International clinical research for secondary pulmonary alveolar proteinosis complicated with myelodysplastic syndrome

• Project/Area Number: 26305028
• Research Category: Grant-in-Aid for Scientific Research (B)
• Allocation Type: Partial Multi-year Fund
• Section: 海外学術
• Research Field: Respiratory organ internal medicine
• Research Institution: Kyorin University
• Principal Investigator: Ishii Haruyuki 杏林大学, 医学部, 准教授 (30406970)
• Co-Investigator(Kenkyū-buntansha): 中田 光 新潟大学, 医歯学総合病院, 教授 (80207802) ; 井上 義一 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, その他 (90240895) ; 田澤 立之 新潟大学, 医歯学総合病院, 准教授 (70301041) ; 皿谷 健 杏林大学, 医学部, 助教 (40549185)
• Project Period (FY): 2014-04-01 – 2017-03-31
• Project Status: Completed (Fiscal Year 2016)
• Budget Amount: ¥15,600,000 (Direct Cost: ¥12,000,000、Indirect Cost: ¥3,600,000); Fiscal Year 2016: ¥4,810,000 (Direct Cost: ¥3,700,000、Indirect Cost: ¥1,110,000); Fiscal Year 2015: ¥5,850,000 (Direct Cost: ¥4,500,000、Indirect Cost: ¥1,350,000); Fiscal Year 2014: ¥4,940,000 (Direct Cost: ¥3,800,000、Indirect Cost: ¥1,140,000)
• Keywords: 肺胞蛋白症 / 骨髄異形成症候群 / 呼吸器内科 / 血液内科 / 内科 / ステロイド / 臨床 / ドイツ

Outline of Final Research Achievements

We investigated the clinical research for secondary pulmonary alveolar proteinosis complicated with myelodysplastic syndrome. There was little clinical information for this disease because SPAP complicated with MDS was extremely rare lung disease. We demonstrated the following outcomes, based on the results from our study. 1)Prevalence of SPAP complicated with MDS in Japan was significant higher than it in Europe. 2)The prognosis of SPAP complicated with MDS was very poor after diagnosis of SPAP(MST: 15 months). 3)It is the most important risk factor for the prognosis of SPAP was the use of steroid therapy at diagnosis of SPAP, and its cox hazard ratio for the death was 2.43. These outcomes will contribute to the medical treatment and new study for SPAP complicated with MDS. Moreover it is good outcome to be able to constitite the international SPAP network.

