Haploinsufficiency of mTR results in defects in telomere elongation

Proc Natl Acad Sci U S A. 2002 Mar 19;99(6):3591-6. doi: 10.1073/pnas.012549799.

Abstract

Telomeres are usually maintained about an equilibrium length, and the set point for this equilibrium differs between species and between strains of a given species. To examine the requirement for telomerase in mediating establishment of a new telomere length equilibrium, we generated interspecies crosses with telomerase mTR knockout mice. In crosses between C57BL/6J (B6) and either of two unrelated mouse species, CAST/Ei and SPRET/Ei, telomerase mediated establishment of a new telomere length equilibrium in wild-type mTR(+/+) mice. This new equilibrium was characterized by elongation of the short telomeres of CAST/Ei or SPRET/Ei origin. In contrast, mTR(-/-) offspring of interspecies crosses failed to elongate telomeres. Unexpectedly, haploinsufficiency was observed in mTR(+/-) heterozygous interspecies mice, which had an impaired ability to elongate short SPRET/Ei or CAST/Ei telomeres to the new equilibrium set point that was achieved in wild-type mTR(+/+) mice. These results demonstrate that elongation of telomeres to a new telomere set point requires telomerase and indicate that telomerase RNA may be limiting in vivo.

Publication types

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

MeSH terms

  • Animals
  • Crosses, Genetic
  • Female
  • Gene Deletion
  • Genotype
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Mice, Inbred Strains
  • Mice, Knockout
  • Molecular Sequence Data
  • Phenotype
  • RNA / genetics*
  • Species Specificity
  • Telomerase / genetics*
  • Telomerase / metabolism
  • Telomere / enzymology*
  • Telomere / genetics
  • Telomere / metabolism*

Substances

  • telomerase RNA
  • RNA
  • Telomerase

Associated data

  • GENBANK/AY058900
  • GENBANK/AY058901