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Analysis of the neural network formation in NZF-2/-3 double knock-out mice that exhibit symptoms of arthrogryposis

Research Project

Project/Area Number 26460259
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Research Field General anatomy (including histology/embryology)
Research InstitutionFujita Health University

Principal Investigator

MATSUSHITA Fumio  藤田医科大学, 保健学研究科, 准教授 (50298543)

Co-Investigator(Kenkyū-buntansha) 亀山 俊樹  藤田医科大学, 総合医科学研究所, 助教 (60298544)
Project Period (FY) 2014-04-01 – 2020-03-31
Project Status Completed (Fiscal Year 2019)
Budget Amount *help
¥4,940,000 (Direct Cost: ¥3,800,000、Indirect Cost: ¥1,140,000)
Fiscal Year 2018: ¥650,000 (Direct Cost: ¥500,000、Indirect Cost: ¥150,000)
Fiscal Year 2017: ¥910,000 (Direct Cost: ¥700,000、Indirect Cost: ¥210,000)
Fiscal Year 2016: ¥780,000 (Direct Cost: ¥600,000、Indirect Cost: ¥180,000)
Fiscal Year 2015: ¥1,170,000 (Direct Cost: ¥900,000、Indirect Cost: ¥270,000)
Fiscal Year 2014: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000)
Keywordsneural zinc finger / myelin / transcription factor / 神経発生 / 神経回路 / 関節拘縮 / 遺伝子破壊マウス / トランスクリプトーム / NZF / Myt / ミエリン / 変異マウス / 転写因子
Outline of Final Research Achievements

We generated the double knockout mice for gene silencing of NZF-2 and NZF-3, and compared these mice with normal or single knockout mice of NZF-2 or NZF-3 by their histology and gene-expression levels.
As a result, the neurons did not innervate from spinal cord into the diaphragm or the distal portion of the extremities. In cerebral cortex, the bundles of nerve fibers were completely lost. In the genes that have moderately changed expression levels in the double knockout mice compared to wild type mice, we found ones that control alternative splicing. This finding suggests that NZF-2 and NZF-3 may influence alternative splicing patterns of other genes.

Academic Significance and Societal Importance of the Research Achievements

本研究で得られた結果は、神経細胞が神経線維を伸ばして情報網をつくる際に、2つの遺伝子NZF-2とNZF-3が協力して必要不可欠な働きをすることを示しています。また、正常マウスと比べて二重変異マウスでは、他の遺伝子の働き方が変化する可能性があります。こうした遺伝子やNZF-2とNZF-3の働きは、再生医療で神経を再生させたり、遺伝子の働き方の変化を検査して神経難病を診断するなどの応用が期待できます。

Report

(7 results)
  • 2019 Annual Research Report   Final Research Report ( PDF )
  • 2018 Research-status Report
  • 2017 Research-status Report
  • 2016 Research-status Report
  • 2015 Research-status Report
  • 2014 Research-status Report
  • Research Products

    (41 results)

All 2019 2018 2017 2016 2015 2014

All Journal Article (13 results) (of which Int'l Joint Research: 1 results,  Peer Reviewed: 12 results,  Open Access: 4 results,  Acknowledgement Compliant: 2 results) Presentation (28 results) (of which Int'l Joint Research: 5 results,  Invited: 4 results)

  • [Journal Article] Cancer-Specifically Re-Spliced TSG101 mRNA Promotes Invasion and Metastasis of Nasopharyngeal Carcinoma2019

    • Author(s)
      Chua Huey-Huey、Kameyama Toshiki、Mayeda Akila、Yeh Te-Huei
    • Journal Title

      International Journal of Molecular Sciences

      Volume: 20 Issue: 3 Pages: 773-773

    • DOI

      10.3390/ijms20030773

    • Related Report
      2018 Research-status Report
    • Peer Reviewed / Open Access / Int'l Joint Research
  • [Journal Article] Effects of plasma glycosyltransferase on the ABO(H) blood group antigens of human von Willebrand factor2018

