Elucidation of the pathogenesis of Langerhans cell histiocytosis from the point of bone immunology

• Project/Area Number: 22591167
• Research Category: Grant-in-Aid for Scientific Research (C)
• Allocation Type: Single-year Grants
• Section: 一般
• Research Field: Pediatrics
• Research Institution: Jichi Medical University
• Principal Investigator: MORIMOTO Akira 自治医科大学, 医学部, 教授 (30326227)
• Co-Investigator(Kenkyū-buntansha): NAKAMURA Sachie 自治医科大学, 医学部, 助教 (20382955) ; HAYASE Tomomi 自治医科大学, 医学部, 助教 (50433587) ; HAYAKAWA Takahiro 自治医科大学, 医学部, 大学院生 (60458312) ; OH Yukiko 自治医科大学, 医学部, 大学院生 (30438650)
• Project Period (FY): 2010 – 2012
• Project Status: Completed (Fiscal Year 2012)
• Budget Amount: ¥4,420,000 (Direct Cost: ¥3,400,000、Indirect Cost: ¥1,020,000); Fiscal Year 2012: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000); Fiscal Year 2011: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000); Fiscal Year 2010: ¥1,560,000 (Direct Cost: ¥1,200,000、Indirect Cost: ¥360,000)
• Keywords: 小児血液学 / ランゲルハンス細胞組織球症 / 未熟樹状細胞 / 破骨細胞 / osteopontin / integrin / CD44 / integrin α V

Research Abstract

LCH cells have characteristics of immature dendritic cell (iDC), which differentiate into osteoclasts in inflammatory environment. Osteopontin has been known as a proinflammatory cytokine, which is involved in bone resorption by osteoclasts. Here we show

