Genetics and variation in phenotype in Noonan syndrome

Horm Res. 2004:62 Suppl 3:56-9. doi: 10.1159/000080500.

Abstract

Noonan syndrome is a well-known clinical entity comprising multiple congenital anomalies characterized by typical facial features, short stature and congenital heart defect. Approximately 50% of cases are sporadic. Familial cases are generally autosomal dominant. In 2001 a gene responsible for Noonan syndrome, PTPN11, encoding for the non-receptor protein tyrosine phosphatase SHP-2, was identified. Mutation analysis of the PTPN11 gene was carried out in Nijmegen in 150 patients with Noonan syndrome. Mutations were found in 68 patients (45%), the most common being A922G in exon 8. In exon 4 a mutation was found that encoded the C-SH2 domain of the PTPN11 gene in two unique patients who shared some uncommon features. A 218C-->T mutation was found in exon 3 in one patient with Noonan syndrome and mild juvenile myelomonocytic leukaemia.

MeSH terms

  • Adenine
  • Adult
  • Child
  • Cytosine
  • Exons / genetics
  • Female
  • Genetic Variation*
  • Genotype
  • Guanine
  • Humans
  • Infant
  • Intracellular Signaling Peptides and Proteins
  • Male
  • Mutation*
  • Noonan Syndrome / genetics*
  • Noonan Syndrome / pathology
  • Phenotype*
  • Protein Tyrosine Phosphatase, Non-Receptor Type 11
  • Protein Tyrosine Phosphatases / genetics*
  • SH2 Domain-Containing Protein Tyrosine Phosphatases
  • Thymine
  • src Homology Domains / genetics

Substances

  • Intracellular Signaling Peptides and Proteins
  • Guanine
  • Cytosine
  • PTPN11 protein, human
  • Protein Tyrosine Phosphatase, Non-Receptor Type 11
  • Protein Tyrosine Phosphatases
  • SH2 Domain-Containing Protein Tyrosine Phosphatases
  • Adenine
  • Thymine