- THIS ARTICLE
- Full Text (Rapid PDF)
-
All Versions of this Article:
genetics.105.050898v1
172/1/437 most recent - Alert me when this article is cited
- Alert me if a correction is posted
- SERVICES
- Similar articles in this journal
- Similar articles in PubMed
- Alert me to new issues of the journal
- Download to citation manager
- Reprints & Permissions
- CITING ARTICLES
- Citing Articles via HighWire
- Citing Articles via Google Scholar
- GOOGLE SCHOLAR
- Articles by Roper, R. J.
- Articles by Reeves, R. H.
- Search for Related Content
- PUBMED
- PubMed Citation
- Articles by Roper, R. J.
- Articles by Reeves, R. H.
doi:10.1534/genetics.105.050898
A more recent version of this article appeared on January 1, 2006.
REGULAR RESEARCH PAPERS |
Perinatal Loss of TS65DN "Down Syndrome" Mice
Randall J. Roper 1, Heidi K. St. John 1, Jessica Philip 1, Ann Lawler 1 and Roger H. Reeves 2*
1 Johns Hopkins University School of Medicine
2 The Johns Hopkins University
* To whom correspondence should be addressed. E-mail: rreeves{at}jhmi.edu.
Submitted on September 8, 2005
Revised on September 13, 2005
Accepted on 13 September 2005
Ts65Dn mice inherit a marker chromosome, T(1716)65Dn, producing segmental trisomy for orthologs of about half of the genes on human chromosome 21. These mice display a number of phenotypes that are directly comparable to those in humans with trisomy 21, and are the most widely used animal model of Down syndrome (DS). However, the husbandry of Ts65Dn mice is complicated. Males are sterile, and only 20-40% of the offspring of Ts65Dn mothers are trisomic at weaning. The lower than expected frequency of trisomic offspring has been attributed to losses at meiosis, during gestation and at postnatal stages, but no systematic studies support any of these suppositions. We show that the T(1716)65Dn marker chromosome is inherited at expected frequency and is fully compatible with development to mid-gestation. Disproportional loss of trisomic offspring occurs in late gestation and continues through birth to weaning. Different maternal H2 haplotypes are significantly associated with the frequency of trisomy at weaning in patterns different from those reported previously. The proportion of trisomic mice per litter decreases with age of the Ts65Dn mother. These results provide the first statistical and numerical evidence supporting the prenatal and perinatal pattern of loss in the Ts65Dn mouse model of DS.
Key Words: Down syndrome, H2, animal models, non-Mendelian inheritance, perinatal loss
This article has been cited by other articles:
 |
|
 |
|
  L. E. Olson, R. J. Roper, C. L. Sengstaken, E. A. Peterson, V. Aquino, Z. Galdzicki, R. Siarey, M. Pletnikov, T. H. Moran, and R. H. Reeves Trisomy for the Down syndrome 'critical region' is necessary but not sufficient for brain phenotypes of trisomic mice Hum. Mol. Genet., April 1, 2007; 16(7): 774 - 782. [Abstract] [Full Text] [PDF]
|
|