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The control of inner ear morphogenesis by FGF genes in mouse models of 22q11.2 deletion syndrome.

Research Project

Project/Area Number 23592499
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Research Field Otorhinolaryngology
Research InstitutionJikei University School of Medicine

Principal Investigator

YUICHIRO Yaguchi  東京慈恵会医科大学, 医学部, 講師 (30307475)

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
Project Status Completed (Fiscal Year 2013)
Budget Amount *help
¥4,940,000 (Direct Cost: ¥3,800,000、Indirect Cost: ¥1,140,000)
Fiscal Year 2013: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000)
Fiscal Year 2012: ¥1,560,000 (Direct Cost: ¥1,200,000、Indirect Cost: ¥360,000)
Fiscal Year 2011: ¥1,950,000 (Direct Cost: ¥1,500,000、Indirect Cost: ¥450,000)
Keywords内耳発生 / FGF / 22q11.2欠失症候群 / Sprouty / Tbx1 / Spouty
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.

Report

(4 results)
  • 2013 Annual Research Report   Final Research Report ( PDF )
  • 2012 Research-status Report
  • 2011 Research-status Report
  • Research Products

    (3 results)

All 2013 2011

All Presentation (3 results)

  • [Presentation] 組織切片からの三次元再構築から見る内耳発生機構の解析2013

    • Author(s)
      広川恵里沙.辰巳徳史.谷口雄一郎.岡部正隆
    • Organizer
      日本耳科学会
    • Place of Presentation
      宮崎
    • Year and Date
      2013-11-24
    • Related Report
      2013 Annual Research Report 2013 Final Research Report
  • [Presentation] M.Albert Basson The control of inner ear morphogenesis by sprouty and Tbx1 genes in mouse model s of 22q11.2deletion syndrome2011

    • Author(s)
      Yuichiro Yaguchi, Tian yu, Katherine Shim, Bernice Morrow
    • Organizer
      28^<th> politzer society meeting
    • Place of Presentation
      Greece
    • Year and Date
      2011-09-28
    • Related Report
      2013 Final Research Report
  • [Presentation] The control of inner ear morphogenesis by Sprouty and Tbx1 genes in mouse models of 22q11.2 deletion syndrome2011

    • Author(s)
      谷口 雄一郎
    • Organizer
      28th politzer society meeting
    • Place of Presentation
      アテネ
    • Related Report
      2011 Research-status Report

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Published: 2011-08-05   Modified: 2019-07-29  

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