Congenital nephrotic syndrome of the Finnish type is not associated with the Pax-2 gene despite the promising transgenic animal model

Genomics. 1994 Feb;19(3):570-2. doi: 10.1006/geno.1994.1109.

Abstract

Congenital nephrotic syndrome of the Finnish type (CNF) is an autosomal recessive disease with an incidence of 1 in 8000 in Finland. CNF is characterized by massive proteinuria and nephrotic syndrome at birth. In a recent report, deregulation of expression of the gene coding for the Pax-2 DNA-binding protein was shown to generate severe kidney abnormalities in transgenic mice resembling the clinical and pathological findings in congenital nephrotic syndrome, making it a candidate gene for CNF. However, in this study, we have unequivocally excluded the Pax-2 gene locus as a causative for congenital nephrotic syndrome of the Finnish type.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Basement Membrane / chemistry
  • Basement Membrane / pathology
  • Chromosome Mapping
  • Chromosomes, Human, Pair 10
  • DNA-Binding Proteins / genetics*
  • Disease Models, Animal*
  • Finland / epidemiology
  • Genes*
  • Genes, Recessive
  • Genetic Markers
  • Humans
  • Incidence
  • Kidney Glomerulus / chemistry
  • Kidney Glomerulus / pathology
  • Lod Score
  • Mice
  • Mice, Transgenic*
  • Nephrotic Syndrome / congenital
  • Nephrotic Syndrome / epidemiology
  • Nephrotic Syndrome / genetics*
  • PAX2 Transcription Factor
  • Transcription Factors / genetics*

Substances

  • DNA-Binding Proteins
  • Genetic Markers
  • PAX2 Transcription Factor
  • PAX2 protein, human
  • Pax2 protein, mouse
  • Transcription Factors