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Functional analyses of heparan sulfate degradation using Drosophila as a model for mucopolysaccharidosis

Research Project

Project/Area Number 26840041
Research Category

Grant-in-Aid for Young Scientists (B)

Allocation TypeMulti-year Fund
Research Field Functional biochemistry
Research InstitutionTokyo Metropolitan Institute of Medical Science

Principal Investigator

KAMIMURA Keisuke  公益財団法人東京都医学総合研究所, 脳発達・神経再生研究分野, 主席研究員 (30529524)

Project Period (FY) 2014-04-01 – 2018-03-31
Project Status Completed (Fiscal Year 2017)
Budget Amount *help
¥4,030,000 (Direct Cost: ¥3,100,000、Indirect Cost: ¥930,000)
Fiscal Year 2016: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
Fiscal Year 2015: ¥1,040,000 (Direct Cost: ¥800,000、Indirect Cost: ¥240,000)
Fiscal Year 2014: ¥1,300,000 (Direct Cost: ¥1,000,000、Indirect Cost: ¥300,000)
Keywordsヘパラン硫酸 / プロテオグリカン / ムコ多糖代謝異常症 / ショウジョウバエ
Outline of Final Research Achievements

Heparan sulfate proteoglycans (HSPGs) are glycoproteins composed of a core protein with covalently attached heparan sulfate (HS) chains. HSPGs mediate interactions with a variety of extracellular ligands and participate in many physiological processes. In human, mutations in the lysosomal enzymes that degrade HS result in mucopolysaccharidosis, cause various organ dysfunction. Here, we examined the role of HS degradation during development using Drosophila melanogaster as a model system. We found that mutations in iduronidase, encoding a HS degradation enzyme, reduce the activity of BMP signaling and affect wing development.

Report

(5 results)
  • 2017 Annual Research Report   Final Research Report ( PDF )
  • 2016 Research-status Report
  • 2015 Research-status Report
  • 2014 Research-status Report
  • Research Products

    (17 results)

All 2017 2016 2015 2014 Other

All Journal Article (2 results) (of which Int'l Joint Research: 1 results,  Peer Reviewed: 2 results,  Open Access: 1 results,  Acknowledgement Compliant: 1 results) Presentation (12 results) (of which Int'l Joint Research: 4 results,  Invited: 3 results) Book (1 results) Remarks (2 results)

  • [Journal Article] Heparan sulfate proteoglycans in Drosophila neuromuscular development2017

    • Author(s)
      Kamimura Keisuke、Maeda Nobuaki
    • Journal Title

      Biochimica et Biophysica Acta (BBA) - General Subjects

      Volume: 1861 Issue: 10 Pages: 2442-2446

    • DOI

      10.1016/j.bbagen.2017.06.015

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed
  • [Journal Article] The Strip-Hippo Pathway Regulates Synaptic Terminal Formation by Modulating Actin Organization at the Drosophila Neuromuscular Synapses.2016

    • Author(s)
      Sakuma C, Saito Y, Umehara T, Kamimura K, Maeda N, Mosca TJ, Miura M, Chihara T.
    • Journal Title

      Cell Rep

      Volume: 16 Issue: 9 Pages: 2289-2297

    • DOI

      10.1016/j.celrep.2016.07.066

    • Related Report
      2016 Research-status Report
    • Peer Reviewed / Open Access / Int'l Joint Research / Acknowledgement Compliant
  • [Presentation] Glypican regulates synaptic plasticity at the Drosophila neuromuscular junction2017

    • Author(s)
      Keisuke Kamimura
    • Organizer
      Internatinal Conference on the Glycobiology of Nervous System
    • Related Report
      2017 Annual Research Report
    • Int'l Joint Research / Invited
  • [Presentation] Glypican regulates synaptic plasticity at the Drosophila neuromuscular junction2017

    • Author(s)
      Keisuke Kamimura, Aiko Odajima, Nobuaki Maeda
    • Organizer
      7 Lakes Proteoglycans Conference
    • Related Report
      2017 Annual Research Report
    • Int'l Joint Research
  • [Presentation] ショウジョウバエの神経筋接合部においてグリピカンはオクトパミンを介したシナプス可塑性を調節する2017

    • Author(s)
      神村圭亮、小田嶋愛子、前田信明
    • Organizer
      ConBio2017
    • Related Report
      2017 Annual Research Report
  • [Presentation] ショウジョウバエの神経筋接合部においてグリピカンはシナプス可塑性を調節する2016

