Literature DB >> 6434827

A new patient with dicarboxylic aciduria suggestive of medium-chain Acyl-CoA dehydrogenase deficiency presenting as Reye's syndrome.

J A Del Valle, M J Garcia, B Merinero, C Pérez-Cerdá, F Roman, A Jimenez, M Ugarte, M Martínez-Pardo, C Ludeña, C Camarero.   

Abstract

A new patient with medium-chain dicarboxylic aciduria and suberyl glycinuria during an attack of acute illness is reported. When, inadvertently he was given medium-chain triglycerides for 2 days, the excretion of abnormal metabolites of medium-chain fatty acids increased and hepatomegaly became more pronounced. During remission a low excretion of the metabolites were observed. After 16 h of fasting hypoglycaemia was accompanied by an increase of urinary dicarboxylic acids and psi-hydroxyacids similar to that found on admission. Interestingly this urinary organic acid pattern persisted 8 h after intravenous administration of glucose. In a blood sample obtained after 16 h of fasting there was hypoketonaemia and increased levels of total free fatty acids, octanoic, decanoic and cis-4-decenoic acids. These biochemical data suggest the existence of a deficiency at the level of medium-chain acyl-CoA dehydrogenase.

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Year:  1984        PMID: 6434827     DOI: 10.1007/bf01805804

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


  8 in total

1.  Suberylglycine excretion in the urine from a patient with dicarboxylic aciduria.

Authors:  N Gregersen; R Lauritzen; K Rasmussen
Journal:  Clin Chim Acta       Date:  1976-08-02       Impact factor: 3.786

2.  In vitro fibroblast studies in a patient with C6-C10-dicarboxylic aciduria: evidence for a defect in general acyl-CoA dehydrogenase.

Authors:  S Kølvraa; N Gregersen; E Christensen; N Hobolth
Journal:  Clin Chim Acta       Date:  1982-11-24       Impact factor: 3.786

3.  Dicarboxylic aciduria: the response to fasting.

Authors:  R J Truscott; L Hick; C Pullin; B Halpern; B Wilcken; H Griffiths; M Silink; H Kilham; F Grunseit
Journal:  Clin Chim Acta       Date:  1979-05-16       Impact factor: 3.786

4.  General (medium-chain) acyl-CoA dehydrogenase deficiency (non-ketotic dicarboxylic aciduria): quantitative urinary excretion pattern of 23 biologically significant organic acids in three cases.

Authors:  N Gregersen; S Kølvraa; K Rasmussen; P B Mortensen; P Divry; M David; N Hobolth
Journal:  Clin Chim Acta       Date:  1983-08-15       Impact factor: 3.786

5.  Dicarboxylic aciduria: deficient [1-14C]octanoate oxidation and medium-chain acyl-CoA dehydrogenase in fibroblasts.

Authors:  W J Rhead; B A Amendt; K S Fritchman; S J Felts
Journal:  Science       Date:  1983-07-01       Impact factor: 47.728

6.  Isovalerylglucuronide, a new urinary metabolite in isovaleric acidemia. Identification problems due to rearrangement reactions.

Authors:  L Dorland; M Duran; S K Wadman; A Niederwieser; L Bruinvis; D Ketting
Journal:  Clin Chim Acta       Date:  1983-10-31       Impact factor: 3.786

7.  Episodic hypoglycemia with psi-hydroxy fatty acid excretion.

Authors:  E Colle; O A Mamer; J A Montgomery; J D Miller
Journal:  Pediatr Res       Date:  1983-02       Impact factor: 3.756

8.  Intermittent non-ketotic dicarboxylic aciduria in two siblings with hypoglycaemia: an apparent defect in beta-oxidation of fatty acids.

Authors:  E W Naylor; L L Mosovich; R Guthrie; J E Evans; H Tieckelmann
Journal:  J Inherit Metab Dis       Date:  1980       Impact factor: 4.982

  8 in total
  5 in total

1.  Early diagnosis and treatment of neonatal medium-chain acyl-CoA dehydrogenase deficiency: report of two siblings.

Authors:  C Catzeflis; C Bachmann; D E Hale; P M Coates; U Wiesmann; J P Colombo; F Joris; G Délèze
Journal:  Eur J Pediatr       Date:  1990-05       Impact factor: 3.183

2.  Molecular survey of a prevalent mutation, 985A-to-G transition, and identification of five infrequent mutations in the medium-chain Acyl-CoA dehydrogenase (MCAD) gene in 55 patients with MCAD deficiency.

Authors:  I Yokota; P M Coates; D E Hale; P Rinaldo; K Tanaka
Journal:  Am J Hum Genet       Date:  1991-12       Impact factor: 11.025

Review 3.  The inborn errors of mitochondrial fatty acid oxidation.

Authors:  C Vianey-Liaud; P Divry; N Gregersen; M Mathieu
Journal:  J Inherit Metab Dis       Date:  1987       Impact factor: 4.982

4.  Investigation of enzyme defects in children with lactic acidosis.

Authors:  B Merinero; C Pérez-Cerda; M Ugarte
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

Review 5.  Medium chain acyl-CoA dehydrogenase deficiency.

Authors:  E H Touma; C Charpentier
Journal:  Arch Dis Child       Date:  1992-01       Impact factor: 3.791

  5 in total

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