Literature DB >> 2279505

Medium-chain acyl CoA dehydrogenase deficiency: electron microscopic differentiation from Reye syndrome.

R Santer1, E Schmidt-Sommerfeld, Y K Leung, J E Fischer, E Lebenthal.   

Abstract

Inborn errors involving the oxidative metabolism of fatty acids may present clinically with a Reye syndrome-like picture. This case report of a patient with medium-chain acyl CoA dehydrogenase (MCAD) deficiency illustrates that electron microscopy may help to differentiate this disorder from Reye syndrome even if a liver biopsy is performed in a patient who recovered from an acute metabolic decompensation. Together with this case, a review of the few reports in the literature of pathological findings in MCAD deficiency is given. Changes uncharacteristic for Reye syndrome are a large-droplet steatosis and the presence of distinctive mitochondrial abnormalities on electron microscopy. The detection of an electron dense mitochondrial matrix and a widened space of inner mitochondrial membranes rules out Reye syndrome and is suggestive of a disorder of mitochondrial fatty acid oxidation.

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Year:  1990        PMID: 2279505     DOI: 10.1007/bf02072051

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


  10 in total

1.  Mitochondrial ultrastructure in Reye's syndrome (encephalopathy and fatty degeneration of the viscera).

Authors:  J C Partin; W K Schubert; J S Partin
Journal:  N Engl J Med       Date:  1971-12-09       Impact factor: 91.245

2.  Ultrastructure of liver in inherited disorders of fat oxidation.

Authors:  B D Lake; P T Clayton; J V Leonard; A K Bhuiyan; K Bartlett; A A Green
Journal:  Lancet       Date:  1987-02-14       Impact factor: 79.321

3.  A simple screening test for medium-chain acyl CoA dehydrogenase deficiency.

Authors:  G Rumsby; J W Seakins; J V Leonard
Journal:  Lancet       Date:  1986-08-23       Impact factor: 79.321

Review 4.  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

5.  Medium-chain acyl-CoA dehydrogenase deficiency. Diagnosis by stable-isotope dilution measurement of urinary n-hexanoylglycine and 3-phenylpropionylglycine.

Authors:  P Rinaldo; J J O'Shea; P M Coates; D E Hale; C A Stanley; K Tanaka
Journal:  N Engl J Med       Date:  1988-11-17       Impact factor: 91.245

6.  Medium-chain and long-chain acyl CoA dehydrogenase deficiency: clinical, pathologic and ultrastructural differentiation from Reye's syndrome.

Authors:  W R Treem; C A Witzleben; D A Piccoli; C A Stanley; D E Hale; P M Coates; J B Watkins
Journal:  Hepatology       Date:  1986 Nov-Dec       Impact factor: 17.425

7.  Diagnostic and therapeutic implications of medium-chain acylcarnitines in the medium-chain acyl-coA dehydrogenase deficiency.

Authors:  C R Roe; D S Millington; D A Maltby; T P Bohan; S G Kahler; R A Chalmers
Journal:  Pediatr Res       Date:  1985-05       Impact factor: 3.756

8.  Recognition of medium-chain acyl-CoA dehydrogenase deficiency in asymptomatic siblings of children dying of sudden infant death or Reye-like syndromes.

Authors:  C R Roe; D S Millington; D A Maltby; P Kinnebrew
Journal:  J Pediatr       Date:  1986-01       Impact factor: 4.406

9.  Familial Reye-like syndrome: a presentation of medium-chain acyl-coenzyme A dehydrogenase deficiency.

Authors:  B Taubman; D E Hale; R I Kelley
Journal:  Pediatrics       Date:  1987-03       Impact factor: 7.124

10.  Genetic deficiency of medium-chain acyl coenzyme A dehydrogenase: studies in cultured skin fibroblasts and peripheral mononuclear leukocytes.

Authors:  P M Coates; D E Hale; C A Stanley; B E Corkey; J A Cortner
Journal:  Pediatr Res       Date:  1985-07       Impact factor: 3.756

  10 in total
  3 in total

1.  Clinical and biological features at diagnosis in mitochondrial fatty acid beta-oxidation defects: a French pediatric study of 187 patients.

Authors:  Julien Baruteau; Philippe Sachs; Pierre Broué; Michèle Brivet; Hendy Abdoul; Christine Vianey-Saban; Hélène Ogier de Baulny
Journal:  J Inherit Metab Dis       Date:  2012-10-03       Impact factor: 4.982

Review 2.  Medium-chain acyl-CoA dehydrogenase deficiency: molecular aspects.

Authors:  Y Matsubara; K Narisawa; K Tada
Journal:  Eur J Pediatr       Date:  1992-03       Impact factor: 3.183

Review 3.  Mitochondrial dysfunction in fatty acid oxidation disorders: insights from human and animal studies.

Authors:  Moacir Wajner; Alexandre Umpierrez Amaral
Journal:  Biosci Rep       Date:  2015-11-20       Impact factor: 3.840

  3 in total

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