A new T677C mutation of the aspartoacylase gene encodes for a protein with no enzymatic activity

Clin Biochem. 2008 May;41(7-8):611-5. doi: 10.1016/j.clinbiochem.2008.01.021. Epub 2008 Feb 7.

Abstract

Objective: To verify the effect of and to date the unknown T677C mutation of the human N-acetylaspartoacylase (hASPA) gene on the function of the mutated enzyme.

Design and methods: Wild type and I226T-mutated proteins were expressed and purified from a transformed Escherichia coli colony. Enzymatic activities were measured in the presence of varying substrate concentrations.

Results: Whilst kinetic parameters of wild type hASPA were in line with data in literature, I226T-mutated hASPA showed no enzymatic activity.

Conclusion: Data indicated that this new mutation might be responsible in homozygosis for the phenotype corresponding to Canavan disease.

Publication types

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

MeSH terms

  • Amidohydrolases / biosynthesis
  • Amidohydrolases / genetics*
  • Amidohydrolases / isolation & purification
  • Amino Acid Substitution / genetics
  • Canavan Disease / diagnosis
  • Canavan Disease / enzymology
  • Canavan Disease / genetics
  • Child, Preschool
  • Cytosine
  • Enzyme Activation / genetics
  • Female
  • Humans
  • Male
  • Mutagenesis, Site-Directed
  • Mutation / genetics*
  • Thymine

Substances

  • Cytosine
  • Amidohydrolases
  • aspartoacylase
  • Thymine