Congenital lactic acidosis due to a defect of pyruvate dehydrogenase complex (E1). Clinical, biochemical, nerve biopsy study and effect of therapy

Eur Neurol. 1990;30(3):123-7. doi: 10.1159/000117327.

Abstract

We report an 8-year-old patient with clinical features suggesting Leigh's syndrome and with a decreased activity of the E1 component of the pyruvate dehydrogenase complex in cultured skin fibroblasts. A nerve biopsy showed the presence of severe peripheral neuropathy, rarely described in the literature. The partial correction of lactic acidosis with oral sodium bicarbonate chronic therapy may result in a slow evolution of the clinical symptoms.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Acidosis, Lactic / congenital*
  • Acidosis, Lactic / drug therapy
  • Acidosis, Lactic / enzymology
  • Bicarbonates / therapeutic use
  • Biopsy
  • Brain Diseases, Metabolic / etiology*
  • Child
  • Fibroblasts / cytology
  • Fibroblasts / enzymology*
  • Humans
  • Leigh Disease / enzymology
  • Leigh Disease / etiology*
  • Male
  • Peripheral Nervous System Diseases / etiology*
  • Peripheral Nervous System Diseases / pathology
  • Pyruvate Dehydrogenase Complex Deficiency Disease*
  • Skin / cytology
  • Sodium / therapeutic use
  • Sodium Bicarbonate

Substances

  • Bicarbonates
  • Sodium Bicarbonate
  • Sodium