Research Products

• [Journal Article] Langerhans cell histiocytosis case with dense metaphyseal band sign (2013)
  • Author(s): Kikkawa I, Aihara T, Morimoto A, Watanabe H, Furukawa R
  • Journal Title: Pediatr Int Volume: 55 Pages: 96-98
• [Journal Article] IL-17A receptor expression differs between subclasses of Langerhans cell histiocytosis, which might settle the IL-17A controversy. (2013)
  • Author(s): Murakami I, Morimoto A, Oka T, Kuwamoto S, Kato M, Horie Y, Hayashi K, Gogusev J, Jaubert F, Imashuku S, Al-Kadar LA, Takata K, Yoshino T
  • Journal Title: Virchows Arch. Volume: 462 Pages: 219-228
• [Journal Article] Therapeutic outcome of multifocal Langerhans cell histiocytosis in adults treated with the Special C regimen t formulated by the Japan LCH Study Group (2013)
  • Author(s): Morimoto A, Shimazaki C, Takahashi S, Yoshikawa K, Nishimura R, Wakita H, Kobayashi Y, Kanegane H, Tojo A, Imamura T, Imashuku S.
  • Journal Title: Int J Hematol. Volume: 97 Pages: 103-108
• [Journal Article] Rapidly progressing fatal adult multi-organ langerhans cell histiocytosis complicated with Fatty liver disease. (2012)
  • Author(s): Yuasa M, Fujiwara S, Oh I, Yamaguchi T, Fukushima N, Morimoto A, Ozawa K
  • Journal Title: J Clin Exp Hematop Volume: 52 Pages: 121-126
  • NAID: 10031120436
• [Journal Article] Treatment of patients with hypothalamic-pituitary lesions as adult-onset Langerhans cell histiocytosis. (2011)
  • Author(s): Imashuku S, Kudo N, Kaneda S, Kuroda H, Shiwa T, Hiraiwa T, Inagaki A, Morimoto A.
  • Journal Title: Int J Hematol Volume: 94 Pages: 556-560
  • NAID: 10030266236
• [Journal Article] for the LCH Committee, the Japanese Pediatric Leukemia/Lymphoma Study Group. Nationwide Survey of Bisphosphonate Therapy for Children with Reactivated Langerhans Cell Histiocytosis in Japan (2011)
  • Author(s): Morimoto A, Shioda Y, Imamura T, Kanegane H, Sato T, Kudo K, Nakagawa S, Nakadate H, Tauchi H, Hama A, Yasui M, Nagatoshi Y, Kinoshita A, Miyaji R, Anan T, Yabe M, Kamizono J
  • Journal Title: Pediatr Blood Cancer Volume: 56 Pages: 110-115
• [Journal Article] Tyrosine phosphatase SHP-1 is expressed higher in multisystem than in single-system Langerhans cell histiocytosis by immunohistochemistry. (2011)
  • Author(s): Murakami I, Oka T, Kuwamoto S, Kato M, Hayashi K, Gogusev J, Imamura T, Morimoto A, Imashuku S, Yoshino T.
  • Journal Title: Virchows Arch Volume: 459 Pages: 227-234
• [Journal Article] Analysis of 43 cases of Langerhans cell histiocytosis (LCH)-induced central diabetes insipidus registered in the JLSG-96 and JLSG-02 studies in Japan (2011)
  • Author(s): Shioda Y, Adachi S, Imashuku S, Kudo K, Imamura T, Morimoto A
  • Journal Title: Int J Hematol Volume: 94 Pages: 545-551
  • NAID: 10030266204
• [Journal Article] Treatment of patients with hypothalamic-pituitary lesions as adult-onset Langerhans cell histiocytosis (2011)
  • Author(s): Imashuku S, Kudo N, Kaneda S, Kuroda H, Shiwa T, Hiraiwa T, Inagaki A, Morimoto A
  • Journal Title: Int J Hematol Volume: 94 Issue: 6 Pages: 556-560
  • DOI: 10.1007/s12185-011-0955-z
  • NAID: 10030266236
  • Peer Reviewed
• [Journal Article] Nationwide survey of bisphosphonate therapy for children with reactivated Langerhans cell histiocytosis in Japan (2011)
  • Author(s): Morimoto A, Shioda Y, Imamura T, Kanegane H, Sato T, Kudo K, Nakagawa S, Nakadate H, Tauchi H, Hama A, Yasui M, Nagatoshi Y, Kinoshita A, Miyaji R, Anan T, Yabe M, Kamizono J
  • Journal Title: Pediatr Blood Cancer Volume: 56 Issue: 1 Pages: 110-115
  • DOI: 10.1002/pbc.22703
  • Peer Reviewed
• [Journal Article] Tyrosine phosphatase SHP-1 is expressed higher in multisystem than in single-system Langerhans cell histiocytosis by immunohistochemistry (2011)
  • Author(s): Murakami I, Oka T, Kuwamoto S, Kato M, Hayashi K, Gogusev J, Imamura T, Morimoto A, Imashuku S, Yoshino T
  • Journal Title: Virchows Arch Volume: 459 Issue: 2 Pages: 227-234
  • DOI: 10.1007/s00428-011-1090-1
  • Peer Reviewed
• [Journal Article] Nationwide survey of bisphosphonate therapy for children with reactivated Langerhans cell histiocytosis in Japan. (2011)
  • Author(s): Morimoto A.
  • Journal Title: Pediatric Blood and Cancer Volume: 56 Pages: 110-115
• [Journal Article] Outcome of pediatric patients with Langerhans cell histiocytosis treated with 2 chlorodeoxyadenosine: a nationwide survey in Japan (2010)
  • Author(s): Imamura T, Sato T, Shiota Y, Kanegane H, Kudo K, Nakagawa S, Nakadate S, Tauchi H, Kamizono J, Morimoto A
  • Journal Title: Int J Hematol Volume: 91 Pages: 646-651
  • NAID: 10026402057
• [Journal Article] VCR/AraC chemotherapy and ND-CNS-LCH (2010)
