2013 Fiscal Year Final Research Report
The control of inner ear morphogenesis by FGF genes in mouse models of 22q11.2 deletion syndrome.
| Project/Area Number |
23592499
|
|---|
| Research Category |
Grant-in-Aid for Scientific Research (C)
|
| Allocation Type | Multi-year Fund |
|---|
| Section | 一般 |
|---|
| Research Field |
Otorhinolaryngology
|
|---|
| Research Institution | Jikei University School of Medicine |
|---|
Principal Investigator |
|
|---|
| Co-Investigator(Kenkyū-buntansha) |
OKABE Masataka 東京慈恵会医科大学, 医学部, 教授 (10300716)
TATSUMI Norifumi 東京慈恵会医科大学, 医学部, 助教 (60514528)
UDAGAWA Tomokatsu 東京慈恵会医科大学, 医学部, 助教 (60328292)
|
|---|
| Project Period (FY) |
2011 – 2013
|
| Keywords | 内耳発生 / FGF / 22q11.2欠失症候群 |
|---|
| Research Abstract |
Multiple signaling molecules are involved in development of the ear. Fibroblast growth factor (Fgf) is known to be indispensable for controlling cell fate determination and differentiation during otic development. Here, we report that Spry1 and Spry2 compound mutant embryos , show an abnormal phenotype in the ventral region of the inner ear, including the cochlea. We found neuralization of the otic vesicle is induced in Spry1,2 knockout mouse. Moreover, Tbx1 prevents neural differentiation in the otic vesicle, and we observed a severe defective inner ear phenotype, coupled with strong induction of neural maker genes, in the Spry1,2 knockout ;Tbx1 hetero mouse. Our results suggest excess Fgf signaling inhibits Shh signaling, thereby inhibiting Tbx1 activation to prevent neural differentiation in the otic vesicle.
|