    • Author(s)
      Kano Taiki、Kondo Kazunao、Hamako Jiharu、Matsushita Fumio、Sakai Kazuya、Matsui Taei
    • Journal Title

      International Journal of Hematology

      Volume: 108 Issue: 2 Pages: 139-144

    • DOI

      10.1007/s12185-018-2452-0

    • Related Report
      2018 Research-status Report
    • Peer Reviewed
  • [Journal Article] シスプラチン抵抗性ヒト膀胱癌細胞株の樹立とRNA-seqによる網羅的遺伝子発現解析2018

    • Author(s)
      飴本剛之介、亀山俊樹、日下守、前田明、白木良一
    • Journal Title

      藤田学園医学会誌

      Volume: 42 Pages: 37-40

    • Related Report
      2018 Research-status Report
    • Peer Reviewed
  • [Journal Article] Rearrangement of VPS13B, a causative gene of Cohen syndrome, in a case of RUNX1-RUNX1T1 leukemia with t(8;12;21).2017

    • Author(s)
      Abe A, Yamamoto Y, Katsumi A, Okamoto A, Tokuda M, Inaguma Y, Yamamoto K, Yanada M, Kanie T, Tomita A, Akatsuka Y, Okamoto M, Kameyama T, Mayeda A, Emi N.
    • Journal Title

      Int J Hematol.

      Volume: - Issue: 2 Pages: 208-212

    • DOI

      10.1007/s12185-017-2387-x

    • Related Report
      2018 Research-status Report 2017 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] MU314, a novel selective estrogen receptor modulator (SERM), improves estrogen-dependent depressive behaviors2017

    • Author(s)
      N Izumo、Y Ishibashi、Y Ono、M Toho、A Sumino、T Kameyama、T Morikawa、Y Shiba、Y Watanabe、T Manabe
    • Journal Title

      Global Drugs and Therapeutics

      Volume: 2 Issue: 5

    • DOI

      10.15761/gdt.1000s1004

    • Related Report
      2017 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Mutant botrocetin-2 inhibits von Willebrand factor-induced platelet agglutination2017

    • Author(s)
      T. Matsui, A. Hori, J. Hamako, F. Matsushita, Y. Ozeki, Y. Sakurai, M. Hayakawa, M. Matsumoto, Y. Fujimura
    • Journal Title

      Journal of Thrombosis and Haemostasis

      Volume: 15 Issue: 3 Pages: 538-548

    • DOI

      10.1111/jth.13617

    • Related Report
      2016 Research-status Report
    • Peer Reviewed
  • [Journal Article] Endogenous Multiple Exon Skipping and Back-Splicing at the DMD Mutation Hotspot2016

    • Author(s)
      Hitoshi Suzuki, Yoshitsugu Aoki, Toshiki Kameyama, Takashi Saito, Satoru Masuda, Jun Tanihata, Tetsuya Nagata, Akila Mayeda, Shin’ichi Takeda, Toshifumi Tsukahara
    • Journal Title

      Int. J. Mol. Sci.

      Volume: 17 Issue: 10 Pages: 1722-1722

    • DOI

      10.3390/ijms17101722

    • Related Report
      2016 Research-status Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] Transcriptional activation of platelet-derived growth factor receptor α and GS homeobox 2 resulting from E26 transformation-specific variant 6 translocation in a case of acute myeloid leukemia with t(4;12)(q12;p13).2016

    • Author(s)
      Abe A, Mizuta S, Okamoto A, Yamamoto Y, Kameyama T, Mayeda A, Emi N.
    • Journal Title

      International journal of laboratory hematology

      Volume: 38(2) Issue: 2 Pages: 15-18

    • DOI

      10.1111/ijlh.12450

    • Related Report
      2015 Research-status Report
    • Peer Reviewed
  • [Journal Article] ETV6-LPXN fusion transcript generated by t(11;12)(q12.1;p13) in a patient with relapsing acute myeloid leukemia with NUP98-HOXA9.2016

