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International comparison of autoimmune pulmonary alveolar proteinosis:Platform of rare lung disease research

Research Project

Project/Area Number 24406027
Research Category

Grant-in-Aid for Scientific Research (B)

Allocation TypePartial Multi-year Fund
Section海外学術
Research Field Respiratory organ internal medicine
Research InstitutionClinical Research Center, National Hospital Organization, Kinki-Chuo Chest Medical Center

Principal Investigator

INOUE Yoshikazu  独立行政法人国立病院機構(近畿中央胸部疾患センター臨床研究センター), その他部局等, 臨床研究センター長 (90240895)

Co-Investigator(Kenkyū-buntansha) NAKATS Koh  新潟大学, 医歯学総合病院, 教授 (80207802)
KITAICHI Masanori  国立病院機構近畿中央胸部疾患センター, 臨床研究センター, 室長 (00161464)
AKIRA Masanori  国立病院機構近畿中央胸部疾患センター, 臨床研究センター, 室長 (20393267)
HIROSE Masaki  国立病院機構近畿中央胸部疾患センター, 臨床研究センター, 流動研究員 (90470195)
Project Period (FY) 2012-04-01 – 2015-03-31
Project Status Completed (Fiscal Year 2014)
Budget Amount *help
¥17,290,000 (Direct Cost: ¥13,300,000、Indirect Cost: ¥3,990,000)
Fiscal Year 2014: ¥5,070,000 (Direct Cost: ¥3,900,000、Indirect Cost: ¥1,170,000)
Fiscal Year 2013: ¥5,460,000 (Direct Cost: ¥4,200,000、Indirect Cost: ¥1,260,000)
Fiscal Year 2012: ¥6,760,000 (Direct Cost: ¥5,200,000、Indirect Cost: ¥1,560,000)
Keywords自己免疫性肺胞蛋白症 / 稀少肺疾患国際共同研究 / 診断 / 環境 / 肺胞蛋白症 / 抗GM-CSF自己抗体 / 国際情報交換 / 抗GM-CSF抗体
Outline of Final Research Achievements

(1) International research network of pulmonary alveolar proteinosis (PAP) (Rare lung research platform): Japan Korea (3 institutions), China(5 institutions) have established collaboration network (AsianPAP). International meetings were held at the Asian Pacific Respiratory Society. We keep contact with USA and EU PAP research groups.
(2) International standardization of the diagnosis of PAP: International validation and standardization of ELISA of anti GM-CSF antibodies was performed, and published J Immunol Methods. 2014. 402:57).
(3) An autoimmune PAP (APAP) international survey: 147 Chinese, 78 Korean, and 248 Japanese APAP patients were registered. In China, higher age, more sever patients were observed, and the antibody levels tend to be high. Japanese cohort revealed association between occupational and prognosis.

Report

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

    (37 results)

All 2015 2014 2013 2012 Other

All Journal Article (22 results) (of which Peer Reviewed: 20 results,  Open Access: 8 results,  Acknowledgement Compliant: 7 results) Presentation (11 results) (of which Invited: 3 results) Book (2 results) Remarks (2 results)

  • [Journal Article] A mathematical model to predict protein wash out kinetics during whole-lung lavage in autoimmune pulmonary alveolar proteinosis.2015

    • Author(s)
      Akasaka K, (12名略), Tazawa R, (7名略) Nakata K.
    • Journal Title

      Am J Physiol Lung Cell Mol Physiol

      Volume: 308 Issue: 2 Pages: 105-117

    • DOI

      10.1152/ajplung.00239.2014

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] Pleuroparenchymal fibroelastosis from a consecutive database: a rare disease entity?2015

    • Author(s)
      Nakatani T, Arai T, Kitaichi M, Akira M, Tachibana K, Sugimoto C, Hirooka A, Tsuji T, Minomo S, Hayashi S, Inoue Y
    • Journal Title

      Eur Respir J

      Volume: 45 Issue: 4 Pages: 1183-6

    • DOI

      10.1183/09031936.00214714

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] Standardized serum GM-CSF autoantibody testing for the routine clinical diagnosis of autoimmune pulmonary alveolar proteinosis2014

