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2011 Fiscal Year Final Research Report

Analysis of molecular mechanism of hypophosphatasia

Research Project

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Project/Area Number 21592355
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeSingle-year Grants
Section一般
Research Field Functional basic dentistry
Research InstitutionNiigata University

Principal Investigator

ODA Kimimitsu  新潟大学, 医歯学系, 教授 (10122681)

Co-Investigator(Kenkyū-buntansha) SOHDA Miwa  新潟大学, 医歯学系, 助教 (20258528)
Project Period (FY) 2009 – 2011
Keywords遺伝子疾患 / 発症メカニズム / 突然変異 / 石灰化不全 / 骨代謝異常
Research Abstract

Hypophosphatasia(HPP), an inborn error of metabolism, is caused by various loss-in-function mutations in the tissue-nonspecific alkaline phosphatase gene. However, little has been known as to how these mutations lead to development of HPP. In this study we have examined two missense mutations related with disulfide bond linkage and one mutation involved in dominantly transmitted HPP at the molecular level.

  • Research Products

    (47 results)

All 2012 2011 2010 2009 Other

All Journal Article (20 results) (of which Peer Reviewed: 19 results) Presentation (27 results)

  • [Journal Article] Characterization of spontaneous bone marrow recovery after sublethal total body irradiation : importance of the osteoblastic/adipocytic balance2012

    • Author(s)
      Poncin G, Beaulieu A, Humblet C, Thiry A, Oda K, Boniver J, Defresne MP
    • Journal Title

      PLoS One

      Volume: 7 Pages: 30818

    • Peer Reviewed
  • [Journal Article] Disulfide bonds are critical for tissue-nonspecific alkaline phosphatase function revealed by analysis of mutant proteins bearing a C(201)-Y or C(489)-S substitution associated with severe hypophosphatasia2012

    • Author(s)
      Satou Y, Al-Shawafi HA, Sultana S, Makita S, Sohda M, Oda K
    • Journal Title

      Biochim Biophys Acta

      Volume: 1822 Pages: 581-8

    • Peer Reviewed
  • [Journal Article] Histology of epiphyseal cartilage calcification and endochondral ossification2012

    • Author(s)
      Amizuka N, Hasegawa T, Oda K, Luiz de Freitas PH, Hoshi K, Li M, Ozawa H
    • Journal Title

      Front Biosci(Elite Ed)

      Volume: 4 Pages: 2085-100

    • Peer Reviewed
  • [Journal Article] Morphological assessment of bone mineralization in tibial metaphyses of ascorbic acid-deficient ODS rats2011

    • Author(s)
      Hasegawa T, Li M, Hara K, Sasaki M, Tabata C, de Freitas PH, Hongo H, Suzuki R, Kobayashi M, Inoue K, Yamamoto T, Oohata N, Oda K, Akiyama Y, Amizuka N
    • Journal Title

      Biomed Res

      Volume: 32 Pages: 259-69

    • Peer Reviewed
  • [Journal Article] Eldecalcitol, a second-generation vitamin D analog, drives bone minimodeling and reduces osteoclastic number in trabecular bone of ovariectomized rats2011

    • Author(s)
      de Freitas PH, Hasegawa T, Takeda S, Sasaki M, Tabata C, Oda K, Li M, Saito H, Amizuka N.
    • Journal Title

      Bone

      Volume: 49 Pages: 335-42

    • Peer Reviewed
  • [Journal Article] A morphological analysis on the osteocytic lacunar canalicular system in bone surrounding dental implants2011

    • Author(s)
      Haga M, Nozawa-Inoue K, Li M, Oda K, Yoshie S, Amizuka N, Maeda T
    • Journal Title

      Anat Rec(Hoboken)

      Volume: 294 Pages: 1074-82

    • Peer Reviewed
  • [Journal Article] Tissue nonspecific alkaline phosphatase is activated via a two-step mechanism by zinc transport complexes in the early secretory pathway2011

    • Author(s)
      Fukunaka A, Kurokawa Y, Teranishi F, Sekler I, Oda K, Ackland ML, Faundez V, Hiromura M, Masuda S, Nagao M, Enomoto S, Kambe T
    • Journal Title

