The jumping SHOX gene--crossover in the pseudoautosomal region resulting in unusual inheritance of Leri-Weill dyschondrosteosis

J Clin Endocrinol Metab. 2011 Feb;96(2):E356-9. doi: 10.1210/jc.2010-1505. Epub 2010 Nov 10.

Abstract

Context: During meiosis I, the recombination frequency in the pseudoautosomal region on Xp and Yp (PAR1) in males is very high. As a result, mutated genes located within the PAR1 region can be transferred from the Y-chromosome to the X-chromosome and vice versa.

Patients: Here we describe three families with SHOX abnormalities resulting in Leri-Weill dyschondrosteosis or Langer mesomelic dysplasia.

Results: In about half of the segregations investigated, a transfer of the SHOX abnormality to the alternate sex chromosome was demonstrated.

Conclusions: Patients with an abnormality of the SHOX gene should receive genetic counseling as to the likelihood that they may transmit the mutation or deletion to a son as well as to a daughter.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Arm / pathology
  • Body Height / physiology
  • Child
  • Chromosome Disorders / genetics*
  • Chromosome Disorders / pathology
  • Chromosomes, Human, X / genetics
  • Chromosomes, Human, Y / genetics
  • Exons / genetics
  • Family
  • Female
  • Growth Disorders / genetics
  • Growth Disorders / pathology
  • Hand / diagnostic imaging
  • Hand / pathology
  • Homeodomain Proteins / genetics*
  • Humans
  • In Situ Hybridization, Fluorescence
  • Interspersed Repetitive Sequences / genetics*
  • Karyotyping
  • Male
  • Nucleic Acid Amplification Techniques
  • Osteochondrodysplasias / genetics
  • Osteochondrodysplasias / pathology
  • Pedigree
  • Pregnancy
  • Radiography
  • Short Stature Homeobox Protein
  • Wrist / diagnostic imaging
  • Wrist / pathology

Substances

  • Homeodomain Proteins
  • SHOX protein, human
  • Short Stature Homeobox Protein

Supplementary concepts

  • Langer mesomelic dysplasia
  • Leri-Weil syndrome