Literature DB >> 3924247

Defects of metabolism of fatty acids in the sudden infant death syndrome.

A J Howat, M J Bennett, S Variend, L Shaw, P C Engel.   

Abstract

Two hundred consecutive cases of the sudden infant death syndrome were reviewed for the presence of fat in the liver; 14 showed diffuse panlobular microvesicular fatty change indistinguishable from that found in Reye's syndrome. Samples of frozen liver were available in five of the 14 cases; histochemical analysis showed well preserved cytochrome oxidase and succinate dehydrogenase activity in all five, uncharacteristic of Reye's syndrome. Fatty acyl-coenzyme A dehydrogenase activity in the liver was assayed biochemically in two of the same five cases with severe hepatic fatty infiltration; both showed a defect in medium chain acyl-coenzyme A dehydrogenase activity using the substrate octanoyl-coenzyme A. Both cases also showed cerebral oedema in association with fatty infiltration of renal tubules, myocardium, and skeletal muscle, characteristic of Reye's syndrome. It is concluded that diffuse panlobular microvesicular fatty change of the liver in victims of the sudden infant death syndrome, although essentially non-specific, indicates that the state of mitochondrial enzymes should be investigated.

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Year:  1985        PMID: 3924247      PMCID: PMC1415947          DOI: 10.1136/bmj.290.6484.1771

Source DB:  PubMed          Journal:  Br Med J (Clin Res Ed)        ISSN: 0267-0623


  15 in total

1.  ENCEPHALOPATHY AND FATTY DEGENERATION OF THE VISCERA. A DISEASE ENTITY IN CHILDHOOD.

Authors:  R D REYE; G MORGAN; J BARAL
Journal:  Lancet       Date:  1963-10-12       Impact factor: 79.321

2.  Diagnostic criteria for influenza B-associated Reye's syndrome: clinical vs. pathologic criteria.

Authors:  L Corey; R J Rubin; D Bregman; M B Gregg
Journal:  Pediatrics       Date:  1977-11       Impact factor: 7.124

3.  Aetiology of Reye's syndrome.

Authors:  M H Bellman; S M Hall
Journal:  Arch Dis Child       Date:  1983-09       Impact factor: 3.791

4.  Reye's syndrome: 20 years on.

Authors:  A P Mowat
Journal:  Br Med J (Clin Res Ed)       Date:  1983-06-25

5.  Acute fatty liver of pregnancy and the microvesicular fat diseases.

Authors:  S Sherlock
Journal:  Gut       Date:  1983-04       Impact factor: 23.059

6.  Deficiency of medium chain fatty acylcoenzyme A dehydrogenase presenting as the sudden infant death syndrome.

Authors:  A J Howat; M J Bennett; S Variend; L Shaw
Journal:  Br Med J (Clin Res Ed)       Date:  1984-03-31

7.  The hepatic lesion in Reye's syndrome.

Authors:  K E Bove; A J McAdams; J C Partin; J S Partin; G Hug; W K Schubert
Journal:  Gastroenterology       Date:  1975-09       Impact factor: 22.682

8.  Comparison of cytosolic and mitochondrial hepatic enzyme alterations in Reye's syndrome.

Authors:  R A Mitchell; M L Ram; E L Arcinue; C H Chang
Journal:  Pediatr Res       Date:  1980-11       Impact factor: 3.756

9.  Medium-chain acyl-CoA dehydrogenase deficiency in children with non-ketotic hypoglycemia and low carnitine levels.

Authors:  C A Stanley; D E Hale; P M Coates; C L Hall; B E Corkey; W Yang; R I Kelley; E L Gonzales; J R Williamson; L Baker
Journal:  Pediatr Res       Date:  1983-11       Impact factor: 3.756

Review 10.  Encephalopathy of Reye's syndrome: a review of pathogenetic hypotheses.

Authors:  G R DeLong; T H Glick
Journal:  Pediatrics       Date:  1982-01       Impact factor: 7.124

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  22 in total

1.  Acquired deficiency of long-chain acyl-CoA dehydrogenase in liver: a cautionary tale.

Authors:  F Allison; M J Bennett; R J Pollitt; S Variend
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

2.  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

3.  Intra-alveolar haemorrhage in sudden infant death syndrome: a cause for concern?

Authors:  N Yukawa; N Carter; G Rutty; M A Green
Journal:  J Clin Pathol       Date:  1999-08       Impact factor: 3.411

4.  Generalised dicarboxylic aciduria: a common finding in neonates.

Authors:  M Downing; P Rose; M J Bennett; N J Manning; R J Pollitt
Journal:  J Inherit Metab Dis       Date:  1989       Impact factor: 4.982

5.  Environmental findings and sudden infant death syndrome.

Authors:  R G Carpenter; A Gardner
Journal:  Lung       Date:  1990       Impact factor: 2.584

6.  Inherited disorders of straight chain fatty acid oxidation.

Authors:  R J Pollitt
Journal:  Arch Dis Child       Date:  1987-01       Impact factor: 3.791

Review 7.  Recent theories on the cause of cot death.

Authors:  A D Milner
Journal:  Br Med J (Clin Res Ed)       Date:  1987-11-28

8.  Abnormal expression of glucose-6-phosphatase in preterm infants.

Authors:  R Hume; A Burchell
Journal:  Arch Dis Child       Date:  1993-02       Impact factor: 3.791

9.  Medium-chain acyl-CoA dehydrogenase deficiency does not correlate with apparent life-threatening events and the sudden infant death syndrome: results from phenylpropionate loading tests and DNA analysis.

Authors:  J M Penzien; G Molz; U N Wiesmann; J P Colombo; R Bühlmann; B Wermuth
Journal:  Eur J Pediatr       Date:  1994-05       Impact factor: 3.183

10.  Biochemical characterization of a variant human medium-chain acyl-CoA dehydrogenase with a disease-associated mutation localized in the active site.

Authors:  B Küchler; A G Abdel-Ghany; P Bross; A Nandy; I Rasched; S Ghisla
Journal:  Biochem J       Date:  1999-01-15       Impact factor: 3.857

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