Homozygous N540K hypochondroplasia--first report: radiological and clinical features

Am J Med Genet A. 2014 Jul;164A(7):1784-8. doi: 10.1002/ajmg.a.36504. Epub 2014 Apr 8.

Abstract

We describe a 16-month-old male with N540K homozygous mutation in the FGFR3 gene who showed a more severe phenotype than hypochondroplasia (HCH). To our knowledge, a homozygous state for this mutation causing HCH has not been reported before. The clinical and radiological characteristics of our patient represent an intermediate condition between achondroplasia and achondroplasia/hypochondroplasia compound heterozygosity. This case represents a new expression of FGFR3 spectrum and it is of considerable importance for the genetic counseling in cases where both parents are affected with HCH.

Keywords: achondroplasia; homozygosity; hypochondroplasia; phenotypic overlap.

Publication types

  • Case Reports

MeSH terms

  • Bone and Bones / abnormalities*
  • Bone and Bones / diagnostic imaging
  • Bone and Bones / pathology
  • Brain / pathology
  • Dwarfism / diagnosis*
  • Dwarfism / genetics*
  • Facies
  • Genetic Association Studies*
  • Heterozygote
  • Homozygote*
  • Humans
  • Infant
  • Limb Deformities, Congenital / diagnosis*
  • Limb Deformities, Congenital / genetics*
  • Lordosis / diagnosis*
  • Lordosis / genetics*
  • Magnetic Resonance Imaging
  • Male
  • Mutation*
  • Phenotype*
  • Radiography
  • Receptor, Fibroblast Growth Factor, Type 3 / genetics*

Substances

  • Receptor, Fibroblast Growth Factor, Type 3

Supplementary concepts

  • Hypochondroplasia