    • Author(s)
      Uchida K, Nakata K, Carey B, Chalk C, Suzuki T, Sakagami T, Koch DE, Stevens C, Inoue Y, Yamada Y, Trapnell BC.
    • Journal Title

      J Immunol Methods

      Volume: 402 Issue: 1-2 Pages: 57-70

    • DOI

      10.1016/j.jim.2013.11.011

    • Related Report
      2014 Annual Research Report 2013 Annual Research Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] Development of microscopic polyangiitis-related pulmonary fibrosis in a patient with autoimmune pulmonary alveolar proteinosis2014

    • Author(s)
      Kinehara Y, Kida H, Inoue Y, Hirose M, Nakabayashi A, Takeuchi Y, Hayama Y, Fukushima K, Hirata H, Inoue K, Minami T, Nagatomo I, Takeda Y, Funakoshi T, Kijima T, Kumanogoh A
    • Journal Title

      BMC Pulm Med

      Volume: 14 Issue: 1 Pages: 172-172

    • DOI

      10.1186/1471-2466-14-172

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] Secondary pulmonary alveolar proteinosis complicating myelodysplastic syndrome results in worsening of prognosis: a retrospective cohort study in Japan2014

    • Author(s)
      Haruyuki Ishii, John F Seymour, Ryushi Tazawa, Yoshikazu Inoue, Naoyuki Uchida, Aya Nishida, Yoshihito Kogure, Takeshi Saraya, Keisuke Tomii, Toshinori Takada, Yuko Itoh, Masayuki Hojo, Toshio Ichiwata, Hajime Goto, Koh Nakata
    • Journal Title

      BMC Pulmonary Medicine

      Volume: 14: 37 Issue: 1 Pages: 1471-2466

    • DOI

      10.1186/1471-2466-14-37

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] CYFRA 21-1 as a disease severity marker for autoimmune pulmonary alveolar proteinosis.2014

    • Author(s)
      Arai T, Inoue Y, Sugimoto C, Inoue Y, Nakao K, Takeuchi N, Matsumuro A, Hirose M, Nakata K, Hayashi S.
    • Journal Title

      Respirology

      Volume: 19 Issue: 2 Pages: 246-252

    • DOI

      10.1111/resp.12210

    • Related Report
      2014 Annual Research Report 2013 Annual Research Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] Duration of benefit in patients with autoimmune pulmonary alveolar proteinosis after inhaled granulocyte-macrophage colony-stimulating factor therapy.2014

    • Author(s)
      Tazawa R, Inoue Y, Arai T, Takada T, Kasahara Y, Hojo M, Ohkouchi S, Tsuchihashi Y, Yokoba M, Eda R, Nakayama H, Ishii H, Nei T, Morimoto K, Nasuhara Y, Ebina M, Akira M, Ichiwata T, Tatsumi K, Yamaguchi E, Nakata K.
    • Journal Title

      Chest

      Volume: 145 Issue: 4 Pages: 729-737

    • DOI

      10.1378/chest.13-0603

    • Related Report
      2014 Annual Research Report 2013 Annual Research Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] INPULSIS Trial Investigators. Efficacy and safety of nintedanib in idiopathic pulmonary fibrosis2014

    • Author(s)
      Richeldi L, du Bois RM, Raghu G, Azuma A, Brown KK, Costabel U, Cottin V, Flaherty KR, Hansell DM, Inoue Y, Kim DS, Kolb M, Nicholson AG, Noble PW, Selman M, Taniguchi H, Brun M, Le Maulf F, Girard M, Stowasser S, Schlenker-Herceg R, Disse B, Collard HR
    • Journal Title

      N Engl J Med

      Volume: 29 Issue: 22 Pages: 2071-82

    • DOI

      10.1056/nejmoa1402584

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Lymphatic manifestations of lymphangioleiomyomatosis2014

    • Author(s)
      Gupta R, Kitaichi M, Inoue Y, Kotloff R, McCormack FX
    • Journal Title

      Lymphology

      Volume: 47 Pages: 106-17

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Design of the INPULSIS™ trials: two phase 3 trials of nintedanib in patients with idiopathic pulmonary fibrosis2014

