Mitochondrial myopathy with lactic acidaemia, Fanconi-De Toni-Debré syndrome and a disturbed succinate: cytochrome c oxidoreductase activity

Eur J Pediatr. 1988 May;147(4):418-21. doi: 10.1007/BF00496424.

Abstract

A patient with severe muscular hypotonia, failure to thrive, a metabolic acidosis and a renal tubular dysfunction is presented. The disease followed a fatal course. Blood lactate and pyruvate levels as well as lactate/pyruvate ratios were strongly elevated. There were a massive excretion of lactate in urine, a generalized hyperaminoaciduria, a proteinuria and a mellituria. The carnitine concentration was diminished in blood and muscle tissue. Biochemical investigations of skeletal muscle and liver tissue revealed a defect in the respiratory chain at the level of succinate: cytochrome c oxidoreductase. The defect could not be demonstrated in cultured fibroblasts.

Publication types

  • Case Reports

MeSH terms

  • Acidosis, Lactic / metabolism
  • Carnitine / metabolism
  • Fanconi Syndrome / enzymology
  • Fanconi Syndrome / metabolism*
  • Female
  • Humans
  • Infant, Newborn
  • Lactates / blood*
  • Lactates / urine
  • Lactic Acid
  • Mitochondria, Muscle / enzymology
  • Mitochondria, Muscle / metabolism*
  • Muscular Diseases / enzymology
  • Muscular Diseases / metabolism*
  • Oxidoreductases / metabolism*
  • Pyruvates / blood
  • Succinate Cytochrome c Oxidoreductase / metabolism*
  • Ubiquinone / metabolism

Substances

  • Lactates
  • Pyruvates
  • Ubiquinone
  • Lactic Acid
  • Oxidoreductases
  • Succinate Cytochrome c Oxidoreductase
  • Carnitine