      J Biol Chem

      Volume: 286 Pages: 16363-73

    • Peer Reviewed
  • [Journal Article] Molecular characterization of tissue-nonspecific alkaline phosphatase with an Ala to Thr substitution at position 116 associated with dominantly inherited hypophosphatasia2011

    • Author(s)
      Ishida Y, Komaru K, Oda K
    • Journal Title

      Biochim Biophys Acta

      Volume: 1812 Pages: 326-32

    • Peer Reviewed
  • [Journal Article] Immunolocalization of DMP1 and sclerostin in the epiphyseal trabecule and diaphyseal cortical bone of osteoprotegerin deficient mice2010

    • Author(s)
      Masuki H, Li M, Hasegawa T, Suzuki R, Ying G, Zhusheng L, Oda K, Yamamoto T, Kawanami M, Amizuka N
    • Journal Title

      Biomed Res

      Volume: 31 Pages: 307-18

    • Peer Reviewed
  • [Journal Article] Interaction of Golgin-84 with the COG complex mediates the intra-Golgi retrograde transport2010

    • Author(s)
      Sohda M, Misumi Y, Yamamoto A, Nakamura N, Ogata S, Sakisaka S, Hirose S, Ikehara Y, Oda K
    • Journal Title

      Traffic

      Volume: 11 Pages: 1552-66

    • Peer Reviewed
  • [Journal Article] Ultrastructural observation on cells meeting the histological criteria for preosteoblasts-a study in the mouse tibial metaphysis2010

    • Author(s)
      Narimatsu K, Li M, de Freitas PH, Sultana S, Ubaidus S, Kojima T, Zhucheng L, Ying G, Suzuki R, Yamamoto T, Oda K, Amizuka N
    • Journal Title

      J Electron Microsc

      Volume: 59 Pages: 427-36

    • Peer Reviewed
  • [Journal Article] FGFR3 down-regulates PTH/PTHrP receptor gene expression by mediating JAK/STAT signaling in chondrocytic cell line2010

    • Author(s)
      Li M, Seki Y, Freitas PH, Nagata M, Kojima T, Sultana S, Ubaidus S, Maeda T, Shimomura J, Henderson JE, Tamura M, Oda K, Liu Z, Guo Y, Suzuki R, Yamamoto T, Takagi R, Amizuka N
    • Journal Title

      J Electron Microsc

      Volume: 59 Pages: 227-36

    • Peer Reviewed
  • [Journal Article] The cellular prion protein interacts with the tissue non-specific alkaline phosphatase in membrane microdomains of bioaminergic neuronal cells2009

    • Author(s)
      Ermonval M, Baudry A, Baychelier F, Pradines E, Pietri M, Oda K, Schneider B, Mouillet-Richard S, Launay JM, Kellermann O
    • Journal Title

      PLoS One

      Volume: 4 Pages: 6497

    • Peer Reviewed
  • [Journal Article] Hormones and osteoporosis update. Histological aspects on the action of parathyroid hormone(PTH) and PTH-related peptide(PTHrP) on bone and cartilage2009

    • Author(s)
      Amizuka N, Li M, Tamura M, Oda K
    • Journal Title

      Clin Calcium

      Volume: 19 Pages: 935-43

  • [Journal Article] FGF23 is mainly synthesized by osteocytes in the regularly distributed osteocytic lacunar canalicular system established after physiological bone remodeling2009

    • Author(s)
      Ubaidus S, Li M, Sultana S, de Freitas PH, Oda K, Maeda T, Takagi R, Amizuka N
    • Journal Title

      J Electron Microsc

      Volume: 58 Pages: 381-92

    • Peer Reviewed
  • [Journal Article] Influence of heat stress to matrix on bone formation2009

    • Author(s)
      Yoshida K, Uoshima K, Oda K, Maeda T
    • Journal Title

      Clin Oral Implants Res

      Volume: 20 Pages: 782-90

    • Peer Reviewed
  • [Journal Article] Intermittent PTH administration stimulates pre-osteoblastic proliferation without leading to enhanced bone formation in osteoclast-less c-fos(-/-) mice2009

    • Author(s)
      Luiz de Freitas PH, Li M, Ninomiya T, Nakamura M, Ubaidus S, Oda K, Udagawa N, Maeda T, Takagi R, Amizuka N
    • Journal Title

