Molecular characterization of a ring X chromosome in a male with short stature

Hum Genet. 2002 Apr;110(4):322-6. doi: 10.1007/s00439-002-0685-7. Epub 2002 Mar 2.

Abstract

We report the molecular characterization of a ring X chromosome that was transmitted from a mother to a male who has short stature and minor dysmorphic features. This represents only the second reported ring X chromosome in a male. The ring is derived from breakage within the Xp pseudoautosomal region (PAR) and just proximal to the Xq PAR. The total amount of deleted material is 700-900 kb DNA and includes six known transcribed genes. Interestingly, SHOX, a gene implicated in short stature, is not deleted from the ring chromosome. Possible pathogenetic explanations for the patient's clinical features include insufficient dosage of deleted genes, a position effect on SHOX expression, and cell death during development because of ring chromosome nondisjunction. The findings are also relevant to observations made of "complete" ring chromosomes.

Publication types

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

MeSH terms

  • Body Height / genetics*
  • Child, Preschool
  • Homeodomain Proteins / genetics
  • Humans
  • Male
  • Ring Chromosomes*
  • Short Stature Homeobox Protein
  • X Chromosome*

Substances

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