Literature DB >> 7432384

Systemic carnitine deficiency--a treatable inherited lipid-storage disease presenting as Reye's syndrome.

P R Chapoy, C Angelini, W J Brown, J E Stiff, A L Shug, S D Cederbaum.   

Abstract

A 3 1/2-year-old boy presented at three months of age with an acute episode of lethargy, somnolence, hypoglycemia, hepatomegaly, and cardiomegaly, which responded poorly to restoration of the blood sugar level to normal. The absence of ketonuria during subsequent episodes of severe hypoglycemia prompted a search for a defect in fatty acid oxidation. Plasma carnitine (2.0 to 5.0 mumol per liter), muscle carnitine (0.01 to 0.02 mumol per gram, wet weight) and liver carnitine (0.021 to 0.065 mumol per gram, wet weight) were all less than 5 per cent of the normal mean. During a 36-hour fast, ketones were barely detectable. Prolonged treatment with oral carnitine over a six-month period resulted in increased muscle strength, a dramatic reduction in cardiac size, relief of cardiomyopathy, partial repletion of carnitine levels in plasma and muscle, and complete repletion in the liver. Systemic carnitine deficiency is an easily treatable cause of recurrent Reye's-like syndrome. Its diagnosis requires measurement of carnitine levels.

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Year:  1980        PMID: 7432384     DOI: 10.1056/NEJM198012113032403

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  43 in total

Review 1.  Role of plasma membrane transporters in muscle metabolism.

Authors:  A Zorzano; C Fandos; M Palacín
Journal:  Biochem J       Date:  2000-08-01       Impact factor: 3.857

2.  Subnormal carnitine levels and their correction in artificially fed patients from a neurological intensive care unit: a pilot study.

Authors:  J Schäfer; H Reichmann
Journal:  J Neurol       Date:  1990-06       Impact factor: 4.849

3.  Carnitine deficiency with cardiomyopathy presenting as neonatal hydrops: successful response to carnitine therapy.

Authors:  P Steenhout; C Elmer; A Clercx; D Blum; D Gnat; S van Erum; F Vertongen; E Vamos
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

4.  Importance of molecular diagnosis in the accurate diagnosis of systemic carnitine deficiency.

Authors:  Toshiaki Hitomi; Norio Matsuura; Yosuke Shigematsu; Yoshiyuki Okano; Eri Shinozaki; Masahiko Kawai; Hatasu Kobayashi; Kouji H Harada; Akio Koizumi
Journal:  J Genet       Date:  2015-03       Impact factor: 1.166

Review 5.  Biochemical relationships between Reye's and Reye's-like metabolic and toxicological syndromes.

Authors:  J Osterloh; W Cunningham; A Dixon; D Combest
Journal:  Med Toxicol Adverse Drug Exp       Date:  1989 Jul-Aug

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

Authors:  M Duran; J B de Klerk; S K Wadman; H R Scholte; R P Beekman; F G Jennekens
Journal:  Eur J Pediatr       Date:  1984-08       Impact factor: 3.183

7.  Carnitine metabolism and inborn errors.

Authors:  A G Engel; C J Rebouche
Journal:  J Inherit Metab Dis       Date:  1984       Impact factor: 4.982

8.  Fatal lipid storage myopathy with deficiency of cytochrome-c-oxidase and carnitine. A contribution to the combined cytochemical-finestructural identification of cytochrome-c-oxidase in longterm frozen muscle.

Authors:  J Müller-Höcker; D Pongratz; T Deufel; J M Trijbels; W Endres; G Hübner
Journal:  Virchows Arch A Pathol Anat Histopathol       Date:  1983

Review 9.  Endogenous ornithine in search for CNS functions and therapeutic applications.

Authors:  N Seiler; G Daune-Anglard
Journal:  Metab Brain Dis       Date:  1993-09       Impact factor: 3.584

10.  Muscle carnitine deficiency: adult onset lipid storage myopathy with sensory neuropathy.

Authors:  Wei Zhang; Jianting Miao; Guangyun Zhang; Rui Liu; Dawei Zhang; Qun Wan; Yingxin Yu; Gang Zhao; Zhuyi Li
Journal:  Neurol Sci       Date:  2009-09-19       Impact factor: 3.307

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