International epidemiological studies and standar dization of diagnosis and treatment of autoimmune pulmonary alveolar proteinosis : Research basis of rare lung diseases.

2011 Fiscal Year Final Research Report

• Project/Area Number: 21406025
• Research Category: Grant-in-Aid for Scientific Research (B)
• Allocation Type: Single-year Grants
• Section: 海外学術
• Research Field: Respiratory organ internal medicine
• Research Institution: 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) (2011); Clinical Research Center, National Hospital Organization, Kinki-Chuo Chest Medical Center (2009-2010)
• 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), 呼吸不全・難治性肺疾患研究, 部長 (90240895)
• Co-Investigator(Kenkyū-buntansha): NAKATA Koh 新潟大学, 医歯学総合病院, 教授 (80207802) ; KITAICHI Masanori 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), 臨床研究センター室長, 病理部長 (00161464) ; AKIRA Masanori 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), 臨床研究センター室長, 放射線部長 (20393267) ; HIROSE Masaki 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), 臨床研究センター流動, 研究員 (90470195)
• Project Period (FY): 2009 – 2011
• Keywords: 肺胞蛋白症 / 疫学 / 国際標準化 / 稀少肺疾患 / 抗GM-CSF自己抗体

Research Abstract

Pulmonary alveolar proteinosis(PAP) is a rare lung disease characterized by accumulation of surfactant-like materials in the lungs. Based on our new classification by auto-anti GM-CSF antibodies and our Japanese epidemiological data, we performed international standardization of measurements of the auto-antibodies and comparison of autoimmune PAP(collaboration of rare lung disease consortium, USA). We have shown our Japanese guideline for PAP in Europe, USA, and Asia, and preparation of international guideline is going on.

Research Products

• [Journal Article] Direct evidence that GM-CSF inhalation improves lung clearance in pulmonary alveolar proteinosis (2012)
  • 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(2) Pages: 284-93
  • Peer Reviewed
• [Journal Article] Reduced GM-CSF autoantibody in improved lung of autoimmune pulmonary alveolar proteinosis (2012)
  • 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(3) Pages: 777-780
  • Peer Reviewed
• [Journal Article] Long-term follow-up high-resolution CT findings in non-specific interstitial pneumonia (2011)
  • Author(s): Akira M, Inoue Y, Arai T, Okuma T, Kawata Y
  • Journal Title: Thorax Volume: 66(1) Pages: 61-5
  • Peer Reviewed
• [Journal Article] Clinical features of secondary pulmonary alveolar proteinosis : pre-mortem cases in Japan (2011)
  • Author(s): Ishii H, Tazawa R, Kaneko C, Saraya T, Inoue Y, Hamano E, Kogure Y, Tomii K, Terada M, Takada T, Hojo M, Nishida A, Ichiwata T, Trapnell BC, Goto H, Nakata K
  • Journal Title: Eur Respir J Volume: 37(2) Pages: 465-8
  • Peer Reviewed
• [Journal Article] National Institutes of Health Rare Lung Diseases Consortium ; MILES Trial Group. Efficacy and Safety of Sirolimus in Lymphangioleiomyomatosis (2011)