    • Author(s)
      Abe A, Yamamoto Y, Iba S, Kanie T, Okamoto A, Tokuda M, Inaguma Y, Yanada M, Morishima S, Mizuta S, Akatsuka Y, Okamoto M, Kameyama T, Mayeda A, Emi N.
    • Journal Title

      Genes, chromosomes & cancer

      Volume: 55(3) Issue: 3 Pages: 242-250

    • DOI

      10.1002/gcc.22327

    • Related Report
      2015 Research-status Report
    • Peer Reviewed
  • [Journal Article] NUP214-RAC1 and RAC1-COL12A1 Fusion in Complex Variant Translocations Involving Chromosomes 6, 7 and 9 in an Acute Myeloid Leukemia Case with DEK-NUP214.2015

    • Author(s)
      Abe A, Yamamoto Y, Iba S, Okamoto A, Tokuda M, Inaguma Y, Yanada M, Morishima S, Kanie T, Tsuzuki M, Akatsuka Y, Mizuta S, Okamoto M, Kameyama T, Mayeda A, Emi N.
    • Journal Title

      Cytogenetic and genome research

      Volume: 146(4) Issue: 4 Pages: 279-284

    • DOI

      10.1159/000441464

    • Related Report
      2015 Research-status Report
    • Peer Reviewed
  • [Journal Article] がん細胞で異常なタンパク質が作られるしくみを「mRNA再スプライシング」現象から探る2015

    • Author(s)
      亀山俊樹、前田明
    • Journal Title

      ファルマシア

      Volume: 51 Pages: 22-26

    • Related Report
      2014 Research-status Report
  • [Journal Article] Fluvoxamine moderates reduced voluntary activity following chronic dexamethasone infusion in mice via recovery of BDNF signal cascades2014

    • Author(s)
      Terada K, Izumo N, Suzuki B, Karube Y, Morikawa T, Ishibashi Y, Kameyama T, Chiba K, Sasaki N, Iwata K,
    • Journal Title

      Neurochemistry International

      Volume: 69 Issue: 1 Pages: 9

    • DOI

      10.1016/j.neuint.2014.02.002

    • Related Report
      2014 Research-status Report
    • Peer Reviewed
  • [Journal Article] Spatiotemporal expression pattern of Myt/NZF family zinc finger transcription factors during mouse nervous system development.2014

    • Author(s)
      Fumio Matsushita, *Toshiki Kameyama, Yuzo Kadokawa and Tohru Marunouchi
    • Journal Title

      Developmental Dynamics.

      Volume: 243 Issue: 4 Pages: 588-600

    • DOI

      10.1002/dvdy.24091

    • Related Report
      2014 Research-status Report
    • Peer Reviewed / Acknowledgement Compliant
  • [Presentation] 流動下で培養した血管内皮細胞が産生するVWF上の糖鎖の解析2019

    • Author(s)
      中村優太 松下文雄 濱子二治 雪竹 潤 秋山秀彦 松井太衛
    • Organizer
      第14回日本臨床検査学教育学会学術大会
    • Related Report
      2019 Annual Research Report
  • [Presentation] EJCが癌特異的な成熟 mRNA再スプライシングを抑制する:mRNAの品質管理に関わる新たな EJCの役割2019

    • Author(s)
      大谷 勇太, 亀山 俊樹, Peter Venhuizen, Maria Kalyna, 前田 明
    • Organizer
      【ワークショップ】真核生物におけるストレスに応答した遺伝子発現制御とその破綻に伴う老化および疾患 第42回日本分子生物学会年会
    • Related Report
      2019 Annual Research Report
    • Invited
  • [Presentation] EJC CORE REPRESSES CANER-SPECIFIC MATURE mRNA RE-SPLICING: EJC AS A SIGNAL TO TERMINATE SPLICING2019

