2015 Fiscal Year Final Research Report
Elucidation of pathogenic mechanism of arthrogryposis multiplex congenita by Ctdnep1 gene deficiency
| Project/Area Number |
25462287
|
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| Research Category |
Grant-in-Aid for Scientific Research (C)
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| Allocation Type | Multi-year Fund |
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| Section | 一般 |
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| Research Field |
Orthopaedic surgery
|
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| Research Institution | University of Tsukuba (2015)
Tokyo Medical and Dental University (2013) |
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Principal Investigator |
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| Project Period (FY) |
2013-04-01 – 2016-03-31
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| Keywords | BMP / TGF-β / 筋肉 / 軟骨 |
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| Outline of Final Research Achievements |
In order to investigate the role of Ctdnep1 for musculoskeletal and joint development, we generated limb- and sternum-specific knockout mice (Ctdnep1 cKO). Ctdnep1 cKO mice showed joint contracture and impaired walking. Histological analysis showed that joint structure was formed normally, but development of muscle fiber was severely affected and chondrocytes of proximal femur proliferated abnormally. Cell culture experiments showed that TGF-β signaling was increased and BMP signaling was decreased in the absence of Ctdnep1. These results suggest that Ctdnep1 maintain BMP signaling by suppressing TGF-β signaling in muscle system and plays crucial role in muscle development.
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| Free Research Field |
骨・軟骨代謝学
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