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Ozono Keiichi

Research Project

Project/Area Number 24390264
Research Category

Grant-in-Aid for Scientific Research (B)

Allocation TypePartial Multi-year Fund
Section一般
Research Field Pediatrics
Research InstitutionOsaka University

Principal Investigator

OZONO Keiichi  大阪大学, 医学(系)研究科(研究院), 教授 (20270770)

Co-Investigator(Kenkyū-buntansha) MICHIGAMI Toshimi  大阪府立母子保健総合医療センター(研究所), 部長 (00301804)
Project Period (FY) 2012-04-01 – 2015-03-31
Project Status Completed (Fiscal Year 2014)
Budget Amount *help
¥17,810,000 (Direct Cost: ¥13,700,000、Indirect Cost: ¥4,110,000)
Fiscal Year 2014: ¥4,290,000 (Direct Cost: ¥3,300,000、Indirect Cost: ¥990,000)
Fiscal Year 2013: ¥4,680,000 (Direct Cost: ¥3,600,000、Indirect Cost: ¥1,080,000)
Fiscal Year 2012: ¥8,840,000 (Direct Cost: ¥6,800,000、Indirect Cost: ¥2,040,000)
Keywords成長障害 / 骨系統疾患 / C型Na利尿ペプチド / 成長軟骨 / cGMP / 創薬 / 骨粗鬆症
Outline of Final Research Achievements

We have established the disease entity associated with tall stature and long hallux due to the gain-of-function type mutations of NPR2.The disease is named as epiphyseal chondrodysplasia, Miura Type and filed (#615923)in OMIM (Online Mendelian Inheritance in Man). Transgenic mice with the collagen type XI promoter/enhancer-driven NPR2 mutation showed the overgrowth and deformity of long bones. The growth plate of the transgenic mice were elongated, especially hypertrophic zone, and gene expression is being analyzed in chondrocytes of the growth plate. We found that NT-pro CNP is a good marker of the height velocity in patients with short stature before and after the growth hormone therapy. Ten small compounds are identified by in silico as candidates of NPR2 activator and being analyzed by the generation of cGMP in cultured cells expressing NPR2.

Report

(4 results)
  • 2014 Annual Research Report   Final Research Report ( PDF )
  • 2013 Annual Research Report
  • 2012 Annual Research Report
  • Research Products

    (36 results)

All 2014 2013 2012 Other

All Journal Article (16 results) (of which Peer Reviewed: 12 results) Presentation (16 results) (of which Invited: 1 results) Book (3 results) Remarks (1 results)

  • [Journal Article] 小児における血清NT-proCNP値測定の意義-年齢,成長速度及びGH治療反応性-2014

    • Author(s)
      窪田拓生,三浦弘司,王薇,北岡太一,大幡泰久,山本景子,藤原誠,橘真紀子,三善陽子,難波範行,大薗恵一
    • Journal Title

      ホルモンと臨床

      Volume: 60 Pages: 41-43

    • Related Report
      2014 Annual Research Report
  • [Journal Article] Overgrowth syndrome associated with a gain-of-functionmutation of the natriuretic   peptide receptor 2 (NPR2) gene.2014

    • Author(s)
      Miura K, Kim OH, Lee HR, Namba N, Michigami T, Yoo WJ, Choi H, Ozono K, Cho TJ.
    • Journal Title

      Am J Med Genet A

      Volume: 164A Issue: 1 Pages: 156-163

    • DOI

      10.1002/ajmg.a.36218

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Serum fibroblast growth factor 23 is a useful marker to distinguish vitamin D-deficient  rickets from hypophosphatemic rickets.2014

    • Author(s)
      Kubota T
    • Journal Title

      Horm Res Paediatr Epub ahead of print

      Volume: - Pages: 220-226

    • Related Report
      2013 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Overgrowth syndrome associated with a gain-of-function mutation of the natriuretic  peptide receptor 2 (NPR2) gene.2014