    • Author(s)
      Richeldi L, Cottin V, Flaherty KR, Kolb M, Inoue Y, Raghu G, Taniguchi H, Hansell DM, Nicholson AG, Le Maulf F, Stowasser S, Collard HR
    • Journal Title

      Respir Med

      Volume: 108 Issue: 7 Pages: 1023-30

    • DOI

      10.1016/j.rmed.2014.04.011

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Safety and pharmacokinetics of nintedanib and pirfenidone in idiopathic pulmonary fibrosis2014

    • Author(s)
      Ogura T, Taniguchi H, Azuma A, Inoue Y, Kondoh Y, Hasegawa Y, Bando M, Abe S, Mochizuki Y, Chida K, Klüglich M, Fujimoto T, Okazaki K, Tadayasu Y, Sakamoto W, Sugiyama Y
    • Journal Title

      Eur Respir J

      Volume: 45 Issue: 5 Pages: 1382-92

    • DOI

      10.1183/09031936.00198013

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Organ Assessment Instrument Investigators TW. The WASOG Sarcoidosis Organ Assessment Instrument: An update of a previous clinical tool2014

    • Author(s)
      Judson MA, Costabel U, Drent M, Wells A, Maier L, Koth L, Shigemitsu H, Culver DA, Gelfand J, Valeyre D, Sweiss N, Crouser E, Morgenthau AS, Lower EE, Azuma A, Ishihara M, Morimoto S, Yamaguchi T, Shijubo N, Grutters JC, Rosenbach M, Li HP, Rottoli P, Inoue Y, Prasse A, Baughman RP
    • Journal Title

      Sarcoidosis Vasc Diffuse Lung Dis

      Volume: 31 Pages: 19-27

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed
  • [Journal Article] An intractable case of hermansky-pudlak syndrome2014

    • Author(s)
      Kanazu M, Arai T, Sugimoto C, Kitaichi M, Akira M, Abe Y, Hozumi Y, Suzuki T, Inoue Y
    • Journal Title

      Intern Med

      Volume: 53 Pages: 2629-2634

    • NAID

      130004713242

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Predictors of the clinical effects of pirfenidone on idiopathic pulmonary fibrosis2014

    • Author(s)
      Arai T, Inoue Y, Sasaki Y, Tachibana K, Nakao K, Sugimoto C, Okuma T, Akira M, Kitaichi M, Hayashi S
    • Journal Title

      Respir Investig

      Volume: 52 Issue: 2 Pages: 136-43

    • DOI

      10.1016/j.resinv.2013.09.002

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed / Acknowledgement Compliant
  • [Journal Article] Tracheobronchial lesions in eosinophilic pneumonia2014

    • Author(s)
      Matsuda Y, Tachibana K, Sasaki Y, Tsuyuguchi K, Kitaichi M, Inoue Y
    • Journal Title

      Respir Investig

      Volume: 52 Issue: 1 Pages: 21-7

    • DOI

      10.1016/j.resinv.2013.05.006

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Light chain (κ/λ) ratio of GM-CSF autoantibodies is associated with disease severity in autoimmune pulmonary alveolar proteinosis.2013

    • Author(s)
      Nei T, Urano S, Itoh Y, Kitamura N, Hashimoto A, Tanaka T, Motoi N, Kaneko C, Tazawa R, Nakagaki K, Arai T, Inoue Y, Nakata K.
    • Journal Title

      Clin Immunol

      Volume: 149(3) Issue: 3 Pages: 357-364

    • DOI

      10.1016/j.clim.2013.10.002

    • Related Report
      2013 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Direct evidence that GM-CSF inhalation improves lung clearance in pulmonary alveolar proteinosis2012

    • Author(s)
      Ohashi K, Sato A, Takada T, Arai T, Nei T, Kasahara Y, Motoi N, Hojo M, Urano S, Isii H, Yokoba M, Eda R, Nakayama H, Nasuhara Y, Tsuchihashi Y, Kaneko C, Kanazawa H, Ebina M, Yamaguchi E, Kirchner J, Inoue Y, Nakata K, Tazawa R
    • Journal Title

      Respir Med.