  • Author(s): McCormack FX, Inoue Y, Moss J, Singer LG, Strange C, Nakata K, Barker AF, Chapman JT, Brantly ML, Stocks JM, Brown KK, Lynch JP 3rd, Goldberg HJ, Young LR, Kinder BW, Downey GP, Sullivan EJ, Colby TV, McKay RT, Cohen MM, Korbee L, Taveira-DaSilva AM, Lee HS, Krischer JP, Trapnell BC
  • Journal Title: N Engl J Med Volume: 364(17) Pages: 1595-6061
  • Peer Reviewed
• [Journal Article] Assembly Head of ILD (2011)
  • Author(s): Inoue Y
  • Journal Title: APSR. APSR RESPIRATORY UPDATES(APSR EDUCATION PUBLICATION) Volume: 3(5)
  • Peer Reviewed
• [Journal Article] Polymyxin-B Hemoperfusion for Acute Exacerbation of Idiopathic Pulmonary Fibrosis : Serum IL-7 as a Prognostic Marker (2011)
  • Author(s): Tachibana K, Inoue Y, Nishiyama A, Sugimoto C, Matsumuro A, Hirose M, Kitaichi M, Akira M, Arai T, Hayashi S, Inoue Y
  • Journal Title: Sarcoidosis vasculitis and diffuse lung diseases Volume: 28(2) Pages: 113-22
  • Peer Reviewed
• [Journal Article] 特定疾患治療研究事業対象疾患リンパ脈管筋腫症(LAM)認定基準の解説-厚生労働省難治性疾患克服研究事業呼吸不全に関する調査研究班 (2011)
  • Author(s): 林田美江,瀬山邦明,井上義一,平井豊博,三嶋理晃
  • Journal Title: 日呼吸会誌 Volume: 49(2) Pages: 67-74
  • Peer Reviewed
• [Journal Article] リンパ脈管筋腫症(LAM)の治療 (2011)
  • Author(s): 井上義一
  • Journal Title: Annual Review呼吸器2011 Pages: 216-222
• [Journal Article] 特定疾患治療研究事業対象疾患リンパ脈管筋腫症(LAM)認定基準の解説 (2011)
  • Author(s): 林田美江,瀬山邦明,井上義一,平井豊博,三嶋理晃
  • Journal Title: 日本呼吸器学会雑誌 Volume: 49(2) Pages: 67-74
  • Peer Reviewed
• [Journal Article] 全肺洗浄を施行した自己免疫性肺胞蛋白症8例の手技,効果,安全性の多面的評価 (2011)
  • Author(s): 杉本親寿,新井徹,西山明秀,井上康,香川智子,審良正則,松室昭子,広瀬雅樹,北市正則,林清二,井上義一
  • Journal Title: 日本呼吸器学会雑誌 Volume: 49(8) Pages: 569-576
  • Peer Reviewed
• [Journal Article] LAMとその周辺に対する新規分子標的治療薬の展望 (2011)
  • Author(s): 井上義一,新井徹,杉本親寿,広瀬雅樹
  • Journal Title: 日本胸部臨床 Volume: 70(10) Pages: 1031-1039
• [Journal Article] Inhaled granulocyte/macrophage-colony stimulating factor as therapy for pulmonary alveolar proteinosis (2010)
  • Author(s): Tazawa R, Trapnell BC, Inoue Y, Arai T, Takada T, Nasuhara Y, Hizawa N, Kasahara Y, Tatsumi K, Hojo M, Ishii H, Yokoba M, Tanaka N, Yamaguchi E, Eda R, Tsuchihashi Y, Morimoto K, Akira M, Terada M, Otsuka J, Ebina M, Kaneko C, Nukiwa T, Krischer JP, Akazawa K, Nakata K
  • Journal Title: Am J Respir Crit Care Med Volume: 181(12) Pages: 1345-54
  • Peer Reviewed
• [Journal Article] Serum Vascular Endothelial Growth Factor-D Prospectively Distinguishes Lymphangioleiomyomatosis from Other Diseases (2010)
  • Author(s): Young LR, Vandyke R, Gulleman PM, Inoue Y, Brown KK, Schmidt LS, Linehan WM, Hajjar F, Kinder BW, Trapnell BC, Bissler JJ, Franz DN, McCormack FX
  • Journal Title: Chest Volume: 138(3) Pages: 674-81
  • Peer Reviewed
• [Journal Article] The international LAM registry : a component of an innovative web-based clinician, researcher, and patient-driven rare disease research platform (2010)
  • Author(s): Nurok M, Eslick I, Carvalho CR, Costabe lU, D' Armiento J, Glanville AR, Harari S, Henske EP, Inoue Y, Johnson SR, Lacronique J, Lazor R, Moss J, Ruoss SJ, Ryu JH, Seyama K, Watz H, Xu KF, Hohmann EL, Moss F
  • Journal Title: Lymphat Res Biol Volume: 8(1) Pages: 81-87
  • Peer Reviewed