    • Author(s)
      Miura K
    • Journal Title

      Am J Med Gene A

      Volume: 164A(1) Pages: 156-163

    • Related Report
      2013 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Elevated fibroblast growth factor 23 exerts its effects on placenta and regulates  vitamin D metabolism in pregnancy of Hyp mice.2014

    • Author(s)
      Ohata Y
    • Journal Title

      J Bone Miner Res Epub ahead of print

      Volume: -

    • Related Report
      2013 Annual Research Report
    • Peer Reviewed
  • [Journal Article] 遺伝性骨粗鬆症2013

    • Author(s)
      大薗恵一
    • Journal Title

      最新の骨粗鬆症学-骨粗鬆症の最新知見-

      Volume: 71  増刊号2 Pages: 583-588

    • Related Report
      2013 Annual Research Report
  • [Journal Article] 骨系統疾患の病態と治療2013

    • Author(s)
      三浦弘司
    • Journal Title

      CLINICAL CALCIUM

      Volume: 23(12) Pages: 97-102

    • Related Report
      2013 Annual Research Report
  • [Journal Article] A human skeletal overgrowth mutation maximal velocity and blocks desensitization of guanylyl cyclase-B2013

    • Author(s)
      Miura K
    • Journal Title

      Bone

      Volume: 56(2) Pages: 375-382

    • Related Report
      2013 Annual Research Report
  • [Journal Article] Weaver syndrome and EZH2 mutations:Clarifying the clinical phenotype.2013

    • Author(s)
      Ozono K
    • Journal Title

      Am J Med Gene A

      Volume: 161A(12) Pages: 2972-2980

    • Related Report
      2013 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Treatment of hypophosphatemic rickets with phosphate and active vitamin D in japan:A questionnaire-based survey.2013

    • Author(s)
      Fujiwara M
    • Journal Title

      Clin Pediatr Endocrinol

      Volume: 22(1) Pages: 9-14

    • Related Report
      2013 Annual Research Report
    • Peer Reviewed
  • [Journal Article] FGF23 suppresses chondrocyte proliferation in the presence of soluble α-Klotho both in vitro and in vivo.2013

    • Author(s)
      Kawai M, Kinoshita S, Kimoto A, Hasegawa Y, Miyagawa K, Yamazaki M, Ohata Y, Ozono K, Michigami T.
    • Journal Title

      J Biol Chem

      Volume: 288 Issue: 4 Pages: 2414-2427

    • DOI

      10.1074/jbc.m112.410043

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Treatment of Hypophosphatemic Rickets with Phosphate and Active Vitamin D in Japan: A Questionnaire-based Survey2013

    • Author(s)
      Fujiwara M
    • Journal Title

      Clinical Pediatric Endocrinology

      Volume: 22 Issue: 1 Pages: 9-14

    • DOI

      10.1297/cpe.22.9

    • NAID

      10031199647

    • ISSN
      0918-5739, 1347-7358
    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] An observational study of the effectiveness and safety of growth hormone (HumatropeR) treatment in Japanese children with growth hormone deficiency or Turner syndrome.2013

    • Author(s)
      Tai S
    • Journal Title

      Endocr J

      Volume: 60(1) Pages: 57-64

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] An overgrowth disorder associated with excessive production of cGMP due to a gain-of-function mutation of the natriuretic peptide receptor 2 gene.2012

    • Author(s)
      Miura K, Namba N, Fujiwara M, Ohata Y, Ishida H, Kitaoka T, Kubota T, Hirai H, Higuchi C, Tsumaki N, Yoshikawa H, Sakai N, Michigami T, Ozono K.
    • Journal Title

      PLoS One

      Volume: 7 Issue: 8 Pages: e42180-e42180

    • DOI

      10.1371/journal.pone.0042180

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Vinculin functions as regulator of chondrogenesis.2012

