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Hippocampal and visuospatial learning defects in mice with a deletion of frizzled 9, a gene in the williams syndrome deletion interval

Hippocampal and visuospatial learning defects in mice with a...

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中国神经科学学会第六届学术会议暨学会成立十周年庆祝大会

作者：赵春杰 Samuel J. Pleasure 东南大学遗传研究中心 UCSF San Francisco 94143

＜正＞Wnt signaling regulates hippocampal development but little is known about the functions of specific Wnt receptors in this structure. Frizzled 9 is selectively expressed in the hippocampus and is one of about 20 genes typically deleted in williams syndrome. Since williams syndrome is associated with severe visuospatial processing defects, we generated a targeted null allele for frizzled 9 to examine its role in hippocampal development. Frizzled 9-null mice had generally normal gross anatomical hippocampal organization but showed large increases in apoptotic cell death in the developing dentate gyrus. This increase in programmed cell death commenced with the onset of dentate gyrus development and persisted into the first postnatal week of life. There was also a perhaps compensatory increase in the number of dividing precursors in the dentate gyrus, which may have been a compensatory response to the increased cell death. These changes in the mutants resulted in a moderate decrease in the number of adult dentate granule cells in null mice and. an increase in the number of hilar mossy cells.

关键词： hippocampus wnt williams syndrome

来源： cnki会议评论

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williams综合征中国儿童的适应性行为(英文)

Williams综合征中国儿童的适应性行为(英文)

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2014年浙江省医学会儿科学分会学术年会暨儿内科疾病诊治新进展国家级继续教育学习班

作者：季钗 Dan Yao Weijun Chen Mingyan Li Zhengyan Zhao Department of child health care The Children’s Hospital of Zhejiang University School of Medicine

Background:williams syndrome(WS) is a neurodevelopmental disease characterized by compelling psychological *** symptoms span multiple cognitive domains and include a distinctive pattern of social *** goal of this study was to explore adaptive behavior in WS patients in ***:We conducted a structured interview including the Infants-Junior Middle School Students Sociallife Abilities Scale in three participant groups:children with WS(n=26),normally-developing children matched for mental age(MA,n=30),and normally-developing children matched for chronological age(CA,n=40).We compared the mean scores for each domain between the three ***:Children with WS had more siblings than children in the two control *** educational level of the caregivers of WS children was lower than that of the control *** found no differences in locomotion,work skill,socialization,or self-management between the WS and MA *** children obtained higher scores of self-dependence(df=54,Z =-2.379,p=0.017) and had better communication skills(df=54,Z =-2.222,p = 0.026) compared with MA *** CA children achieved higher scores than the WS children for all dimensions of adaptive ***:WS children have better adaptive behavior skills regarding communication and self-dependence than normal children matched for mental *** intervention techniques should be designed to promote social development in this population.

关键词： williams syndrome children adaptive behavior

来源： cnki会议评论

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