• [Journal Article] 稀少肺疾患に対する薬剤開発の現状と課題 (2010)
  • Author(s): 井上義一
  • Journal Title: THE LUNG perspectives Volume: Vol.18, No.2 Pages: 49-51
• [Journal Article] 乳び胸,診断,治療と病態 (2010)
  • Author(s): 杉本親寿,井上義一
  • Journal Title: 呼吸器内科 Volume: 17(4) Pages: 398-404
• [Journal Article] 『肺胞蛋白症』病態解明への新展開(up-date)特集びまん性肺疾患診療の進歩 (2010)
  • Author(s): 井上義一
  • Journal Title: 成人病と生活習慣病 Volume: Vol.40, No.6 Pages: 704-707
• [Journal Article] リンパ脈管筋腫症に関する分子生物学的解析 (2010)
  • Author(s): 広瀬雅樹,井上義一
  • Journal Title: 最新医学65巻9月増号(2010) Pages: 2102-2110
• [Journal Article] LAMの内科的治療と管理わが国の手引きと欧州呼吸器学会ガイドライン-特集リンパ脈管筋腫症(LAM)の新展開 (2010)
  • Author(s): 井上義一
  • Journal Title: 呼吸と循環 Volume: Vol.58, No.12 Pages: 217-1224
• [Journal Article] Comparative Study of High-Resolution CT Findings between Autoimmune and Secondary Pulmonary Alveolar Proteinosis (2009)
  • Author(s): Ishii H, Trapnell BC, Tazawa R, Inoue Y, Akira M, Kogure Y, Tomii K, Takada T, Hojo M, Ichiwata T, Goto H, Nakata K
  • Journal Title: Chest Volume: 136 Pages: 1348-1355
  • Peer Reviewed
• [Journal Article] Usual interstitial pneumonia and nonspecific interstitial pneumonia with and without concurrent emphysema : thin-section CT findings (2009)
  • Author(s): Akira M, Inoue Y, Kitaichi M, Yamamoto S, Arai T, Toyokawa K
  • Journal Title: Radiology Volume: 251(1) Pages: 271-9
  • Peer Reviewed
• [Journal Article] Quatitative CT in Chronic Obstructive Pulmonary Disease : Inspiratory and Expiratory Assessment (2009)
  • Author(s): Akira M, Toyokawa K, Inoue Y, Arai T
  • Journal Title: AJR Volume: 192 Pages: 1-6
  • Peer Reviewed
• [Presentation] Serum CYFRA reflects disease severity of autoimmune pulmonary alveolar proteinosis, Session 89, Thematic Poster Session : Rare diffuse lung diseases (2010)
  • Author(s): T. Arai, Y. Inoue, C. Sugimoto, A. Matsumuro, Y. Iwaki, M. Hirose, M. Kitaichi, M. Akira, S. Hayashi, K. Nakata, M. Sakatani
  • Organizer: ERS 2010, Fira Barcelona.
  • Place of Presentation: Barcelona, Spain
  • Year and Date: 20100918-22
• [Presentation] Quantitative CT in pulmonary lymphangioleiomyomatosis : Comparisonwith pulmonary dysfunction, Session 89, Thematic Poster Session : Rare diffuse lung diseases (2010)
  • Author(s): Y. Inoue, M. Akira, M. Hirose, T. Arai, T. Okuma, Y. Iwaki, C. Sugimoto, K. Tachibana, M. Akiko, M. Nakagawa, M. Kitaichi, S. Hayashi, M. Okada, M. Sakatani
  • Organizer: ERS 2010, Fira Barcelona.
  • Place of Presentation: Barcelona, Spain
  • Year and Date: 20100918-22
• [Presentation] Serum lymphangiogenic/angiogenic factors in lymphangioleiomyomatosis, as diagnostic biomarkers (2010)
  • Author(s): Yoshikazu Inoue
  • Organizer: The LAM Foundation 2010 International Research Conference
  • Place of Presentation: Cincinnati Ohio, USA
  • Year and Date: 2010-04-12
• [Presentation] Don't Miss Rare Diffuse Parenchymal Lung Disease : Pulmonary Alveolar Proteinosis Update (2009)
  • Author(s): Yoshikazu Inoue
  • Organizer: 14^<th> Congress of the APSR and 3^<rd> Joint Congress of the APSR/ACCP
  • Place of Presentation: COEX, Seoul, Korea.