    • Author(s)
      Yuta Ohtani, Toshiki Kameyama, Peter Venhuizen, Maria Kalyna, Akila Mayeda
    • Organizer
      Cold Spring Harbor Laboratory Meeting “EUKARYOTIC mRNA PROCESSING”
    • Related Report
      2019 Annual Research Report
    • Int'l Joint Research
  • [Presentation] Cancer specific aberrant mRNA re-splicing and its repressors: What are the splicing termination mechanisms destroyed in cancer cells2019

    • Author(s)
      亀山俊樹
    • Organizer
      招待セミナー、ウィーン天然資源・生命科学大学(ウィーン農科大学)応用遺伝学・細胞生物学科
    • Related Report
      2018 Research-status Report
    • Int'l Joint Research / Invited
  • [Presentation] VWF 依存性血小板凝集を惹起する組換えビチセチン-3 とビチセチンの活性比較2018

    • Author(s)
      中村優太,内藤菜月,藤田佑奈,大場愛梨,岩田奈津美,小田祐人,小野佑介,松下文雄,浜子二治,松井太衛
    • Organizer
      第40回日本血栓止血学会学術集会
    • Related Report
      2018 Research-status Report
  • [Presentation] 培養血管内皮細胞が産生するVWF上の血液型抗原に関する研究2018

    • Author(s)
      中村優太 濱子二治 松下文雄 雪竹 潤 秋山秀彦 松井太衛
    • Organizer
      第13回日本臨床検査学教育学会学術大会
    • Related Report
      2018 Research-status Report
  • [Presentation] EJCががん特異的な成熟mRNA再スプライシングを抑制する:スプライシング正常化にはたらく新たなEJCの役割2018

    • Author(s)
      大谷勇太、亀山俊樹、前田明
    • Organizer
      第41回日本分子生物学会年会
    • Related Report
      2018 Research-status Report
  • [Presentation] がん抑制遺伝子RBM4aはがん細胞特異的成熟mRNA再スプライシングを抑制する2018

    • Author(s)
      亀山俊樹
    • Organizer
      第77回日本癌学会学術総会
    • Related Report
      2018 Research-status Report
  • [Presentation] がん抑制因子RBM4aはがん細胞特異的成熟mRNA再スプライシングを抑制する2018

    • Author(s)
      亀山俊樹、福村和宏、井上邦夫、廣瀬哲郎、前田明
    • Organizer
      第20回日本RNA学会年会
    • Related Report
      2018 Research-status Report
  • [Presentation] がん抑制因子RBM4aはがん細胞特異的成熟mRNA再スプライシングを抑制する: mRNA品質保証の鍵となる因子か?2017

    • Author(s)
      亀山俊樹、福村和宏、井上邦夫、廣瀬哲郎、前田明
    • Organizer
      2017年度生命科学系学会合同年次大会(ConBio2017)
    • Related Report
      2017 Research-status Report
  • [Presentation] A Repressor Candidate of Cancer Specific mRNA Re-splicing: A Key factor for splicing fidelity or mRNA quality control?2017

    • Author(s)
      亀山俊樹、福村和宏、井上邦夫、廣瀬哲朗、前田明
    • Organizer
      RNA2017:第22回国際RNA学会年会
    • Related Report
      2017 Research-status Report
    • Int'l Joint Research
  • [Presentation] A Repressor Candidate of Cancer Specific mRNA Re-splicing: A Key factor for splicing fidelity or mRNA quality control?2017

    • Author(s)
      亀山俊樹、福村和宏、井上邦夫、廣瀬哲朗、前田明
    • Organizer
      International Symposium on the Hallmarks of Cancer: Focus on RNA, a satellite meeting of “RNA 2017”
    • Related Report
      2017 Research-status Report
    • Int'l Joint Research
  • [Presentation] Cisplatin-stimulated Cancer-specific Mature mRNA Re-splicing Is under the Control of Tumor Suppressor p532016

