Literature DB >> 1245071

Glutaric aciduria type II: report on a previously undescribed metabolic disorder.

H Przyrembel, U Wendel, K Becker, H J Bremer, L Bruinvis, D Ketting, S K Wadman.   

Abstract

A report is given on a hitherto undescribed metabolic disorder, characterized clinically by fatal neonatal acidosis, hypoglycemia and a strong 'sweaty-feet' odour. Biochemical features were a massive urinary excretion of glutaric and lactic acids. Isobutyric, isovaleric and alpha-methylbutyric acids were also greatly increased, followed by adipic, ethylmalonic, alpha-hydroxybutyric, n-butyric, beta-hydroxybutyric, sebacic, suberic, propionic, alpha-hydroxyisovaleric and hexanoic acids. The serum level of glutaric acid was highly elevated. In the serum there were also abnormal levels of lactic, alpha-hydroxybutyric, adipic, suberic, p-hydroxyphenyllactic, myristic, hexadecenoic, palmitic, oleic and stearic acids. Plasma lysine and valine were also elevated. Degradation of 14C-labelled glutaric acid and 14C-labelled branched-chain amino acids, alpha-ketoisovaleric and alpha-ketoisocaproic acids in intact fibroblasts was decreased, whereas that of pyruvic acid was normal. The defect was tentatively supposed to be localized at the level of the metabolism of a range of acyl-CoA compounds. The name glutaric aciduria 'type II' is proposed for the patient's disease.

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Year:  1976        PMID: 1245071     DOI: 10.1016/0009-8981(76)90060-7

Source DB:  PubMed          Journal:  Clin Chim Acta        ISSN: 0009-8981            Impact factor:   3.786


  58 in total

1.  Glutaric aciduria in two brothers.

Authors:  N J Brandt; N Gregersen; E Christensen; K Rasmussen
Journal:  J Inherit Metab Dis       Date:  1978       Impact factor: 4.982

2.  Purification and characterization of Put1p from Saccharomyces cerevisiae.

Authors:  Srimevan Wanduragala; Nikhilesh Sanyal; Xinwen Liang; Donald F Becker
Journal:  Arch Biochem Biophys       Date:  2010-05-05       Impact factor: 4.013

3.  Evaluation of diagnostic fasting in the investigation of hypoglycemia in children omani experienc.

Authors:  Bhasker Bappal; Waad-Allah Mula-Abed
Journal:  Oman Med J       Date:  2007-10

4.  A new variant of glutaric aciduria type II: deficiency of beta-subunit of electron transfer flavoprotein.

Authors:  S Yamaguchi; T Orii; K Maeda; M Oshima; T Hashimoto
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

5.  Specific glutaryl-CoA dehydrogenating activity is deficient in cultured fibroblasts from glutaric aciduria patients.

Authors:  D B Hyman; K Tanaka
Journal:  J Clin Invest       Date:  1984-03       Impact factor: 14.808

6.  Biosynthesis of electron transfer flavoprotein in a cell-free system and in cultured human fibroblasts. Defect in the alpha subunit synthesis is a primary lesion in glutaric aciduria type II.

Authors:  Y Ikeda; S M Keese; K Tanaka
Journal:  J Clin Invest       Date:  1986-10       Impact factor: 14.808

7.  Symptoms and signs in organic acidurias.

Authors:  N J Brandt
Journal:  J Inherit Metab Dis       Date:  1984       Impact factor: 4.982

8.  Characterizing the transcriptional regulation of let-721, a Caenorhabditis elegans homolog of human electron flavoprotein dehydrogenase.

Authors:  Derek S Chew; Allan K Mah; David L Baillie
Journal:  Mol Genet Genomics       Date:  2009-09-23       Impact factor: 3.291

9.  Complementation analysis of fatty acid oxidation disorders.

Authors:  A Moon; W J Rhead
Journal:  J Clin Invest       Date:  1987-01       Impact factor: 14.808

10.  Glutaric acidaemia type II (multiple acyl-CoA dehydrogenation deficiency).

Authors:  S I Goodman; F E Frerman
Journal:  J Inherit Metab Dis       Date:  1984       Impact factor: 4.982

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