Embryonic and not maternal trisomy causes developmental attenuation in the Ts65Dn mouse model for Down syndrome
Article first published online: 9 APR 2010
Copyright © 2010 Wiley-Liss, Inc.
Volume 239, Issue 6, pages 1645–1653, June 2010
How to Cite
Blazek, J. D., Billingsley, C. N., Newbauer, A. and Roper, R. J. (2010), Embryonic and not maternal trisomy causes developmental attenuation in the Ts65Dn mouse model for Down syndrome. Dev. Dyn., 239: 1645–1653. doi: 10.1002/dvdy.22295
- Issue published online: 17 MAY 2010
- Article first published online: 9 APR 2010
- Manuscript Accepted: 11 MAR 2010
- Down syndrome;
- mouse models;
- embryonic development;
- developmental delay
Trisomy 21 results in Down syndrome (DS) and causes phenotypes that may result from alterations of developmental processes. The Ts65Dn mouse is the most widely used genetic and phenotypic model for DS. We used over 1,500 offspring from Ts65Dn and two nontrisomic genetically similar control strains to investigate the influence of trisomy on developmental alterations and number of offspring. For the first time, we demonstrate gross developmental attenuation of Ts65Dn trisomic offspring at embryonic day (E) 9.5 and E13.5 and show that the major determinant of the developmental changes is segmental trisomy of the embryo and not the trisomic maternal uterine environment. Maternal alleles of nontrisomic genes linked to Pde6b may also influence the development of Ts65Dn offspring. Both developmental attenuation and the contribution of trisomic and nontrisomic genes are important components in the genesis of DS phenotypes. Developmental Dynamics 239:1645–1653, 2010. © 2010 Wiley-Liss, Inc.