    • Author(s)
      Toshiki Kameyama, Yoshinosuke Amemoto, Akila Mayeda
    • Organizer
      The 21th Annual Meeting of the RNA Society
    • Place of Presentation
      The International Conference Center in Kyoto
    • Year and Date
      2016-06-28
    • Related Report
      2016 Research-status Report
    • Int'l Joint Research
  • [Presentation] 臍帯血管内皮細胞由来VWFの血液型抗原に関する研究2016

    • Author(s)
      松井 太衛, 石橋 香里, 岩坪 里奈, 浜子 二治, 松下 文雄, 狩野 泰輝, 近藤 一直
    • Organizer
      第38回日本血栓止血学会学術集会
    • Place of Presentation
      奈良(奈良春日野国際フォーラム)
    • Year and Date
      2016-06-16
    • Related Report
      2016 Research-status Report
  • [Presentation] t(8;12;21)(q22:p12;q22)を有するRUNX1-RUNX1T1白血病に認められたTM7SF4-VPS13B融合遺伝子2015

    • Author(s)
      安部明弘、山本幸也、岡本晃直、徳田倍将、稲熊容子、山本起代子、柳田正光、森島聡子、蟹江匡治、赤塚美樹、水田秀一、岡本昌隆、亀山俊樹、前田 明、恵美宣彦
    • Organizer
      第76回日本血液学会学術集会
    • Place of Presentation
      石川県立音楽堂(石川県金沢市)
    • Year and Date
      2015-10-16
    • Related Report
      2015 Research-status Report
  • [Presentation] Hypoxia had an effect on cancer-specific mature mRNA re-splicing.2015

    • Author(s)
      Toshiki Kameyama, Shota Matsumiya and Akila Mayeda
    • Organizer
      Cold Spring Harbor Laboratory Meeting “Eukaryotic mRNA Processing”
    • Place of Presentation
      Cold Spring Harbor Laboratory, NY USA
    • Year and Date
      2015-08-18
    • Related Report
      2015 Research-status Report
  • [Presentation] 低酸素ストレスは癌細胞特異的成熟mRNA再スプライシングに影響を与える2015

    • Author(s)
      亀山俊樹、松宮翔太、前田明
    • Organizer
      第17回日本RNA学会年会
    • Place of Presentation
      ホテルライフォート札幌(北海道札幌市)
    • Year and Date
      2015-07-15
    • Related Report
      2015 Research-status Report
  • [Presentation] Regulation of VWF-GPIB interaction with modified recombinant botrocetin2015

    • Author(s)
      Matsui T, Hori A, Hamako J, Matsushita F, Takagishi N, Kondo K, Kano T, Hayakawa M, Matsumoto M and Fujimura Y
    • Organizer
      The International Society on Thrombosis and Haemostasis 2015 Congress and 61st Annual SSC Meeting
    • Place of Presentation
      Metro Toronto Convention Center (MTCC), Toronto, Canada
    • Year and Date
      2015-06-20
    • Related Report
      2015 Research-status Report
  • [Presentation] Modification of ABO(H) blood group antigens on human vwf with plasma glycosyltransferase2015

    • Author(s)
      Kano T, Matsushita F, Hamako J, Fujimura Y, Kondo K and Matsui T
    • Organizer
      The International Society on Thrombosis and Haemostasis 2015 Congress and 61st Annual SSC Meeting
    • Place of Presentation
      Metro Toronto Convention Center (MTCC), Toronto, Canada
    • Year and Date
      2015-06-20
    • Related Report
      2015 Research-status Report
  • [Presentation] ヒトVWF に存在するABO(H)血液型抗原の付加経路の解析2015

    • Author(s)
      狩野泰輝,松下文雄,浜子二治,松本雅則,藤村吉博,近藤一直,松井太衛
    • Organizer
      第37回日本血栓止血学会学術集会
    • Place of Presentation
      甲府市総合市民会館(山梨県甲府市)
    • Year and Date
      2015-05-21
    • Related Report
      2015 Research-status Report
  • [Presentation] 変異導入組換えボトロセチン-2 を用いた血小板凝集の制御2015

