Description and functional implications of a novel mutation in the sex-determining gene SRY

Hum Mutat. 1994;3(3):200-4. doi: 10.1002/humu.1380030305.

Abstract

The sex-determining gene SRY was screened for molecular alteration in an XY sex-reversed female by single-strand conformation polymorphism (SSCP) technique. An A-to-G transition was detected which leads to an exchange of a tyrosine by a cysteine in the SRY protein. The affected tyrosine residue located at the C terminus of the DNA binding protein is evolutionarily strongly conserved among the members of the HMG box containing proteins. Using gel shift assay and peptide synthesis such a mutation is shown to abolish the SRY protein DNA binding ability. The involvement of this particular amino acid in the binding specificity is also discussed.

Publication types

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

MeSH terms

  • Adolescent
  • Amenorrhea / genetics*
  • Amino Acid Sequence
  • Animals
  • Base Sequence
  • DNA / blood
  • DNA / isolation & purification
  • DNA Primers
  • DNA-Binding Proteins / genetics*
  • Female
  • Humans
  • Leukocytes / metabolism
  • Molecular Sequence Data
  • Nuclear Proteins*
  • Point Mutation*
  • Polymerase Chain Reaction
  • Sequence Homology, Amino Acid
  • Sex Determination Analysis*
  • Sex-Determining Region Y Protein
  • Transcription Factors / genetics
  • X Chromosome
  • Y Chromosome

Substances

  • DNA Primers
  • DNA-Binding Proteins
  • Nuclear Proteins
  • SRY protein, human
  • Sex-Determining Region Y Protein
  • Transcription Factors
  • DNA