    • Author(s)
      Koshimizu T, Kawai M, Kondou H, Tachikawa K, Sakai N, Ozono K, Michigami T.
    • Journal Title

      J Biol Chem

      Volume: 287 Issue: 19 Pages: 15760-15775

    • DOI

      10.1074/jbc.m111.308072

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Pediatric aspects of skeletal dysplasia.2012

    • Author(s)
      Ozono K
    • Journal Title

      Pediatr Endocrinol Rev

      Volume: 10 Suppl 1 Pages: 35-43

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Presentation] Serum Levels of Amino-terminal Propepride of C-type Natriuretic Peptide may Predict Growth Response to Groeth Hormone Treatment in Patients with Achondroplasia /Hypochondroplasia.2014

    • Author(s)
      Kubota k,Miura K,Wang W,Yamamoto K,Fujiwara M,Ohata Y,Tachabana M,Kitaoka T,Miyoshi Y,Namba N,Ozono K
    • Organizer
      ASBMR2014
    • Place of Presentation
      アメリカ合衆国,ヒューストン
    • Year and Date
      2014-09-12 – 2014-09-15
    • Related Report
      2014 Annual Research Report
  • [Presentation] 低身長症例における血清NT-proCNP値測定の有用性-成長速度及びGH治療反応性-2014

    • Author(s)
      窪田拓生,三浦弘司,王薇,北岡太一,大幡泰久,山本景子,藤原誠,橘真紀子,三善陽子,難波範行,大薗恵一
    • Organizer
      第87回 日本内分泌学会学術総会
    • Place of Presentation
      福岡
    • Year and Date
      2014-04-24 – 2014-04-26
    • Related Report
      2014 Annual Research Report
  • [Presentation] 小児内分泌-骨ミネラル異常症を中心として-2014

    • Author(s)
      大薗恵一
    • Organizer
      第23回 臨床内分泌代謝Update
    • Place of Presentation
      名古屋国際会議場
    • Related Report
      2013 Annual Research Report
  • [Presentation] 骨芽細胞・骨細胞の制御と疾患2013

    • Author(s)
      大薗恵一
    • Organizer
      第57回 日本リウマチ学会総会・学術集会 第22回 国際リウマチシンポジウム
    • Place of Presentation
      国立京都国際会館
    • Related Report
      2013 Annual Research Report
  • [Presentation] 成長障害診療の現在と未来2013

    • Author(s)
      大薗恵一
    • Organizer
      第24回 日本小児科医会総会フォーラム
    • Place of Presentation
      大阪市中央公会堂
    • Related Report
      2013 Annual Research Report
  • [Presentation] 骨系統疾患の新たな治療法2012

    • Author(s)
      大薗恵一
    • Organizer
      第10回 日本胎児治療学会 第5回 胎児骨系統疾患フォーラム併催
    • Place of Presentation
      仙台市情報・産業プラザ(宮城県)(招待講演)
    • Year and Date
      2012-12-02
    • Related Report
      2012 Annual Research Report
  • [Presentation] An Overgrowth Disorder Associated with Excessive Production of cGMP Due to a Gain-of-Function Mutation of the Natriuretic Peptide Receptor 2 (NPR2) Gene.2012

    • Author(s)
      Miura K
    • Organizer
      7th Biennial Asia Pacific Paediatric Endocrine Society(APPES) Scientific Meeting
    • Place of Presentation
      Bali Nusa Dua Convention Centre (Bali, Indonesia)
    • Year and Date
      2012-11-16
    • Related Report
      2012 Annual Research Report
  • [Presentation] N540K変異を有する軟骨低形成症の臨床像および成長ホルモン治療の効果について(第2報)2012

    • Author(s)
      北岡太一
    • Organizer
      第46回日本小児内分泌学会学術集会
    • Place of Presentation
      大阪国際会議場(大阪府)
    • Year and Date
      2012-09-29
    • Related Report
      2012 Annual Research Report
  • [Presentation] 小児における血清NT-proCNP値測定の意義一年齢、成長速度及びGH治療反応性-2012