    • Author(s)
      高岸波穂,堀有沙1,浜子二治,松下文雄,松本雅則,早川正樹,藤村吉博,狩野泰揮,近藤一直,松井太衛
    • Organizer
      第37回日本血栓止血学会学術集会
    • Place of Presentation
      甲府市総合市民会館(山梨県甲府市)
    • Year and Date
      2015-05-21
    • Related Report
      2015 Research-status Report
  • [Presentation] Mature mRNA Re-splicing in Cancer Cells2015

    • Author(s)
      Toshiki Kameyama
    • Organizer
      Invited Seminar for Harvard Medical School
    • Place of Presentation
      Boston (USA)
    • Year and Date
      2015-02-05
    • Related Report
      2014 Research-status Report
    • Invited
  • [Presentation] Novel cancer specific mature mRNA re-splicing postulates undiscovered mRNA quality control mechanism in normal cells2015

    • Author(s)
      Toshiki Kameyama and Akila Mayeda
    • Organizer
      The 10th Cold Spring Harbor Conference “Systems Biology: Global Regulation of Gene Expression”
    • Place of Presentation
      Rio Grande (Puerto Rico)
    • Year and Date
      2015-01-28 – 2015-02-01
    • Related Report
      2014 Research-status Report
  • [Presentation] Additional NUP214-RAC1 fusion gene in an AML patient with DEK-NUP2142014

    • Author(s)
      安部明弘、山本幸也、岡本晃直、徳田倍将、稲熊容子、柳田正光、森島聡子、蟹江匡治、都築基弘、赤塚美樹、水田秀一、岡本昌隆、亀山俊樹、前田 明、恵美宣彦
    • Organizer
      第76回日本血液学会学術集会
    • Place of Presentation
      大阪国際会議場(大阪)
    • Year and Date
      2014-10-31 – 2014-11-02
    • Related Report
      2014 Research-status Report
  • [Presentation] Novel mRNA re-splicing event: important aspects for understanding robustness and catastrophe in gene expression systems.2014

    • Author(s)
      Toshiki Kameyama and Akila Mayeda
    • Organizer
      RIKEN Symposium & The 15th Tokyo RNA Club “Noncoding RNA Regulation”
    • Place of Presentation
      鈴木梅太郎記念ホール(埼玉・和光)
    • Year and Date
      2014-10-01
    • Related Report
      2014 Research-status Report
    • Invited
  • [Presentation] VWF-GPIb依存性血小板凝集を制御する組換えボトロセチン-2変異体の発現2014

    • Author(s)
      髙岸波穂,堀有沙,狩野泰揮,近藤一直,松下文雄,濵子二治,松本雅則,藤村吉博,松井太衛
    • Organizer
      第9回 日本臨床検査学教育学会学術大会
    • Place of Presentation
      大田区産業プラザ(東京)
    • Year and Date
      2014-08-20 – 2014-08-22
    • Related Report
      2014 Research-status Report
  • [Presentation] がん細胞での成熟mRNA再スプライシング活性に影響を与える核外輸送因子2014

    • Author(s)
      亀山 俊樹、増田 誠司、前田 明
    • Organizer
      RNA2014 第16回日本RNA学会年会
    • Place of Presentation
      ウィンクあいち(愛知・名古屋)
    • Year and Date
      2014-07-23 – 2014-07-25
    • Related Report
      2014 Research-status Report
  • [Presentation] 長鎖ノンコーディングRNA Malat1におけるスプライシング抑制機構の存在2014

    • Author(s)
      佐々木(原口) 典子、亀山  俊樹、眞部  孝幸、秋光 信佳、前田  明
    • Organizer
      RNA2014 第16回日本RNA学会年会
    • Place of Presentation
      ウィンクあいち(愛知・名古屋)
    • Year and Date
      2014-07-23 – 2014-07-25
    • Related Report
      2014 Research-status Report

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Published: 2014-04-04   Modified: 2021-02-19  

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