      J Bone Miner Res

      Volume: 24 Pages: 1586-97

    • Peer Reviewed
  • [Journal Article] Warfarin administration disrupts the assembly of mineralized nodules in the osteoid2009

    • Author(s)
      Amizuka N, Li M, Hara K, Kobayashi M, de Freitas PH, Ubaidus S, Oda K, Akiyama Y
    • Journal Title

      J Electron Microsc

      Volume: 58 Pages: 55-65

    • Peer Reviewed
  • [Journal Article] Immunolocalization of sclerostin synthesized by osteocytes in relation to bone remodeling in the interradicular septa of ovariectomized rats

    • Author(s)
      Guo Y., Li M., Liu Z., Sasaki M., Hasegawa T., Hongo H., Tabata C., Suzuki R., Oda K., Yamamoto T., Kawanami M., Amizuka N.
    • Journal Title

      J Electron Microsc

      Volume: (in press)

    • Peer Reviewed
  • [Journal Article] Morphological Aspects on Osteocytic Function on Bone Mineralization

    • Author(s)
      Sasaki M., Hongo H., Hasegawa T., Suzuki R., Liu Z., Freitas PHL., Oda K., Yamamoto T., Li M., Totsuka Y., Amizuka N.
    • Journal Title

      Oral Science International

      Volume: (in press)

    • Peer Reviewed
  • [Presentation] Histological examination on bone regeneration induced by a porous beta-TCP block mingled with bone marrow stromal cells2011

    • Author(s)
      Kojima T.
    • Organizer
      International Bone-Tissue-Engineering Congress
    • Place of Presentation
      Hannover, Germany
    • Year and Date
      20111012-15
  • [Presentation] 卵巣摘出ラットの下顎歯槽骨におけるsclerostinの組織化学的観察2011

    • Author(s)
      郭頴
    • Organizer
      第53回歯科基礎医学会学術大会
    • Place of Presentation
      岐阜
    • Year and Date
      20110930-1002
  • [Presentation] 骨組織におけるオステオカルシンと基質グラ蛋白の局在について.2011

    • Author(s)
      韋山良
    • Organizer
      第53回歯科基礎医学会学術大会
    • Place of Presentation
      岐阜
    • Year and Date
      20110930-1002
  • [Presentation] Endochondral ossification in mice over-expressing tissue non-specific alkaline phosphatase driven by type II collagen promoter2011

    • Author(s)
      Hasegawa T.
    • Organizer
      IOF Regionals 2nd Asia-Pacific Osteoporosis and Bone Meeting, ANZBMS annual scientific meeting
    • Place of Presentation
      Gold Coast, Australia
    • Year and Date
      20110904-08
  • [Presentation] 卵巣摘出ラット脛骨の海綿骨におけるビタミンDアナログ-Eldecalcitol-の投与効果について2011

    • Author(s)
      李敏啓
    • Organizer
      第29回日本骨代謝学会学術集会
    • Place of Presentation
      大阪
    • Year and Date
      20110728-30
  • [Presentation] アルカリフォスファターゼトランスジェニックマウスにおける基質石灰化機構の解明2011

    • Author(s)
      長谷川智香
    • Organizer
      第29回日本骨代謝学会学術集会
    • Place of Presentation
      大阪
    • Year and Date
      20110728-30
  • [Presentation] Interaction of Golgin-84 with the conserved oligomeric Golgi(COG) complex mediates the intra-Golgi retrograde transport2010

    • Author(s)
      Sohda M.
    • Organizer
      第33回日本分子生物学会・第83回日本生化学会合同学会
    • Place of Presentation
      神戸
    • Year and Date
      20101207-08
  • [Presentation] 歯限局型低フォスファターゼ症で報告された変異型組織非特異型アルカリフォスファターゼの解析2010

    • Author(s)
      金城奈津子
    • Organizer
      平成22年度日本小児歯科学会秋季大会抄録小児歯誌
    • Place of Presentation
      郡山
    • Year and Date
      20101202-03
  • [Presentation] 骨芽細胞特異的PTHrPトランジェニックマウスの骨組織における解析2010

    • Author(s)
      李骨芽
    • Organizer
      第52回歯科基礎医学会学術大会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] アスコルビン酸欠乏ラットの歯根膜異常における組織化学的検索2010

