Autosomal recessive chronic granulomatous disease associated with 18q-syndrome and end-stage renal failure due to Henoch-Schönlein nephritis

Eur J Pediatr. 1991 Mar;150(5):325-6. doi: 10.1007/BF01955932.

Abstract

Chronic granulomatous disease (CGD) is an inherited disorder in which phagocytes, including polymorphonuclear neutrophils, are unable to generate oxygen-derived microbicidal compounds, among them superoxide. Two main types of CGD are known, an X-linked form which is normally associated with the absence of cytochrome b558, a component of the membrane-associated reduced nicotinamide adenine dinucleotide phosphate (NADPH) oxidase which generates superoxide and an autosomal recessive form, in which cytochrome b558 is present, caused by the deficiency of a cytosolic factor required to activate NADPH oxidase. Patients with the X-linked type are highly susceptible to infections; those with the autosomal recessive form may be less severely affected. We report the unusual association of autosomal CGD with 18q--syndrome in a girl who developed terminal renal insufficiency caused by Henoch-Schönlein nephritis, and speculate on the possibility that the gene defect of autosomal recessive CGD may be located on chromosome 18.

Publication types

  • Case Reports

MeSH terms

  • Chromosome Aberrations / genetics*
  • Chromosome Disorders
  • Chromosomes, Human, Pair 18*
  • Female
  • Genes, Recessive*
  • Granulomatous Disease, Chronic / complications
  • Granulomatous Disease, Chronic / genetics*
  • Humans
  • IgA Vasculitis / complications*
  • Infant, Newborn
  • Kidney Failure, Chronic / etiology*
  • Nephritis, Hereditary / complications*
  • Neutrophils / metabolism
  • Oxidation-Reduction
  • Syndrome