      Volume: 106 Pages: 284-293

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Reduced GM-CSF autoantibody in improved lung of autoimmune pulmonary alveolar proteinosis2012

    • Author(s)
      Ohashi K, Sato A, Takada T, Arai T, Kasahara Y, Hojo M, Nei T, Nakayama H, Motoi N, Urano S, Eda R, Yokoba M, Tsuchihashi Y, Nasuhara Y, Ishii H, Ebina M, Yamaguchi E, Inoue Y, Nakata K, Tazawa R
    • Journal Title

      Eur Respir J.

      Volume: 39 Pages: 777-80

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Tetraspanin CD151 protects against pulmonary fibrosis by maintaining epithelial integrity.2012

    • Author(s)
      Tsujino K, Takeda Y, Arai T, Shintani Y, Inagaki R, Saiga H, Iwasaki T, Tetsumoto S, Jin Y, Ihara S, Minami T, Suzuki M, Nagatomo l, Inoue K, Kida H, Kijima T, Ito M, Kitaichi M, Inoue Y, Tachibana l, Takeda K, Okumura M, Hemler ME, Kumanogoh A
    • Journal Title

      Am J Respir Crit Care Med.

      Volume: 186 Pages: 170-80

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] A case of combined sarcoidosis and usual interstitial pneumonia2012

    • Author(s)
      Tachibana K, Arai T, Kagawa T, Minomo S, Akira M, Kitaichi M, Inoue Y
    • Journal Title

      Intern Med.

      Volume: 51 Pages: 1893-7

    • NAID

      130002062237

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] リンパ脈管筋腫症克服に向けて:患者とともに2012

    • Author(s)
      井上義一
    • Journal Title

      呼吸と循環

      Volume: 60 Pages: 355-62

    • Related Report
      2012 Annual Research Report
  • [Journal Article] リンパ脈筋腫症の新たな分子標的治療2012

    • Author(s)
      井上義一
    • Journal Title

      日本サルコイドーシス/肉芽腫性疾患学会雑誌

      Volume: 32 Pages: 89-89

    • Related Report
      2012 Annual Research Report
  • [Presentation] Long term follow-up of serum autoantibody against GM-CSF levels in patients with autoimmune pulmonary alveolar proteinosis2014

    • Author(s)
      Matsumuro A, Hirose M, Arai T, Sugimoto C, Kitaichi M, Akira M, Inoue Y
    • Organizer
      19th Congress of Asian Pasific Society of Respirology
    • Place of Presentation
      Bali, Indonesia
    • Year and Date
      2014-11-14
    • Related Report
      2014 Annual Research Report
  • [Presentation] Serum B cell activating factor belonging to the tumor necrosis factor family (BAFF) in Autoimmune Pulmonary Alveolar Proteinosis2014

    • Author(s)
      Hirose M, Matsumuro A, Arai T, Sugimoto C, Inoue Y
    • Organizer
      9th Congress of Asian Pasific Society of Respirology
    • Place of Presentation
      Bali, Indonesia
    • Year and Date
      2014-11-14
    • Related Report
      2014 Annual Research Report
  • [Presentation] Clinical features and prognosis of 108 patients with lymphangioleiomyomatosis2014

    • Author(s)
      Inoue Y, Hirose M, Nakata K, Arai T, Sugimoto C, Matsuda Y, Takeuchi N, Hirooka A, Tachibana K, Matsumuro A, Honnma T, Kitaichi M, Akira M, Okada M, Hayashi S
    • Organizer
      EUROPIAN RESPIRATORY SOCIETY INTERNATIONAL CONGRTSS 2014
    • Place of Presentation
      Munich, Germany
    • Year and Date
      2014-09-06 – 2014-09-10
    • Related Report
      2014 Annual Research Report
  • [Presentation] B Cell Activating Factor Belonging To The Tumor Necrosis Factor Family In Autoimmune Pulmonary Alveolar Proteinosis2014

