[Multiple acyl-CoA dehydrogenase deficiency (MADD): a curable cause of genetic muscular lipidosis]

Rev Neurol (Paris). 2010 Mar;166(3):289-94. doi: 10.1016/j.neurol.2009.05.009. Epub 2009 Jul 9.
[Article in French]

Abstract

Introduction: Multiple acyl-CoA dehydrogenase deficiency (MADD) is a rare genetic disease involving fatty acid oxidation. It is due to the deficiency of one of the two electron transporters: electron transfer flavoprotein (ETF) or electron transfer flavoprotein ubiquinone oxydoreductase (ETF-QO). Symptoms begin more often in childhood or in young adulthood with a multisystemic disease with encephalopathy or muscular weakness.

Case reports: We report here two adult cases with ETF-QO deficiency, confirmed by mutation analysis (ETFDH gene), revealed by a muscular weakness associated with muscle lipidosis. One of our patients presented an acute encephalopathy with vomiting ten years before the onset of muscular symptoms. The second patient exhibited a slowly progressive pelvic girdle muscle weakness. Diagnosis was established by characteristic abnormalities of acylcarnitine profile by tandem mass spectrometry. For both patients, a dramatic clinical improvement was observed under treatment with riboflavine and L-carnitine.

Conclusion: Since it is a treatable disorder, this diagnosis must be considered by performing an acylcarnitine profile in all patients presenting with an unexplained muscular weakness.

Publication types

  • Case Reports
  • English Abstract

MeSH terms

  • Acyl-CoA Dehydrogenases / deficiency*
  • Adult
  • Biopsy
  • Brain Diseases, Metabolic / genetics
  • Carnitine / analogs & derivatives
  • Carnitine / analysis
  • Carnitine / metabolism
  • Coloring Agents
  • DNA Mutational Analysis
  • Electron Transport / genetics
  • Electron-Transferring Flavoproteins / genetics
  • Electron-Transferring Flavoproteins / metabolism
  • Female
  • Humans
  • Lipid Metabolism, Inborn Errors / enzymology*
  • Lipid Metabolism, Inborn Errors / genetics*
  • Lipid Metabolism, Inborn Errors / pathology
  • Lipidoses / drug therapy*
  • Lipidoses / genetics*
  • Lipidoses / pathology
  • Male
  • Middle Aged
  • Muscle Weakness / etiology
  • Muscle Weakness / genetics
  • Muscle, Skeletal / pathology
  • Riboflavin / metabolism
  • Tandem Mass Spectrometry
  • Young Adult

Substances

  • Coloring Agents
  • Electron-Transferring Flavoproteins
  • acylcarnitine
  • Acyl-CoA Dehydrogenases
  • Carnitine
  • Riboflavin