Research Products

• [Int'l Joint Research] エッセン大学/デュッセルドルフ大学(ドイツ)
• [Int'l Joint Research] Peter MacCallum Cancer Centre(オーストラリア)
• [Int'l Joint Research] パビア大学(イタリア)
• [Journal Article] 骨髄異形成症候群と肺胞マクロファージー続発性肺胞蛋白症を中心にー (2016)
  • Author(s): 石井晴之
  • Journal Title: 日本サーファクタント・界面医学界雑誌 Volume: 47 Pages: 17-19
• [Journal Article] Pulmonary alveolar proteinosis-like change: A fairly common reaction asociated with the severity of idiopathic pulmonary fibrosis. (2016)
  • Author(s): Nunomura S, Tanaka T, Nakayama T, Otani K, Ishii H, Tabata K, Kondoh Y, Kataoka K, Johkoh T, Taniguchi H, Fukuoka J.
  • Journal Title: Respiratory Investigation Volume: 54 Issue: 4 Pages: 272-279
  • DOI: 10.1016/j.resinv.2016.02.004
  • Peer Reviewed / Open Access
• [Journal Article] 続発性肺胞蛋白症 最新の知見と今後の課題 (2016)
  • Author(s): 石井晴之
  • Journal Title: 日本胸部臨床 Volume: 75 (11) Pages: 1126-35
  • Peer Reviewed
• [Journal Article] Clinical significance of the galaxy sign in patients with pulmonary sarcoidosis in a Japanese single-center cohort. (2016)
  • Author(s): Koide T, Saraya T, Tsukahara Y, Bonella F, Borner E, Ishida M, Ogawa Y, Hirukawa I, Oda M, Shimoda M, Ohkuma K, Fujiwara M, Takata S, Yokoyama T, KUrai D, Ishii H, Goto H, Takizawa H.
  • Journal Title: Sarcoidosis Vascular and Diffuse Lung Disease Volume: 33 Pages: 247-252
  • Peer Reviewed / Int'l Joint Research
• [Journal Article] A new diagnostic approarch for bilious pleural effusion. (2016)
  • Author(s): Saraya T, Light RW, Sakuma S, Nakamoto Y, Wada S, Ishida M, Inui T, Koide T, Ishii H, Takizawa H.
  • Journal Title: Respiratory Investigation Volume: 54 Issue: 5 Pages: 364-68
  • DOI: 10.1016/j.resinv.2016.03.009
  • Peer Reviewed / Open Access / Int'l Joint Research
• [Journal Article] Molecular evolution of the fusion protein gene in human respiratory syncytial virus subgroup A. (2016)
  • Author(s): Kimura H, Nagasawa K, Tsugagoshi H, Matsushima Y, Fujita K, Yoshida LM, Tabnaka R, Ishii H, Shimojo N, Kuroda M, Ryu A.
  • Journal Title: Infect Genet Evol Volume: 9 Pages: 398-406
  • DOI: 10.1016/j.meegid.2016.06.019
  • Peer Reviewed / Open Access
• [Journal Article] Two cases of autoimmune and secondary pulmonary alveolar proteinosis during immunosuppressive therapy in dermatomyositis with interstitial lung disease. (2016)
  • Author(s): Imura Y, Yukawa N, Handa T, Nakashima R, Murakami K, Yoshifuji H, Ohmura K, Ishii H, Nakata K, Mimori T.
  • Journal Title: Mod Rheumatol Volume: 11 Issue: 4 Pages: 1-6
  • DOI: 10.3109/14397595.2016.1153443
  • Peer Reviewed / Open Access / Int'l Joint Research
• [Journal Article] Outcome of corticosteroid administration in autoimmune pulmonary alveolar proteinosis: a retrospective cohort study (2015)
  • Author(s): Keiichi Akasaka, Takahiro Tanaka, Nobutaka Kitamura, Shinya Ohkouchi, Ryushi Tazawa, Toshinori Takada, Toshio Ichiwata, Etsuro Yamaguchi, Masaki Hirose, Toru Arai, Kentaro Nakano, Takahito Nei, Haruyuki Ishii, Tomohiro Handa, Yoshikazu Inoue and Koh Nakata
  • Journal Title: BMC Pulmonary Medicine Volume: 15 Issue: 1 Pages: 88-88
  • DOI: 10.1186/s12890-015-0085-0
  • NAID: 120005758416
  • Peer Reviewed / Open Access / Int'l Joint Research
• [Journal Article] Possible Involvement of Lung Cells Harboring an Abnormal Karyotype in the Pathogenesis of Pulmonary Alveolar Proteinosis Associated with Myelodysplastic Syndrome (2015)
  • Author(s): Moriyama M, Yano T, Furukawa T, Takada T, Ushiki T, Masuko M, Takizawa J, Sone H, Tazawa R, Saijo Y, Ishii H, Nakata K.
  • Journal Title: Ann Am Thorac Soc Volume: 12 (8) Pages: 1251-1253
  • Open Access / Int'l Joint Research
• [Journal Article] 続発性肺胞蛋白症の臨床的特徴―全国調査結果と更なる課題― (2014)
  • Author(s): 石井晴之
  • Journal Title: 日本肺サーファクタント・界面医学会雑誌 Volume: 45 Pages: 13-15
  • Peer Reviewed