    • Author(s)
      神村圭亮、小田嶋愛子、前田信明
    • Organizer
      第89回日本生化学会大会
    • Place of Presentation
      宮城県仙台市、仙台国際センター
    • Year and Date
      2016-09-25
    • Related Report
      2016 Research-status Report
  • [Presentation] ショウジョウバエの神経筋接合部においてグリピカンはシナプス可塑性を調節する2016

    • Author(s)
      神村圭亮、小田嶋愛子、前田信明
    • Organizer
      第35回日本糖質学会年会
    • Place of Presentation
      高知県高知市、高知市文化プラザかるぽーと
    • Year and Date
      2016-09-01
    • Related Report
      2016 Research-status Report
  • [Presentation] Glypican regulates synaptic plasticity at the Drosophila neuromuscular junction.2016

    • Author(s)
      Keisuke Kamimura, Aiko Odajima, Nobuaki Maeda.
    • Organizer
      Gordon Research Conference “Proteoglycan”
    • Place of Presentation
      Andover, NH, USA
    • Year and Date
      2016-07-08
    • Related Report
      2016 Research-status Report
    • Int'l Joint Research
  • [Presentation] Drosophila glypican regulates octopamine-mediated synaptic plasticity.2016

    • Author(s)
      Keisuke Kamimura, Aiko Odajima, Nobuaki Maeda
    • Organizer
      The 3rd International Synposium on Glyco-Neuroscience
    • Place of Presentation
      ウェスティンホテル淡路 (兵庫県淡路市)
    • Year and Date
      2016-01-14
    • Related Report
      2015 Research-status Report
    • Int'l Joint Research
  • [Presentation] ショウジョウバエにおいてグリピカンはオクトパミンを介した神経筋シナプスの可塑的な形態変化を調節する2015

    • Author(s)
      神村圭亮、小田嶋愛子、前田信明.
    • Organizer
      第88回日本生化学会大会
    • Place of Presentation
      神戸ポートアイランド (兵庫県神戸市)
    • Year and Date
      2015-12-01
    • Related Report
      2015 Research-status Report
  • [Presentation] ショウジョウバエの神経筋接合部におけるヘパラン硫酸プロテオグリカンの役割2015

    • Author(s)
      神村圭亮、前田信明
    • Organizer
      第34回日本糖質学会
    • Place of Presentation
      東京大学 (東京都文京区)
    • Year and Date
      2015-07-31
    • Related Report
      2015 Research-status Report
    • Invited
  • [Presentation] Heparan sulfate proteoglycans at the Drosophila neuromuscular junction.2015

    • Author(s)
      Keisuke Kamimura, Nobuaki Maeda
    • Organizer
      第38回日本神経科学大会
    • Place of Presentation
      神戸コンベンションセンター (兵庫県神戸市)
    • Year and Date
      2015-07-28
    • Related Report
      2015 Research-status Report
    • Invited
  • [Presentation] Glypican and the heparan sulfate fine structure proteoglycans at the Drosophila neuromuscular junction2014

    • Author(s)
      Keisuke Kamimura, Rie Hamada, Nobuaki Maeda
    • Organizer
      Joint Meeting of the Society for Glycobiology and the Japanese Society of Carbohydrate Research
    • Place of Presentation
      Hilton Hawaiian Village, Honolulu, Hawaii, USA
    • Year and Date
      2014-11-16 – 2014-11-19
    • Related Report
      2014 Research-status Report
  • [Presentation] ショウジョウバエの神経筋接合部におけるグリピカンとヘパラン硫酸微細構造の役割2014

    • Author(s)
      神村圭亮、濵田理絵、前田信明
    • Organizer
      第87回日本生化学会大会
    • Place of Presentation
      京都府京都市国立京都国際会館
    • Year and Date
      2014-10-15 – 2014-10-18
    • Related Report
      2014 Research-status Report
  • [Book] Glycoscience: Biology and Medicine2015

    • Author(s)
      Keisuke Kamimura, Nobuaki Maeda (Naoyuki Taniguchi et al (Eds.))
    • Total Pages
      1569
    • Publisher
      Springer, New York
    • Related Report
      2014 Research-status Report
  • [Remarks] 東京都医学総合研究所ホームページ

    • URL

      http://www.igakuken.or.jp/project/detail/regeneration.html

    • Related Report
      2017 Annual Research Report 2016 Research-status Report
  • [Remarks] 神経回路形成プロジェクトホームページ

    • URL

      http://www.igakuken.or.jp/regeneration/

    • Related Report
      2015 Research-status Report

URL: 

Published: 2014-04-04   Modified: 2019-03-29  

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