Literature DB >> 4258782

Succinyl-CoA: 3-ketoacid CoA-transferase deficiency. A cause for ketoacidosis in infancy.

J T Tildon, M Cornblath.   

Abstract

To explain the cause of a unique form of severe and intermittent ketoacidosis in an infant who expired after 6 months of life, tissue culture fibroblasts and post mortem tissue were examined for enzyme activities that catalyze glucose and ketoacid oxidation. No measurable succinyl-CoA: 3-ketoacid CoA-transferase (CoA-transferase) activity could be detected in homogenates of the post mortem brain, muscle and kidney tissue, or in the cultured skin fibroblasts. Since seven other enzyme activities involving both glycolysis and ketone body oxidation were present in these same tissues, it was reasonable to conclude that the observed absence of CoA-transferase activity was not an artifact of homogenate preparation. It was concluded that the absence of CoA-transferase activity resulted in a loss of intracellular homeostasis leading to ketoacidosis. In addition, the absence of this enzyme appears to be a reasonable explanation for the alteration in glucose metabolism that was previously reported in fibroblasts from this patient.

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Year:  1972        PMID: 4258782      PMCID: PMC302154          DOI: 10.1172/JCI106837

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  17 in total

1.  Fasting hypoglycaemia and metabolic acidosis associated with deficiency of hepatic fructose-1,6-diphosphatase activity.

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2.  Inherited propionyl-Coa carboxylase deficiency in "ketotic hyperglycinemia".

Authors:  Y E Hsia; K J Scully; L E Rosenberg
Journal:  J Clin Invest       Date:  1971-01       Impact factor: 14.808

3.  Activities of enzymes involved in acetoacetate utilization in adult mammalian tissues.

Authors:  D H Williamson; M W Bates; M A Page; H A Krebs
Journal:  Biochem J       Date:  1971-01       Impact factor: 3.857

4.  Coenzyme A transferase activity in rat brain.

Authors:  J T Tildon; A L Cone; M Cornblath
Journal:  Biochem Biophys Res Commun       Date:  1971-04-02       Impact factor: 3.575

5.  A new syndrome of ketoacidosis in infancy.

Authors:  M Cornblath; R L Gingell; G A Fleming; J T Tildon; A T Leffler; R A Wapnir
Journal:  J Pediatr       Date:  1971-09       Impact factor: 4.406

6.  Cell composition and metabolism in Kwashiorkor. (Severe protein-calorie malnutrition in children).

Authors:  J Metcoff; S Frenk; T Yoshida; R T Pinedo; E Kaiser; J D Hansen
Journal:  Medicine (Baltimore)       Date:  1966-09       Impact factor: 1.889

7.  Familial chronic acidosis due to an error in lactate and pyruvate metabolism.

Authors:  J C Haworth; J D Ford; M K Younoszai
Journal:  Can Med Assoc J       Date:  1967-09-23       Impact factor: 8.262

8.  Methylmalonic aciduria. An inborn error of metabolism leading to chronic metabolic acidosis.

Authors:  V G Oberholzer; B Levin; E A Burgess; W F Young
Journal:  Arch Dis Child       Date:  1967-10       Impact factor: 3.791

9.  Activity and intracellular distribution of enzymes of ketone-body metabolism in rat liver.

Authors:  D H Williamson; M W Bates; H A Krebs
Journal:  Biochem J       Date:  1968-07       Impact factor: 3.857

10.  Acetoacetate metabolism in infant and adult rat brain in vitro.

Authors:  T Ito; J H Quastel
Journal:  Biochem J       Date:  1970-02       Impact factor: 3.857

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  30 in total

1.  A new case of succinyl-CoA:acetoacetate transferase deficiency: favourable course despite very low residual activity.

Authors:  I Barić; V Sarnavka; K Fumić; M Maradin; D Begović; J P Ruiter; R J Wanders
Journal:  J Inherit Metab Dis       Date:  2001-02       Impact factor: 4.982

2.  Altered systemic ketone body metabolism in advanced heart failure.

Authors:  Ajit Janardhan; Jane Chen; Peter A Crawford
Journal:  Tex Heart Inst J       Date:  2011

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 case of succinyl-CoA: acetoacetate transferase deficiency.

Authors:  C Pérez-Cerdá; B Merinero; P Sanz; A Jiménez; C Hernández; M J García; M Ugarte
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

5.  Neonatal hypoglycaemia in severe succinyl-CoA: 3-oxoacid CoA-transferase deficiency.

Authors:  G T Berry; T Fukao; G A Mitchell; A Mazur; M Ciafre; J Gibson; N Kondo; M J Palmieri
Journal:  J Inherit Metab Dis       Date:  2001-10       Impact factor: 4.982

6.  Clinical approach to inherited metabolic diseases in the neonatal period: a 20-year survey.

Authors:  J M Saudubray; H Ogier; J P Bonnefont; A Munnich; A Lombes; F Hervé; G Mitchel; B P Thé; N Specola; P Parvy
Journal:  J Inherit Metab Dis       Date:  1989       Impact factor: 4.982

Review 7.  β-Hydroxybutyrate in the Brain: One Molecule, Multiple Mechanisms.

Authors:  Lavanya B Achanta; Caroline D Rae
Journal:  Neurochem Res       Date:  2016-11-08       Impact factor: 3.996

8.  Obligate role for ketone body oxidation in neonatal metabolic homeostasis.

Authors:  David G Cotter; D André d'Avignon; Anna E Wentz; Mary L Weber; Peter A Crawford
Journal:  J Biol Chem       Date:  2011-01-05       Impact factor: 5.157

9.  Symptoms and signs in organic acidurias.

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

10.  An alteration in glucose metabolism associated with a defect in ketone body metabolism.

Authors:  J T Tildon
Journal:  Proc Natl Acad Sci U S A       Date:  1973-01       Impact factor: 11.205

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