Literature DB >> 7951672

Genetic disorders of mitochondrial fatty acid oxidation.

C A Stanley1, D E Hale.   

Abstract

Genetic diseases of mitochondrial fatty acid oxidation have recently emerged as important disorders to consider in the differential diagnosis of hypoglycemia, cardiomyopathy, or skeletal muscle weakness in infants and children. A total of 16 different defects have been identified over the past decade that involve almost all of the possible enzyme steps in the pathway. One of these disorders, medium-chain acyl-coenzyme A dehydrogenase deficiency has a frequency as high as 1 in 10,000 births and is the single most common genetic defect of intermediary metabolism. The disorders are frequently mistaken for Reye syndrome or sudden infant death syndrome. Improved methods have simplified the diagnosis of some of the fatty acid oxidation defects. However, recognition of these disorders remains challenging. Rapid advances have continued to be made over the past year in defining clinical phenotypes, diagnostic methods, and therapeutic strategies. Familiarity with this new group of disorders is becoming increasingly important for general pediatricians as well as subspecialists in metabolism, endocrinology, gastroenterology, cardiology, neurology, and genetics.

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Year:  1994        PMID: 7951672     DOI: 10.1097/00008480-199408000-00021

Source DB:  PubMed          Journal:  Curr Opin Pediatr        ISSN: 1040-8703            Impact factor:   2.856


  3 in total

1.  Fatty acid oxidation in fibroblasts from patients with defects in beta-oxidation and in the respiratory chain.

Authors:  N Venizelos; U von Döbeln; L Hagenfeldt
Journal:  J Inherit Metab Dis       Date:  1998-06       Impact factor: 4.982

Review 2.  Knowledge gaps and research needs for understanding and treating neonatal hypoglycemia: workshop report from Eunice Kennedy Shriver National Institute of Child Health and Human Development.

Authors:  William W Hay; Tonse Nk Raju; Rosemary D Higgins; Satish C Kalhan; Sherin U Devaskar
Journal:  J Pediatr       Date:  2009-11       Impact factor: 4.406

3.  Measurement of tissue acyl-CoAs using flow-injection tandem mass spectrometry: acyl-CoA profiles in short-chain fatty acid oxidation defects.

Authors:  Andrew A Palladino; Jie Chen; Staci Kallish; Charles A Stanley; Michael J Bennett
Journal:  Mol Genet Metab       Date:  2012-10-18       Impact factor: 4.797

  3 in total

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