Studies on molecular mechanisms of mammalian early embryonic development by means of recessive lethal mutations.

• Project/Area Number: 18370094
• Research Category: Grant-in-Aid for Scientific Research (B)
• Allocation Type: Single-year Grants
• Section: 一般
• Research Field: Developmental biology
• Research Institution: Research Institute, Osaka Medical Center for Maternal and Child Health
• Principal Investigator: MATSUO Isao Research Institute, Osaka Medical Center for Maternal and Child Health, 大阪府立母子保健総合医療センター(研究所) (10264285)
• Project Period (FY): 2006 – 2009
• Project Status: Completed (Fiscal Year 2009)
• Budget Amount: ¥17,860,000 (Direct Cost: ¥15,400,000、Indirect Cost: ¥2,460,000); Fiscal Year 2009: ¥2,470,000 (Direct Cost: ¥1,900,000、Indirect Cost: ¥570,000); Fiscal Year 2008: ¥3,900,000 (Direct Cost: ¥3,000,000、Indirect Cost: ¥900,000); Fiscal Year 2007: ¥4,290,000 (Direct Cost: ¥3,300,000、Indirect Cost: ¥990,000); Fiscal Year 2006: ¥7,200,000 (Direct Cost: ¥7,200,000)
• Keywords: マウス / 劣性致死変異体 / ヘパラン硫酸鎖 / 発生 / プロテオグリカン / FGF / 遺伝学 / Ext2 / 胚体外外胚葉 / 劣性致死変異 / へパラン硫酸鎖

Research Abstract

We identified the transgene insertion allele of mouse Ext2, one of the causative genes of human multiple hereditaryexostoses, which catalyzes elongation of HS-chains. Marker expression analyses of Ext2-deficient embryos revealed that HS-chains are essential for response to Fibroblast growth factor (FGF) signaling and, in particular, local distribution of the FGF ligands as well as FGF signaling. Moreover, HS-chains expression attached with cell surface is distinctively specific to those areas in which FGF signaling is potentially active. Fur

