct Biol Cryst Commun       Date:  2007-11-30

3.  Identification and Quantitation of Malonic Acid Biomarkers of In-Born Error Metabolism by Targeted Metabolomics.

Authors:  Chandra Shekar R Ambati; Furong Yuan; Lutfi A Abu-Elheiga; Yiqing Zhang; Vivekananda Shetty
Journal:  J Am Soc Mass Spectrom       Date:  2017-03-17       Impact factor: 3.109

4.  Use of a long-chain triglyceride-restricted/medium-chain triglyceride-supplemented diet in a case of malonyl-CoA decarboxylase deficiency with cardiomyopathy.

Authors:  E J Footitt; J Stafford; M Dixon; M Burch; C Jakobs; G S Salomons; M A Cleary
Journal:  J Inherit Metab Dis       Date:  2010-06-15       Impact factor: 4.982

Review 5.  Metabolic cardiomyopathies.

Authors:  B Guertl; C Noehammer; G Hoefler
Journal:  Int J Exp Pathol       Date:  2000-12       Impact factor: 1.925

6.  The molecular basis of malonyl-CoA decarboxylase deficiency.

Authors:  D R FitzPatrick; A Hill; J L Tolmie; D R Thorburn; J Christodoulou
Journal:  Am J Hum Genet       Date:  1999-08       Impact factor: 11.025

Review 7.  Malonyl-CoA decarboxylase is a major regulator of myocardial fatty acid oxidation.

Authors:  Karalyn D Cuthbert; Jason R B Dyck
Journal:  Curr Hypertens Rep       Date:  2005-12       Impact factor: 5.369

8.  Malonic aciduria in Maltese dogs: normal methylmalonic acid concentrations and malonyl-CoA decarboxylase activity in fibroblasts.

Authors:  D P O'Brien; B A Barshop; K K Faunt; G C Johnson; K M Gibson; G D Shelton
Journal:  J Inherit Metab Dis       Date:  1999-12       Impact factor: 4.982

9.  Cardiomyopathy and hypotonia in a 5-month-old infant with malonyl-coa decarboxylase deficiency: potential for preclinical diagnosis with expanded newborn screening.

Authors:  C Ficicioglu; M R K Chrisant; I Payan; D H Chace
Journal:  Pediatr Cardiol       Date:  2005 Nov-Dec       Impact factor: 1.655

10.  Combined malonic and methylmalonic aciduria with normal malonyl-coenzyme A decarboxylase activity: a case supporting multiple aetiologies.

Authors:  A R Gregg; A W Warman; D R Thorburn; W E O'Brien
Journal:  J Inherit Metab Dis       Date:  1998-06       Impact factor: 4.982

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