Literature DB >> 10604140

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

D P O'Brien1, B A Barshop, K K Faunt, G C Johnson, K M Gibson, G D Shelton.   

Abstract

A family of Maltese dogs with malonic aciduria is reported. The propositus presented at 3 years of age with episodes of seizures and stupor with hypoglycaemia, acidosis, and ketonuria. Urinary organic acid assays showed elevated malonic acid without elevation of methylmalonic acid. Cultured fibroblasts had normal malonyl-CoA decarboxylase activity. Treatment with frequent feedings of a low-fat diet high in medium-chain triglycerides resulted in normalization of clinical signs and a resolution of the malonic aciduria. Two full siblings of the propositus had died at a young age of undiagnosed metabolic and neurological disease. Urine organic acid assays were performed on other family members. A half-sister showed mild malonic aciduria and other organic acid changes similar to the propositus, while the mother and half-brother showed mildly elevated ketone bodies. This family suggests further genetic and clinical heterogeneity in the malonic acidurias.

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Year:  1999        PMID: 10604140     DOI: 10.1023/a:1005635306257

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


  13 in total

1.  Cloning and mutational analysis of human malonyl-coenzyme A decarboxylase.

Authors:  J Gao; L Waber; M J Bennett; K M Gibson; J C Cohen
Journal:  J Lipid Res       Date:  1999-01       Impact factor: 5.922

2.  Fatal neonatal malonic aciduria.

Authors:  B Buyukgebiz; C Jakobs; H R Scholte; J G Huijmans; W J Kleijer
Journal:  J Inherit Metab Dis       Date:  1998-02       Impact factor: 4.982

Review 3.  Regulation of hepatic fatty acid oxidation and ketone body production.

Authors:  J D McGarry; D W Foster
Journal:  Annu Rev Biochem       Date:  1980       Impact factor: 23.643

4.  Malonic aciduria and cardiomyopathy.

Authors:  R Matalon; K Michaels; R Kaul; V Whitman; J Rodriguez-Novo; S Goodman; D Thorburn
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

5.  Methylmalonic and malonic aciduria in a dog with progressive encephalomyelopathy.

Authors:  M Podell; G D Shelton; W L Nyhan; S O Wagner; A Genders; M Oglesbee; W R Fenner
Journal:  Metab Brain Dis       Date:  1996-09       Impact factor: 3.584

6.  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

7.  Malonyl coenzyme A decarboxylase deficiency.

Authors:  G K Brown; R D Scholem; A Bankier; D M Danks
Journal:  J Inherit Metab Dis       Date:  1984       Impact factor: 4.982

8.  Malonyl coenzyme A decarboxylase deficiency.

Authors:  G B MacPhee; R W Logan; J S Mitchell; D W Howells; E Tsotsis; D R Thorburn
Journal:  Arch Dis Child       Date:  1993-10       Impact factor: 3.791

9.  Malonyl coenzyme A decarboxylase deficiency. Clinical and biochemical findings in a second child with a more severe enzyme defect.

Authors:  E A Haan; R D Scholem; H B Croll; G K Brown
Journal:  Eur J Pediatr       Date:  1986-04       Impact factor: 3.183

10.  Malonic aciduria.

Authors:  P T Ozand; W L Nyhan; A al Aqeel; J Christodoulou
Journal:  Brain Dev       Date:  1994-11       Impact factor: 1.961

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  3 in total

1.  Clinical, enzymatic and molecular characterization of nine new patients with malonyl-coenzyme A decarboxylase deficiency.

Authors:  G S Salomons; C Jakobs; L Landegge Pope; A Errami; M Potter; M Nowaczyk; S Olpin; N Manning; J A J Raiman; T Slade; M P Champion; D Peck; D Gavrilov; R Hillman; G E Hoganson; K Donaldson; J P H Shield; D Ketteridge; M Wasserstein; K M Gibson
Journal:  J Inherit Metab Dis       Date:  2006-12-20       Impact factor: 4.982

2.  Metabolic Encephalopathy and Lipid Storage Myopathy Associated with a Presumptive Mitochondrial Fatty Acid Oxidation Defect in a Dog.

Authors:  Vanessa R Biegen; John P McCue; Taryn A Donovan; G Diane Shelton
Journal:  Front Vet Sci       Date:  2015-11-26

3.  The effect of acupuncture on depression and its correlation with metabolic alterations: A randomized controlled trial.

Authors:  Wei Li; Manqin Sun; Xuan Yin; Lixing Lao; Zaoyuan Kuang; Shifen Xu
Journal:  Medicine (Baltimore)       Date:  2020-10-23       Impact factor: 1.817

  3 in total

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