Occurrence of thanatophoric dysplasia type I (R248C) and hypochondroplasia (N540K) mutations in two patients with achondroplasia phenotype

Am J Med Genet. 2001 Dec 15;104(4):277-81. doi: 10.1002/ajmg.10092.

Abstract

We report two patients with clinical and radiological findings of achondroplasia, who had the most common FGFR3 mutation occurring in thanatophoric dysplasia type I and hypochondroplasia, respectively. Thanatophoric dysplasia is usually a lethal condition, but the patient carrying this mutation is alive and presents a medical history similar to that of patients with achondroplasia. The events leading to such a discrepancy between genotype and phenotype are unclear. These rare cases may influence an appropriate medical and genetic counseling.

Publication types

  • Case Reports

MeSH terms

  • Achondroplasia / genetics*
  • Achondroplasia / pathology
  • Base Sequence
  • DNA / chemistry
  • DNA / genetics
  • DNA Mutational Analysis
  • Humans
  • Infant, Newborn
  • Male
  • Mutation, Missense
  • Phenotype
  • Protein-Tyrosine Kinases*
  • Receptor, Fibroblast Growth Factor, Type 3
  • Receptors, Fibroblast Growth Factor / genetics*
  • Thanatophoric Dysplasia / genetics*
  • Thanatophoric Dysplasia / pathology

Substances

  • Receptors, Fibroblast Growth Factor
  • DNA
  • FGFR3 protein, human
  • Protein-Tyrosine Kinases
  • Receptor, Fibroblast Growth Factor, Type 3