    • Author(s)
      Hirose M, Matsumuro A, Arai T, Sugimoto C, Kitaichi M, Akira M, Inoue Y
    • Organizer
      ATS 2014 International Conference
    • Place of Presentation
      Sun Diego, USA
    • Year and Date
      2014-05-16 – 2014-05-21
    • Related Report
      2014 Annual Research Report
  • [Presentation] Roles of bronchofiberscopy for the diagnosis of interstitial lung diseases2014

    • Author(s)
      Inoue. Y
    • Organizer
      18th WCBIP/WCBE
    • Place of Presentation
      Kyoto, Japan
    • Year and Date
      2014-04-13 – 2014-04-16
    • Related Report
      2014 Annual Research Report
    • Invited
  • [Presentation] 肺胞蛋白症2013

    • Author(s)
      井上義一
    • Organizer
      第53回臨床呼吸機能講習会
    • Place of Presentation
      東京
    • Related Report
      2013 Annual Research Report
    • Invited
  • [Presentation] Surveillance of whole lung lavage for autoimmune pulmonary alveolar proteinosis in Japan2013

    • Author(s)
      T. Ichiwata, S. Oh-ishi, K. Uchida, Y. Inoue, T. Arai, H. Ishii, K. Nakata
    • Organizer
      Europian Respiratory Society Annul Congress 2013
    • Place of Presentation
      Barcelona
    • Related Report
      2013 Annual Research Report
  • [Presentation] Vital Capacity And Recurrence After Granulocyte-Macrophage Colony Stimulating Factor (GM-CSF) Inhalation Therapy For Pulmonary Alveolar Proteinosis2013

    • Author(s)
      Tazawa R, Arai T, Kasahara Y, Hojo M, Ohkouchi S, Eda R, Yokoba M, Tsuchihashi Y, Nei T, Nakayama H, Ishii H, Morimoto K, Nasuhara Y, Takada T, Ebina M, Yamaguchi E, Inoue Y, Nakata K
    • Organizer
      American Thoracic Society International Conference 2013
    • Place of Presentation
      Philadelphia
    • Related Report
      2013 Annual Research Report
  • [Presentation] Longitudinal Cohort Of Autoimmune Pulmonary Alveolar Proteinosis2013

    • Author(s)
      Inoue Y, Arai T, Nakata K, Yamaguchi E, Ichiwata T, Ebina M, Tazawa R, Ishii H, Setoguchi Y, Kitaichi M, Akira M, Tatsumi K, Nasuhara Y, Cho K, Tsuchihashi Y, Uchida K, Takada T, Nakayama H, Tomii K, Sugimoto C, Kohashi Y, Ohkouchi S, Kasahara Y, Morimoto K, Nakatani T, Tsuyuguchi K
    • Organizer
      American Thoracic Society International Conference 2013
    • Place of Presentation
      Philadelphia
    • Related Report
      2013 Annual Research Report
  • [Presentation] 稀少肺難病の克服に向けて

    • Author(s)
      中田光
    • Organizer
      第53回日本呼吸器学会総会
    • Place of Presentation
      東京
    • Related Report
      2013 Annual Research Report
    • Invited
  • [Presentation] Rare Lung Diseases Recent advances in rare lung diseases

    • Author(s)
      Costabel U, Inoue Y (Chair)
    • Organizer
      18th Congress of the Asian Pacific Society of Respirology
    • Place of Presentation
      Yokohama
    • Related Report
      2013 Annual Research Report
  • [Book] 肺胞蛋白症 今日の診療のための ガイドライン外来診療2013

    • Author(s)
      新井徹,井上義一
    • Total Pages
      2
    • Publisher
      日経メディカル開発
    • Related Report
      2013 Annual Research Report
  • [Book] 肺胞蛋白症,別冊・医学のあゆみ2013

    • Author(s)
      井上義一
    • Total Pages
      3
    • Publisher
      医歯薬出版株式会社
    • Related Report
      2013 Annual Research Report
  • [Remarks] 国立病院機構近畿中央胸部疾患センター

    • URL

      http://www.hosp.go.jp/~kch/

    • Related Report
      2014 Annual Research Report
  • [Remarks] 日本肺胞蛋白研究グループ英語版ホームページ

    • URL

      http://www.pap-guide.jp/en/index.html

    • Related Report
      2012 Annual Research Report

URL: 

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

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