Cerebro-hepato-renal syndrome of Zellweger. A report of eight cases with comments upon the incidence, the liver lesion, and a fault in pipecolic acid metabolism

J Pediatr. 1975 Mar;86(3):382-7. doi: 10.1016/s0022-3476(75)80967-x.

Abstract

Studies of eight patients with this syndrome confirm the characteristic pattern of abnormalities and the autosomal recessive mode of inheritance. The incidence is estimated to be approximately 1 in 100,000 live births. The liver lesion proved to be variable, with features of progressive parenchymal damage rather than a developmental defect of small bile ducts as previously suggested. Elevated levels of pipecolic acid were found in blood and urine and may be related to the basic defect.

MeSH terms

  • Acidosis / blood
  • Acidosis / epidemiology
  • Acidosis / genetics
  • Australia
  • Bone Diseases / blood
  • Bone Diseases / epidemiology*
  • Bone Diseases / genetics
  • Brain Diseases / blood
  • Brain Diseases / epidemiology*
  • Brain Diseases / genetics
  • Humans
  • Iron / blood
  • Iron / metabolism
  • Kidney / pathology
  • Kidney Diseases / blood
  • Kidney Diseases / epidemiology*
  • Kidney Diseases / genetics
  • Liver / pathology
  • Liver Diseases / blood
  • Liver Diseases / epidemiology*
  • Liver Diseases / genetics
  • Pedigree
  • Pipecolic Acids / blood
  • Pipecolic Acids / urine
  • Syndrome

Substances

  • Pipecolic Acids
  • Iron