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

Autor: Faycal Guedj, Diana W. Bianchi, Roderick T. Bronson, Ashley E. Siegel, Millie A. Ferres, Gordon S. Huggins
Rok vydání: 2016
Předmět:
Male
0301 basic medicine
Embryology
Physiology
Developmental Disabilities
lcsh:Medicine
Pediatrics
Chromosomal Disorders
Mice
Child Development
0302 clinical medicine
Pregnancy
Genotype
Medicine and Health Sciences
Morphogenesis
lcsh:Science
Multidisciplinary
Heart
Embryo
Animal Models
Neomycin
Congenital Heart Defects
Testis determining factor
Physiological Parameters
In utero
Female
Anatomy
Research Article
medicine.drug
Heart Defects
Congenital
Down syndrome
medicine.medical_specialty
Child Growth
Cardiology
Mouse Models
Biology
Research and Analysis Methods
Andrology
03 medical and health sciences
Model Organisms
Internal medicine
Congenital Disorders
medicine
Animals
Weaning
Birth Defects
Sex Distribution
Clinical Genetics
Growth Restriction
lcsh:R
Embryos
Body Weight
Biology and Life Sciences
Neonates
medicine.disease
Disease Models
Animal
030104 developmental biology
Endocrinology
Animals
Newborn
Cardiovascular Anatomy
Ventricular Septal Defects
lcsh:Q
Down Syndrome
Multiplex Polymerase Chain Reaction
030217 neurology & neurosurgery
Developmental Biology
Zdroj: PLoS ONE
PLoS ONE, Vol 11, Iss 12, p e0168009 (2016)
ISSN: 1932-6203
Popis: Background 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. Methods 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. Results 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
Databáze: OpenAIRE
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