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| Distinct Defects in Spine Formation or Pruning in Two Gene Duplication Mouse Models of Autism | |
| 2017-04 | |
| Source Publication | NEUROSCIENCE BULLETIN
 |
| ISSN | 1673-7067 |
| Volume | 33Issue:2Pages:143-152 |
| Status | 已发表 |
| DOI | 10.1007/s12264-017-0111-8 |
| Abstract | Autism spectrum disorder (ASD) encompasses a complex set of developmental neurological disorders, characterized by deficits in social communication and excessive repetitive behaviors. In recent years, ASD is increasingly being considered as a disease of the synapse. One main type of genetic aberration leading to ASD is gene duplication, and several mouse models have been generated mimicking these mutations. Here, we studied the effects of MECP2 duplication and human chromosome 15q11-13 duplication on synaptic development and neural circuit wiring in the mouse sensory cortices. We showed that mice carrying MECP2 duplication had specific defects in spine pruning, while the 15q11-13 duplication mouse model had impaired spine formation. Our results demonstrate that spine pathology varies significantly between autism models and that distinct aspects of neural circuit development may be targeted in different ASD mutations. Our results further underscore the importance of gene dosage in normal development and function of the brain. |
| Keyword | Autism Autism spectrum disorder Spine Spine formation Spinogenesis Spine pruning Gene duplication MECP2 15q11-13 duplication |
| Indexed By | SCI |
| Language | 英语 |
| Funding Project | Science and Technology Commission of Shanghai Municipality, China[16XD1404800] |
| WOS Research Area | Neurosciences & Neurology |
| WOS Subject | Neurosciences |
| WOS ID | WOS:000398725200003 |
| Publisher | SPRINGER |
| WOS Keyword | COPY-NUMBER VARIATION ; DOWN-SYNDROME ; DENDRITIC SPINES ; SPECTRUM DISORDERS ; CEREBRAL-CORTEX ; FRAGILE-X ; BEHAVIORAL ABNORMALITIES ; DIVERSE REGIONS ; RETT-SYNDROME ; MECP2 |
| Original Document Type | Article |
| Citation statistics | |
| Document Type | 期刊论文 |
| Identifier | https://kms.shanghaitech.edu.cn/handle/2MSLDSTB/1444 |
| Collection | 生命科学与技术学院_特聘教授组_于翔组 |
| Corresponding Author | Xu, Xiu; Yu, Xiang; Bian, Wen-Jie |
| Affiliation | 1.Chinese Acad Sci, Inst Neurosci, State Key Lab Neurosci, CAS Ctr Excellence Brain Sci & Intelligence Techn, Shanghai 200031, Peoples R China 2.Univ Chinese Acad Sci, Beijing 100049, Peoples R China 3.Fudan Univ, Dept Child Healthcare, Childrens Hosp, Shanghai 201102, Peoples R China 4.RIKEN Brain Sci Inst, Wako, Saitama 3510198, Japan 5.ShanghaiTech Univ, Sch Life Sci & Technol, Shanghai 201210, Peoples R China |
| Corresponding Author Affilication | School of Life Science and Technology |
| Recommended Citation GB/T 7714 | Wang, Miao,Li, Huiping,Takumi, Toru,et al. Distinct Defects in Spine Formation or Pruning in Two Gene Duplication Mouse Models of Autism[J]. NEUROSCIENCE BULLETIN,2017,33(2):143-152. |
| APA | Wang, Miao.,Li, Huiping.,Takumi, Toru.,Qiu, Zilong.,Xu, Xiu.,...&Bian, Wen-Jie.(2017).Distinct Defects in Spine Formation or Pruning in Two Gene Duplication Mouse Models of Autism.NEUROSCIENCE BULLETIN,33(2),143-152. |
| MLA | Wang, Miao,et al."Distinct Defects in Spine Formation or Pruning in Two Gene Duplication Mouse Models of Autism".NEUROSCIENCE BULLETIN 33.2(2017):143-152. |
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