    • Author(s)
      長谷川智香
    • Organizer
      第52回歯科基礎医学会学術大会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] オステオプロテジェリン遺伝子欠損マウスにおける骨細胞産生蛋白の局在2010

    • Author(s)
      木英郎
    • Organizer
      第52回歯科基礎医学会学術大会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] 根間中隔歯槽骨の骨細胞は骨改造に応じてsclerostin産生を調節する2010

    • Author(s)
      郭頴
    • Organizer
      第52回歯科基礎医学会学術大会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] Klotho遺伝子欠損マウスの脛骨骨端部における骨細胞産生蛋白の局在について2010

    • Author(s)
      佐々木宗輝
    • Organizer
      第52回歯科基礎医学会学術大会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] ラット臼歯の歯周組織におけるペリオスチンとファイブロネクチンの局在について2010

    • Author(s)
      田幡千尋
    • Organizer
      第52回歯科基礎医学会学術大会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] RANKL遺伝子欠損マウスにおける骨・軟骨基質を取り込む細胞と活性型骨芽細胞の組織化学的検索2010

    • Author(s)
      宮本幸奈
    • Organizer
      第52回歯科基礎医学会学術大会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] 骨芽細胞特異的PTHrP過剰発現マウスの骨組織における組織化化学的解析2010

    • Author(s)
      李敏啓
    • Organizer
      第52回歯科基礎医学会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] 根間中隔歯槽骨の骨細胞は骨改造に応じてsclerostin産生を調節する2010

    • Author(s)
      郭頴
    • Organizer
      第52回歯科基礎医学会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] アスコルビン酸欠乏ラットの歯根膜異常における組織化学的検索2010

    • Author(s)
      長谷川智香
    • Organizer
      第52回歯科基礎医学会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] Klotho遺伝子欠損マウスの脛骨骨端部における骨細胞産生蛋白の局在について2010

    • Author(s)
      佐々木宗輝
    • Organizer
      第52回歯科基礎医学会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] ラット臼歯の歯周組織におけるペリオスチンとファイブロネクチンの局在について2010

    • Author(s)
      田幡千尋
    • Organizer
      第52回歯科基礎医学会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] RANKL遺伝子欠損マウスにおける骨・軟骨基質を取り込む細胞と活性型骨芽細胞の組織化学的検索2010

    • Author(s)
      宮本幸奈
    • Organizer
      第52回歯科基礎医学会
    • Place of Presentation
      東京
    • Year and Date
      20100920-22
  • [Presentation] Histological examination on bone augmentation withβ-tri-calcium phosphate and a thermoplastic bioresorbable plate2010

    • Author(s)
      Kojima T.
    • Organizer
      20th Congress of the European Association for Cranio-Maxillo-Facial Surgery
    • Place of Presentation
      ruges, Belgium
    • Year and Date
      20100914-18
  • [Presentation] 卵巣摘出ラットの下顎歯槽骨における骨細胞機能に関連する蛋白の局在について2010

    • Author(s)
      郭穎
    • Organizer
      第28回日本骨代謝学会学術集会
    • Place of Presentation
      東京
    • Year and Date
      20100720-22
  • [Presentation] アスコルビン酸合成能欠如ラットの骨基質石灰化における微細構造学的観察2010

    • Author(s)
      李敏啓
    • Organizer
      第115回日本解剖学会
    • Place of Presentation
      岩手
    • Year and Date
      20100328-30
  • [Presentation] c-fos遺伝子欠損マウスにおける骨細胞の組織化学的解析2010

    • Author(s)
      佐々木理絵
    • Organizer
      第115回日本解剖学会総会・全国学術集会
    • Place of Presentation
      岩手
    • Year and Date
      20100328-30
  • [Presentation] マウス歯胚発育過程におけるグリコーゲンおよびグルコース輸送体の局在2009

    • Author(s)
      依田浩子
    • Organizer
      第51回歯科基礎医学会
    • Place of Presentation
      新潟市
    • Year and Date
      2009-09-11
  • [Presentation] ラット血中骨型酒石酸抵抗性酸性フォスファターゼ(TRACP-5b)測定試薬の開発とその基礎性能2009

    • Author(s)
      三浦俊英
    • Organizer
      第42回新潟歯学会総会
    • Place of Presentation
      新潟市
    • Year and Date
      2009-04-18

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Published: 2013-07-31  

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