Research Products

• [Journal Article] FGF signaling directs a center-to-pole expansion of tubulogenesis in mouse testis differentiation (2010)
  • Author(s): Ryuji Hiramatsu, Kyoko Harikae, Naoki Tsunekawa, Masamichi Kurohmaru, Isao Matsuo, Yoshiakira Kanai
  • Journal Title: Development 137 Pages: 303-312
  • Peer Reviewed
• [Journal Article] FGF signaling directs a center-to-pole expansion of tubulogenesis in mousetestis differentiation. (2010)
  • Author(s): Ryuji Hiramatsu, et al.
  • Journal Title: Development 137 Pages: 303-312
  • Peer Reviewed
• [Journal Article] Dosage-dependent hedgehog signals integrated with Wnt/β-catenin signaling regulate external genitalia formation as an appendicular program (2009)
  • Author(s): Shinichi Miyagawa, Anne Moon, Ryuma Haraguchi, Chie Inoue, Masayo Harada, Chiaki Nakahara, Kentaro Suzuki, Daisuke Matsumaru, Takehito Kaneko, Isao Matsuo, Lei Yang, Makoto M. Taketo, Taisen Iguchi, Sylvia M. Evans, Gen Yamada
  • Journal Title: Development 136 Pages: 3969-3978
  • NAID: 120002469485
  • Peer Reviewed
• [Journal Article] Dosage-dependent hedgehog signals integrated with Wnt/β-catenin signaling regulate external genitalia formation as an appendicular program. (2009)
  • Author(s): Shinichi Miyagawa, et al.
  • Journal Title: Development 136 Pages: 3969-3978
  • Peer Reviewed
• [Journal Article] Alain Prochiantz and Takao K Hensch Experience-dependent transfer of Otx2 homeoprotein into the visual cortex activates postnatal plasticity (2008)
  • Author(s): Sayaka Sugiyama, Ariel A Di Nardo, Shinichi Aizawa, Isao Matsuo, Michel Volovitch
  • Journal Title: Cell 134 Pages: 508-520
  • Peer Reviewed
• [Journal Article] Possible involvement of SINEs in mammalian-specific brain formation (2008)
  • Author(s): Takeshi Sasaki, Hidenori Nishihara, Mika Hirakawa, Koji Fujimura, Mikiko Tanaka, Nobuhiro Kokubo, Chiharu Kimura-Yoshida, Isao Matsuo, Kenta Sumiyama, Naruya Saitou, Tomomi Shimogori, Norihiro Okada
  • Journal Title: Proc. Natl. Acad. Sci. USA 105 Pages: 4220-4225
  • Peer Reviewed
• [Journal Article] Experience-dependent transfer of Otx2homeoprotein into the visual cortex activatespostnatal plasticity (2008)
  • Author(s): Sayaka Sugiyama, Ariel A Di Nardo, Shinichi Aizawa, Isao Matsuo, Michel Volovitch, Alain Prochiantz and Takao K Hensch
  • Journal Title: Cell 134 Pages: 508-520
  • Peer Reviewed
• [Journal Article] Possible involvement of SINEs inmammalian-specific brain formation (2008)
  • Author(s): Takeshi Sasaki, Hidenori Nishihara, Mika Hirakawa, Koji Fujimura, Mikiko Tanaka, Nobuhiro Kokubo, Chiharu Kimura-Yoshida, Isao Matsuo , Kenta Sumiyama, Naruya Saitou, Tomomi Shimogori and Norihiro Okada
  • Journal Title: Proc. Natl. Acad. Sci. USA 105 Pages: 4220-4225
  • Peer Reviewed
• [Journal Article] Experience-dependent transfer of Otx2 homeoprotein into the visual cortex activates postnatal plasticity (2008)
  • Author(s): Sayaka Sugiyama, et al.
  • Journal Title: Cell 134 Pages: 508-520
  • Peer Reviewed
• [Journal Article] Possible involvement of SINEs in mammalian-specific brain formation. (2008)
  • Author(s): Takeshi Sasaki, et. al.
  • Journal Title: Proc. Natl. Acad. Sci. USA 105 Pages: 4220-4225
  • Peer Reviewed
• [Journal Article] Functional roles of Otx2 transcription factor in postnatal mouse retinal development (2007)
  • Author(s): Chieko Koike, Akihiro Nishida, Shinji Ueno, Hiromitsu Saito, Rikako Sanuki, Shigeru Sato, Akiko Furukawa, Shinichi Aizawa, Isao Matsuo, Noboru Suzuki, Mineo Kondo, Takahisa Furukawa
  • Journal Title: Mol. Cell. Biol 27 Pages: 8318-8329
  • Peer Reviewed
• [Journal Article] Dkk3-Cre BAC transgenic mouse line: A tool for highly-efficient deletion in retinal progenitor cells (2007)
  • Author(s): Shigeru Sato, Tatsuya Inoue, Koji Terada, Isao Matsuo, Shinichi Aizawa, Yasuo Tano, Takashi Fujikado, Takahisa Furukawa
  • Journal Title: Genesis 45 Pages: 502-507
  • Peer Reviewed
• [Journal Article] Crucial roles of Foxa2 in mouse anterior-posterior axis polarization via regulation of anterior visceral endoderm-specific genes (2007)
  • Author(s): Chiharu Kimura-Yoshida, E Tian, Hiroshi Nakano, Saori Amazaki, Kayo Shimokawa, Janet Rossant, Shinichi Aizawa, Isao Matsuo
