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Perinatal Natural History of the Ts1Cje Mouse Model of Down Syndrome: Growth Restriction, Early Mortality, Heart Defects, and Delayed Development

PloS one, 2016-12, Vol.11 (12), p.e0168009-e0168009 [Peer Reviewed Journal]

COPYRIGHT 2016 Public Library of Science ;COPYRIGHT 2016 Public Library of Science ;2016 Ferrés et al. This is an open access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License. ;2016 Ferrés et al 2016 Ferrés et al ;ISSN: 1932-6203 ;EISSN: 1932-6203 ;DOI: 10.1371/journal.pone.0168009 ;PMID: 27930746

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Title:
Perinatal Natural History of the Ts1Cje Mouse Model of Down Syndrome: Growth Restriction, Early Mortality, Heart Defects, and Delayed Development
Author: Ferrés, Millie A ; Bianchi, Diana W ; Siegel, Ashley E ; Bronson, Roderick T ; Huggins, Gordon S ; Guedj, Faycal
Herault, Yann
Subjects: Amniocentesis ; Analysis ; Animal cognition ; Animals ; Animals, Newborn ; Biology and Life Sciences ; Births ; Brain research ; Care and treatment ; Chromosomes ; Congenital diseases ; Cyclin-dependent kinase inhibitor p21 ; Defects ; Developmental Disabilities - etiology ; Developmental Disabilities - pathology ; Diagnosis ; Disease Models, Animal ; Down syndrome ; Down Syndrome - complications ; Down Syndrome - embryology ; Down Syndrome - mortality ; Down Syndrome - pathology ; Down's syndrome ; Embryonic development ; Embryos ; Female ; Females ; Fetuses ; Gender aspects ; Genotype ; Genotype & phenotype ; Genotypes ; Heart ; Heart Defects, Congenital - etiology ; Heart Defects, Congenital - pathology ; Heart diseases ; Hospitals ; Infant mortality ; Infants ; Laboratory animals ; Male ; Males ; Medicine and Health Sciences ; Mice ; Mortality ; Multiplex Polymerase Chain Reaction ; Neomycin ; Neonates ; Newborn babies ; Patient outcomes ; Pregnancy ; Proteins ; Research and Analysis Methods ; Risk factors ; Rodents ; Sex ; Sex differences ; Sex Distribution ; Statistical analysis ; Studies ; Ventricle ; Weaning
Is Part Of: PloS one, 2016-12, Vol.11 (12), p.e0168009-e0168009
Description: The Ts1Cje model of Down syndrome is of particular interest for perinatal studies because affected males are fertile. This permits affected pups to be carried in wild-type females, which is similar to human pregnancies. Here we describe the early natural history and growth profiles of Ts1Cje embryos and neonates and determine if heart defects are present in this strain. Pups were studied either on embryonic (E) day 15.5, or from postnatal (P) day 3 through weaning on P21. PCR amplification targeting the neomycin cassette (present in Ts1Cje) and Sry (present in males) was used to analyze pup genotypes and sex ratios. Body weights and lengths, as well as developmental milestones, were recorded in Ts1Cje mice and compared to their wild-type (WT) littermates. Histological evaluations were performed at E15.5 to investigate the presence or absence of heart defects. Pups were divided into two groups: Ts1Cje-I pups survived past weaning and Ts1Cje-II pups died at some point before P21. Ts1Cje mouse embryos showed expected Mendelian ratios (45.8%, n = 66 for Ts1Cje embryos; 54.2%, n = 78 for WT embryos). Histological analysis revealed the presence of ventricular septal defects (VSDs) in 21% of Ts1Cje E15.5 embryos. After weaning, only 28.2% of pups were Ts1Cje (185 Ts1Cje out of 656 total pups generated), with males predominating (male:female ratio of 1.4:1). Among the recovered dead pups (n = 207), Ts1Cje (63.3%, n = 131, p<0.01) genotype was found significantly more often than WT (36.7%, n = 76). Retrospective analysis of Ts1Cje-II (pre-weaning deceased) pups showed that they were growth restricted compared to Ts1Cje-I pups (post-weaning survivors). Growth restriction correlated with statistically significant delays in achieving several neonatal milestones between P3 and P21 compared to Ts1Cje-I (post-weaning survivors) neonates and WT littermates. Ts1Cje genotype is not associated with increased early in utero mortality. Cardiac defects, specifically VSDs, are part of the phenotype in this strain. There is increased neonatal mortality in Ts1Cje pups, with sex differences observed. Ts1Cje mice that died in the neonatal period were more likely to be growth restricted and delayed in achieving neonatal developmental milestones.
Publisher: United States: Public Library of Science
Language: English
Identifier: ISSN: 1932-6203
EISSN: 1932-6203
DOI: 10.1371/journal.pone.0168009
PMID: 27930746
Source: Public Library of Science (PLoS) Journals Open Access
GFMER Free Medical Journals
MEDLINE
PubMed Central
ProQuest Central
DOAJ Directory of Open Access Journals

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Perinatal Natural History of the Ts1Cje Mouse Model of Down Syndrome: Growth Restriction, Early Mortality, Heart Defects, and Delayed Development

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