Understanding mental retardation in Down's syndrome using trisomy 16 mouse models

被引:68
作者
Galdzicki, Z [1 ]
Siarey, RJ [1 ]
机构
[1] Uniformed Serv Univ Hlth Sci, Dept Anat Physiol & Genet, Program Neurosci, F Edward Hebert Sch Med, Bethesda, MD 20814 USA
关键词
Down's syndrome; mental retardation; mouse; neurodevelopment; synaptic plasticity; trisomy; 16; 21; Ts1Cje; Ts65Dn;
D O I
10.1034/j.1601-183X.2003.00024.x
中图分类号
B84 [心理学]; C [社会科学总论]; Q98 [人类学];
学科分类号
03 ; 0303 ; 030303 ; 04 ; 0402 ;
摘要
Mental retardation in Down's syndrome, human trisomy 21, is characterized by developmental delays, language and memory deficits and other cognitive abnormalities. Neurophysiological and functional information is needed to understand the mechanisms of mental retardation in Down's syndrome. The trisomy mouse models provide windows into the molecular and developmental effects associated with abnormal chromosome numbers. The distal segment of mouse chromosome 16 is homologous to nearly the entire long arm of human chromosome 21. Therefore, mice with full or segmental trisomy 16 (Ts65Dn) are considered reliable animal models of Down's syndrome. Ts65Dn mice demonstrate impaired learning in spatial tests and abnormalities in hippocampal synaptic plasticity. We hypothesize that the physiological impairments in the Ts65Dn mouse hippocampus can model the suboptimal brain function occuring at various levels of Down's syndrome brain hierarchy, starting at a single neuron, and then affecting simple and complex neuronal networks. Once these elements create the gross brain structure, their dysfunctional activity cannot be overcome by extensive plasticity and redundancy, and therefore, at the end of the maturation period the mind inside this brain remains deficient and delayed in its capabilities. The complicated interactions that govern this aberrant developmental process cannot be rescued through existing compensatory mechanisms. In summary, overexpression of genes from chromosome 21 shifts biological homeostasis in the Down's syndrome brain to a new less functional state.
引用
收藏
页码:167 / 178
页数:12
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