  • Journal Title: Proc. Natl. Acad. Sci. USA 104 Pages: 5919-5924
  • Peer Reviewed
• [Journal Article] Functional roles of Otx2 transcription factor inpostnatal mouse retinal development (2007)
  • Author(s): Chieko Koike, Akihiro Nishida, Shinji Ueno, Hiromitsu Saito, Rikako Sanuki, Shigeru Sato, Akiko Furukawa, Shinichi Aizawa, Isao Matsuo ,Noboru Suzuki, Mineo Kondo, and Takahisa Furukawa
  • Journal Title: Mol. Cell. Biol 27 Pages: 8318-8329
  • Peer Reviewed
• [Journal Article] Dkk3-Cre BAC transgenic mouse line : A tool forhighly-efficient deletion in retinal progenitorcells (2007)
  • Author(s): Shigeru Sato, Tatsuya Inoue, Koji Terada, Isao Matsuo, Shinichi Aizawa, Yasuo Tano, Takashi Fujikado and Takahisa Furukawa
  • Journal Title: Genesis 45 Pages: 502-507
  • Peer Reviewed
• [Journal Article] Crucial roles of Foxa2 in mouseanterior-posterior axis polarization viaregulation of anterior visceral endoderm-specificgenes (2007)
  • Author(s): Chiharu Kimura-Yoshida, E Tian, Hiroshi Nakano, Saori Amazaki, Kayo Shimokawa, Janet Rossant, Shinichi Aizawa and Isao Matsuo
  • Journal Title: Proc. Natl. Acad. Sci. USA 104 Pages: 5919-5924
  • Peer Reviewed
• [Journal Article] Dkk3-Cre BAC transgenic mouse line : A tool for highly-efficient deletion in retinal progenitor cells. (2007)
  • Author(s): Shigeru Sato, et. al.
  • Journal Title: Genesis 45 Pages: 502-507
  • Peer Reviewed
• [Journal Article] Functional roles of Otx2 transcription factor in postnatal mouse retinal development (2007)
  • Author(s): Chieko Koike, et. al.
  • Journal Title: Mol. Cell. Biol. 27 Pages: 8318-8329
  • Peer Reviewed
• [Journal Article] Crucial roles of Foxa2 in mouse anterior-posterior axis polarization via regulation of anterior visceral endoderm-specific genes (2007)
  • Author(s): Chiharu Kimura-Yoshida et al.
  • Journal Title: Proc.Natl.Acad.Sci.USA 104・13 Pages: 5919-5924
• [Presentation] Spatio-temporal expression of heparan sulfate glycosaminoglycan chains modulates distribution of Fgf signaling during mammalian embrvogenesis (2009)
  • Author(s): Kayo Shimokawa, et al.
  • Organizer: 16th International Society of Developmental Biologists Congress
  • Place of Presentation: Edinburgh, Scotland, UK
  • Year and Date: 2009-09-08
• [Presentation] Spatio-temporal expression of heparan sulfate glycosaminoglycan chains modulates distribution of Fgf signaling during mammalian embryogenesis (2009)
  • Author(s): Isao Matsuo
  • Organizer: 16th International Society of Developmental Biologists Congress
  • Place of Presentation: UK. Edinburgh
• [Presentation] Isao Matsuo Specific expression of heparan sulfate glycosaminoglycan chains is crucial for local distribution of FGF signaling in mouse extraembryonic ectoderm of the (2009)
  • Author(s): Isao Matsuo
  • Organizer: 第32回日本分子生物学会年会
  • Place of Presentation: 横浜
• [Presentation] Heparan sulfate chains synthesized by Ext2 regulate Fgf signaling during mammalian embryogenesis (2008)
  • Author(s): Kazuhiro Mukai, et al.
  • Organizer: Mouse Genetics & Genomics : Development and Disease
  • Place of Presentation: CSH, New York, USA
  • Year and Date: 2008-11-01
• [Presentation] Heparan sulfate chains synthesized by Ext2 regulate Fgf signaling during mammalian embryogenesis (2008)
  • Author(s): Isao Matsuo
  • Organizer: Mouse Genetics, Genomics: Development and Disease
  • Place of Presentation: Cold Spring Harbor Laboratory, USA
• [Presentation] Identification and characterization of a transgene insertion mutation of Ext2 gene encoding a glycosyltransferase. (2007)
  • Author(s): Kazuhiro Mukai, et. al.
  • Organizer: Mouse Molecular Genetics Meeting
  • Place of Presentation: Cambridge, UK
  • Year and Date: 2007-09-07
• [Presentation] Identification and characterization of a transgene insertion mutation of Ext2 gene encoding a glycosyltransferase (2007)
  • Author(s): Isao Matsuo
  • Organizer: Mouse Molecular Genetics Meeting
  • Place of Presentation: Wellcome Trust Genome Campus, Cambridge, UK
• [Book] Wntシグナルとマウス前後軸決定～Wnt拮抗因子Dickkopf1遺伝子に着目して～医学のあゆみ (2010)
  • Author(s): 吉田千春、平松竜司、松尾勲
  • Publisher: (印刷中)
• [Remarks]
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