  • Year and Date: 20091114-18
• [Presentation] Serum Vascular Endothelial Growth Factor(VEGF)-D in lymphangioleiomyomatosis(LAM) (2009)
  • Author(s): Masaki Hirose, Yoshikazu Inoue, Akiko Matsumuro, Toru Arai, Chikatoshi Sugimoto, Kazunobu Tachibana, Akihide Nishiyama, Seiji. Hayashi, Masaji Okada, Mitsunori Sakatani
  • Organizer: 14^<th> Congress of the APSR and 3^<rd> Joint Congress of the APSR/ACCP
  • Place of Presentation: COEX, Seoul, Korea
  • Year and Date: 20091114-18
• [Presentation] Japanese Center of RLDC, Osaka Symposium on Respiratory Disease. Intercurrent intractable factors in autoimmune pulmonary alveolar proteinosis(Thematic Poster Session : Various 5 backgrounds in diffuse parenchymal Lung P. 757) (2009)
  • Author(s): Y. Inoue, T. Arai, M. Kitaichi, M. Akira, K. Nakata, R. Tazawa, J. Otsuka, M. Hirose, A. Matsumuro, M. Sakatani
  • Organizer: ERS Vienna 2009 Austria
  • Place of Presentation: Sakai, Niigata, Japan
  • Year and Date: 20090912-16
• [Presentation] Profile of Th1/Th2 Cytokines in Autoimmune Pulmonary Alveolar Proteinosis (2009)
  • Author(s): Y Inoue, A Matsumuro, M Hirose, T Arai, J Otsuka, C Sugimoto, S Minamoto, A Nishiyama, K Sugiyama, M Nakagawa, M Kitaichi, M Akira, S Hayashi, K Nakata, M Okada, and M Sakatani
  • Organizer: ATS 2009 San Diego International conference
  • Place of Presentation: San Diego, California, USA
  • Year and Date: 20090515-20
• [Presentation] Impact of Th2 cytokines in Lymphangioleiomyomatos (2009)
  • Author(s): M Hirose, Y Inoue, A Matsumuro, T Arai, C Sugimoto, S Minamoto, K Tachibana, M Akira, M Kitaichi, S Hayashi, K Sugiyama, M Nakagawa, M Okada, and M Sakatani
  • Organizer: ATS 2009 San Diego International conference
  • Place of Presentation: San Diego, California, USA
  • Year and Date: 20090515-20
• [Presentation] Serum lymphangiogenic/angiogenic factors in lymphangioleiomyomatosis, as diagnostic biomarkers (2009)
  • Author(s): Yoshikazu Inoue
  • Organizer: The LAM Foudation/NHLBI 2009 International Research Conference
  • Place of Presentation: Ohio, USA
  • Year and Date: 20090417-19
• [Presentation] Clinical Features of Disorders of Surfactant Clearance : East Meets West (2009)
  • Author(s): Y. Inoue
  • Organizer: 3^<rd>. Congresso Internazionale Pavia, Collegio Cardinal Riboldi
  • Place of Presentation: Pavia, Italy
  • Year and Date: 20090330-0401
• [Book] 肺胞蛋白症-呼吸器疾患最新の治療 (2012)
  • Author(s): 井上義一
  • Total Pages: 331-334
  • Publisher: 南光堂
• [Book] リンパ脈管筋腫症(LAM)の治療, LAMの病態, LAMの治療,具体的薬物療法 Annual Review呼吸器2011 (2011)
  • Author(s): 井上義一
  • Total Pages: 216-222
  • Publisher: 中外医学社(株)
• [Book] Autoimmune pulmonary alveolar proteinosis. Molecular Basis of Pulmonary Disease Insights from Rare Lung Disorders (2010)
  • Author(s): Bruce C. Trapnell, Koh Nakata, and Yoshikazu Inoue
  • Publisher: Humana Press, Springer New York Dordrecht Heidelberg London
• [Book] Lymphangioleio-myomatosis. Murray & Nadel's textbook of respiratory medicine (2010)
  • Author(s): McCormack FX, Sullivan EJ, Inoue Y.
  • Total Pages: 1496-1515
  • Publisher: Saunders Elsevier
• [Book] リンパ脈管筋腫症-看護学テキスト疾病と治療I (2010)
  • Author(s): 井上義一
  • Total Pages: 94
  • Publisher: 南江堂