Literature DB >> 6468448

Systemic carnitine deficiency: benefit of oral carnitine supplements vs. persisting biochemical abnormalities.

M Duran, J B de Klerk, S K Wadman, H R Scholte, R P Beekman, F G Jennekens.   

Abstract

A patient is described who was admitted with a condition similar to the Reye syndrome at the age of 9 months. Hypoglycemia, hyperammonemia, hepatomegaly, and lethargy were present. The plasma concentrations of free and acylcarnitine were extremely low and the urine contained excessive amounts of dicarboxylic acids. Extensive biochemical and histological investigations of biopsied liver and muscle led to the diagnosis of systemic carnitine deficiency. The patient was put on oral carnitine treatment, upon which he remained clinically well. A prolonged fasting test during this treatment gave abnormal results: there was no ketonemia, but an increase of omega-oxidation of fatty acids. In spite of the treatment the liver and muscle carnitine content remained below normal.

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Year:  1984        PMID: 6468448     DOI: 10.1007/bf00442456

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


  22 in total

1.  Lethal hypoglycemia in a child with a deficiency of 3-hydroxy-3-methylglutarylcoenzyme A lyase.

Authors:  R B Schutgens; H Heymans; A Ketel; H A Veder; M Duran; D Ketting; S K Wadman
Journal:  J Pediatr       Date:  1979-01       Impact factor: 4.406

2.  Gas chromatography of urinary N-phenylacetylglutamine.

Authors:  J P Kamerling; M Brouwer; D Ketting; S K Wadman
Journal:  J Chromatogr       Date:  1979-10-11

3.  Fasting hypoglycemia resulting from hepatic carnitine palmitoyl transferase deficiency.

Authors:  P F Bougnères; J M Saudubray; C Marsac; O Bernard; M Odièvre; J Girard
Journal:  J Pediatr       Date:  1981-05       Impact factor: 4.406

4.  Systemic carnitine deficiency simulating recurrent Reye syndrome.

Authors:  A M Glasgow; G Eng; A G Engel
Journal:  J Pediatr       Date:  1980-05       Impact factor: 4.406

5.  Carnitine metabolism in normal-weight and obese human subjects during fasting.

Authors:  C L Hoppel; S M Genuth
Journal:  Am J Physiol       Date:  1980-05

6.  Fatal cases of lipid storage myopathy with carnitine deficiency.

Authors:  F Cornelio; S Di Donato; D Peluchetti; A Bizze; B Bertagnolio; A D'Angelo; U Wiesmann
Journal:  J Neurol Neurosurg Psychiatry       Date:  1977-02       Impact factor: 10.154

7.  Hypoketosis as a cause of symptoms in childhood hypoglycemia.

Authors:  H L Teijema; H H van Gelderen; M A Giesberts
Journal:  Eur J Pediatr       Date:  1980-06       Impact factor: 3.183

8.  Multiple acyl-Co A dehydrogenation deficiency (MADD) in a boy with nonketotic hypoglycemia, hepatomegaly, muscle hypotonia and cardiomyopathy. Detection of N-isovalerylglutamic acid and its monoamide.

Authors:  A Niederwieser; B Steinmann; U Exner; F Neuheiser; U Redweik; M Wang; S Rampini; U Wendel
Journal:  Helv Paediatr Acta       Date:  1983-03

9.  Variation in plasma ketone bodies during a 24-hour fast in normal and in hypoglycemic children: relationship to age.

Authors:  J M Saudubray; C Marsac; J M Limal; E Dumurgier; C Charpentier; H Ogier; F X Coudè
Journal:  J Pediatr       Date:  1981-06       Impact factor: 4.406

10.  Medium-chain triglyceride medication as a pitfall in the diagnosis of non-ketotic C6-C10-dicarboxylic acidurias.

Authors:  P B Mortensen; N Gregersen
Journal:  Clin Chim Acta       Date:  1980-04-11       Impact factor: 3.786

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  4 in total

1.  Mitochondrial very long-chain acyl-CoA dehydrogenase deficiency with a mild clinical course.

Authors:  B Merinero; C Pérez-Cerdá; M J Garcia; J Gangoiti; L M Font; M T Garcia Silva; C Vianey-Saban; M Duran; M Ugarte
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

2.  The effect of fasting, long-chain triglyceride load and carnitine load on plasma long-chain acylcarnitine levels in mitochondrial very long-chain acyl-CoA dehydrogenase deficiency.

Authors:  C G Costa; L Dorland; I T de Almeida; C Jakobs; M Duran; B T Poll-The
Journal:  J Inherit Metab Dis       Date:  1998-06       Impact factor: 4.982

3.  Oral carnitine therapy in children with cystinosis and renal Fanconi syndrome.

Authors:  W A Gahl; I Bernardini; M Dalakas; W B Rizzo; G S Harper; J M Hoeg; O Hurko; J Bernar
Journal:  J Clin Invest       Date:  1988-02       Impact factor: 14.808

4.  Hyperammonemic encephalopathy caused by carnitine deficiency.

Authors:  Berkeley N Limketkai; Stephen D Zucker
Journal:  J Gen Intern Med       Date:  2007-12-13       Impact factor: 5.128

  4 in total

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