• [Journal Article] Spontaneous resolution of pneumocystis jirovecii pneumonia on high resolution computed tomography in a patient with renal cell carcinoma (2014)
  • Author(s): Tanaka Y, Saraya T, Kurai D, Ishii H, Takizawa H, Goto H
  • Journal Title: American Journal of Case Reports Volume: 15 Pages: 496-500
  • DOI: 10.12659/ajcr.890947
  • Peer Reviewed / Open Access
• [Presentation] Characteristics of secondary pulmonary alveolar proteinosis complicated with myelodysplastic syndrome in Japan. (2016)
  • Author(s): Ishii H, Tazawa R, Saraya T, Inoue Y, Nakata K.
  • Organizer: 21th congress of Asian Pacific Society of Respirology.
  • Place of Presentation: Bangkok, Thailand
  • Year and Date: 2016-11-12
  • Int'l Joint Research
• [Presentation] Clinical information of secondary pulmonary alveolar proteinosis: Follow-up report. (2016)
  • Author(s): Ishii H, Tazawa R, Inoue Y, Nakata K.
  • Organizer: Rare Lung Disease Consortium 2016
  • Place of Presentation: Cincinnati, USA
  • Year and Date: 2016-09-22
  • Int'l Joint Research
• [Presentation] Clinical infermation of secondary pulmonary alveolar proteinosis in Japan. (2016)
  • Author(s): Ishii H, Tazawa R, Inoue Y, Saraya T, Ishida M, Nakata K.
  • Organizer: American Thoracic Society 2016 International conference
  • Place of Presentation: San Francisco, USA
  • Year and Date: 2016-05-14
  • Int'l Joint Research
• [Presentation] Significance of high-resolution CT findings by using densitometry in patient with Pulmonary Alveolar Proteinosis (2015)
  • Author(s): Haruyuki Ishii
  • Organizer: 20th Congress of Asian Pacific Society of Respirology
  • Place of Presentation: クアラルンプール
  • Year and Date: 2015-12-03
  • Int'l Joint Research
• [Presentation] 骨髄異形成症候群と肺胞マクロファージ:続発性肺胞蛋白症を中心に (2015)
  • Author(s): 石井晴之
  • Organizer: 第51回学術研究会 日本肺サーファクタント-界面医学会
  • Place of Presentation: 大阪
  • Year and Date: 2015-10-31
  • Invited
• [Presentation] The analysis of alveolar macrophage form on BALF speciemens in secondary alveolar proteinosis. (2015)
  • Author(s): Haruyuki Ishii
  • Organizer: American Thoracic Society 2015
  • Place of Presentation: デンバー
  • Year and Date: 2015-05-15
  • Int'l Joint Research
• [Presentation] Clinical characteristics of secondary pulmonary alveolar proteinosis (sPAP) (2014)
  • Author(s): Ishii H, Tazawa R, Inoue Y, Nakata K
  • Organizer: 19th Congress of Asian Pacific Society of Respirology
  • Place of Presentation: Bali, Indonesia
  • Year and Date: 2014-11-13 – 2014-11-16
• [Presentation] Secondary pulmonary alveolar proteinosis complicating myelodysplastic syndrome results in a worsening of prognosis (2014)
  • Author(s): Ishii H, Tazawa R, Inoue Y, Mikura S, Yomota M, Nagatomo T, Inui T, Handa T, Hashimoto K, Tomii K, Nakata K
  • Organizer: American Thoracic Society 2014 International Conference
  • Place of Presentation: San Diego, USA
  • Year and Date: 2014-05-17 – 2014-05-21
• [Presentation] わが国における骨髄異形成症候群に続発した肺胞蛋白症の予後について (2014)
  • Author(s): 石井晴之、田澤立之、皿谷 健、倉井大輔、横山恵美、横山琢磨、滝澤 始、井上義一、
  • Organizer: 第54回日本呼吸器学会学術講演会
  • Place of Presentation: 大阪
  • Year and Date: 2014-04-25 – 2014-04-27
• [Book] 内科疾患の診断基準・病型分類・重症度 ANCA関連肺疾患 (2015)
  • Author(s): 石井晴之
  • Total Pages: 2
  • Publisher: 南江堂
• [Book] 間質性肺炎の臨床up-to-date 気管支肺胞洗浄検査はどのように解釈すべきでしょうか？ (2015)
  • Author(s): 石田学、石井晴之
  • Total Pages: 4
  • Publisher: 新興医学出版社
• [Book] New専門医を目指すケース・メソッド・アプローチ呼吸器疾患 (2015)
  • Author(s): 石井晴之
  • Total Pages: 8
  • Publisher: 日本医事新報社
• [Book] 胸膜疾患のすべて (2015)
  • Author(s): 石井晴之
  • Total Pages: 15
  • Publisher: 診断と治療社
• [Book] 今日の治療指針 私はこう治療している (2015)
  • Author(s): 石井晴之
  • Total Pages: 2
  • Publisher: 医学書院
• [Remarks] 続発性肺胞蛋白症：情報サイト
  • URL: http://spap-project.jp/
• [Funded Workshop] MDS-sPAP conference 2016 (2016)
  • Place of Presentation: 杏林大学、東京