    • Author(s)
      窪田拓生
    • Organizer
      第46回日本小児内分泌学会学術集会
    • Place of Presentation
      大阪国際会議場(大阪府)
    • Year and Date
      2012-09-28
    • Related Report
      2012 Annual Research Report
  • [Presentation] ナトリウム利尿ペプチド受容体2 (NPR-2)機能獲得型変異に伴う高身長症-2家系目2012

    • Author(s)
      三浦弘司
    • Organizer
      第46回日本小児内分泌学会学術集会
    • Place of Presentation
      大阪国際会議場(大阪府)
    • Year and Date
      2012-09-27
    • Related Report
      2012 Annual Research Report
  • [Presentation] Height determination and chondrocyte development-JSPE/ESPE2012

    • Author(s)
      Ozono K
    • Organizer
      51st Annual Meeting of the European Society for Paediatric Endocrinology
    • Place of Presentation
      The Congress Center Leipzig (Leipzig, Germany)(招待講演)
    • Year and Date
      2012-09-22
    • Related Report
      2012 Annual Research Report
  • [Presentation] 骨系統疾患の基礎と臨床-小児科医の立場から-2012

    • Author(s)
      大薗恵一
    • Organizer
      第28回小児成長研究会
    • Place of Presentation
      霞山会館霞ヶ関コモンゲート(東京都)(招待講演)
    • Year and Date
      2012-07-28
    • Related Report
      2012 Annual Research Report
  • [Presentation] 骨系統疾患2012

    • Author(s)
      大薗恵一
    • Organizer
      第30回日本骨代謝学会学術集会
    • Place of Presentation
      京王プラザホテル(東京都)(招待講演)
    • Year and Date
      2012-07-19
    • Related Report
      2012 Annual Research Report
  • [Presentation] 軟骨無形成症の合併症とその治療、先進医療も含めて

    • Author(s)
      大薗恵一
    • Organizer
      第29回 つくしんぼ総会
    • Place of Presentation
      大阪市長居障がい者スポーツセンター
    • Related Report
      2013 Annual Research Report
  • [Presentation] 治療対象となってきた骨系統疾患

    • Author(s)
      大薗恵一
    • Organizer
      第13回 熊本内分泌代謝フォーラム(特別講演)
    • Place of Presentation
      くまもと県民交流館パレア会議室2
    • Related Report
      2013 Annual Research Report
    • Invited
  • [Presentation] 症例をもとに解説するカルシウム代謝異常症Update

    • Author(s)
      大薗恵一
    • Organizer
      第18回 鹿児島県小児内分泌研究会(特別講演)
    • Place of Presentation
      ホテルパレスイン鹿児島4F鳳凰の間
    • Related Report
      2013 Annual Research Report
  • [Book] 骨研究最前線 アンチ・エイジングシリーズ No.3 代謝・疾患のメカニズムから再生医療・創薬・リハビリ機器・機能性食品開発まで2013

    • Author(s)
      大薗恵一
    • Publisher
      エヌ・ティー・エス
    • Related Report
      2013 Annual Research Report
  • [Book] ビタミンDと疾患 改訂版 -基礎の理解と臨床への応用-2013

    • Author(s)
      大薗恵一
    • Publisher
      医薬ジャーナル社
    • Related Report
      2013 Annual Research Report
  • [Book] 骨粗鬆症診療ハンドブック 改訂5版2012

    • Author(s)
      大薗恵一
    • Publisher
      医薬ジャーナル社
    • Related Report
      2012 Annual Research Report
  • [Remarks] 大阪大学 小児科 大阪大学大学院医学系研究科 小児科学 研究紹介

    • URL

      http://www.med.osaka-u.ac.jp/~ped/www/study/study.html

    • Related Report
      2013 Annual Research Report

URL: 

Published: 2012-04-24   Modified: 2019-07-29  

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