  • Author(s): Imashuku S, Shioda Y, Tsunematsu Y, Imamura T, Morimoto A.
  • Journal Title: Pediatr Blood Cancer Volume: 55 Pages: 215-216
• [Journal Article] Improved outcome of refractory Langerhans cell histiocytosis in children with hematopoietic stem cell transplantation in Japan (2010)
  • Author(s): Kudo K, Ohga S, Morimoto A, Ishida Y, Suzuki N, Hasegawa D, Nagatoshi Y, Kato S, Ishii E.
  • Journal Title: Bone Marrow Transplant Volume: 45 Pages: 901-906
• [Journal Article] Outcome of pediatric patients with Langerhans cell histiocytosis treated with 2 chlorodeoxyadenosine : a nationwide survey in Japan. (2010)
  • Author(s): Imamura T.
  • Journal Title: International Journal of Hematology Volume: 91 Pages: 46-51
• [Journal Article] VCR/AraC chemotherapy and ND-CNS-LCH. (2010)
  • Author(s): Imashuku S.
  • Journal Title: Pediatric Blood and Cancer Volume: 55 Pages: 215-216
• [Presentation] ランゲルハンス細胞組織球症における血清オステオポンチンの解析 (2013)
  • Author(s): 翁 由紀子, 森本 哲
  • Organizer: 第35回日本 LCH 研究会
  • Place of Presentation: 東京
• [Presentation] Intensified and prolonged therapy improved the outcome in multi-system Langerhans cell histiocytosis. (2012)
  • Author(s): Morimoto A, Shioda Y, Imamura T, Kudo K, Sato T, Shiohara M, Yasui M, Koga Y, Kobayashi R, Ishii E, Fujimoto J, Horibe K, Bessho F, Tsunematsu Y, Imashuku S
  • Organizer: 28th Annual Meeting of the Histiocyte Society.
  • Place of Presentation: London UK
• [Presentation] A case of myelodysplastic syndrome after 2-chlorodeoxyadenosine therapy for recurrent Langerhans cell histiocytosis. (2012)
  • Author(s): Oh Y, Morimoto A, Itoh T, Masuzawa A, Kashii Y, Gunji Y, Momoi MY
  • Organizer: 28th Annual Meeting of the Histiocyte Society
  • Place of Presentation: London UK
• [Presentation] Intensified and prolonged therapy improved the outcome in multi-system Langerhans cell histiocytosis; results of JLSG-02 protocol study. (2012)
  • Author(s): 森本 哲
  • Organizer: 第54回日本小児血液・がん学会
  • Place of Presentation: 横浜
• [Presentation] Preliminary result of comparative study on Korean and Japanese patients with multisystem -LCH : Part 2. Treatment and outcomes (2011)
  • Author(s): Morimoto A
  • Organizer: International Symposium of Rare Diseases
  • Place of Presentation: Seoul, Korea
  • Year and Date: 2011-12-08
• [Presentation] Multifocal LCH in adult patients treated with special C regimen of JLSG-02 protocol (2011)
  • Author(s): Morimoto A, Shimazaki C, Takahashi S, Yoshikawa K, Kuroda R, Wakiya H, Kobayashi H, Kanegane H, Akiyama S, Tojyo A, Imamura T, Imashuku S
  • Organizer: 27th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Viena Austria
  • Year and Date: 2011-10-17
• [Presentation] Gene expression profiling of PTPN6 (SHP-1) transfected Langerhas cell-like cell line (ELD-1)-compared with open data of LCH subtype (GSE16395) (2011)
  • Author(s): Murakami I, Oka T, Hayashi K, Gogusev J, Jaubert F, Morimoto A, Imashuku S, Yoshino T
  • Organizer: 27th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Viena Austria
  • Year and Date: 2011-10-17
• [Presentation] Recent advance in Langerhans cell histiocytosis (2011)
  • Author(s): Morimoto A
  • Organizer: 52nd Spring Meeting of the Korean Society of Hematology
  • Place of Presentation: Seoul, Korea
  • Year and Date: 2011-05-28
• [Presentation] Recent advance in Langerhans cell histiocytosis. (2011)
  • Author(s): Morimoto A
  • Organizer: 52nd Spring Meeting of the Korean Society of Hematology Abstract
  • Place of Presentation: Seoul Korea
• [Presentation] Multifocal LCH in adult patients treated with special C regimen of JLSG-02 protocol (2011)
  • Author(s): Morimoto A, Shimazaki C, Takahashi S, Yoshikawa K, Kuroda R, Wakiya H, Kobayashi H, Kanegane H, Akiyama S, Tojyo A, Imamura T, Imashuku S.
  • Organizer: 27th Annual Meeting of the Histiocyte Society.
  • Place of Presentation: Vienna Austria
• [Presentation] Gene expression profiling of PTPN6 (SHP-1) transfected Langerhas cell-like cell line (ELD-1) -compared with open data of LCH subtype (GSE16395). (2011)
  • Author(s): Murakami I, Oka T, Hayashi K, Gogusev J, Jaubert F, Morimoto A, Imashuku S, Yoshino T.
  • Organizer: 27th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Vienna Austria
• [Presentation] Preliminary result of comparative study on Korean and Japanese patients with multisystem-LCH: Part 2. Treatment and outcomes. (2011)
  • Author(s): Morimoto A.
  • Organizer: International Symposium of Rare Diseases
  • Place of Presentation: Seoul Korea
• [Presentation] LCHの病態解明と治療の進歩 (2010)
  • Author(s): 森本哲
  • Organizer: 第52回日本小児血液学会
  • Place of Presentation: 大阪(教育講演)
  • Year and Date: 2010-12-18
• [Presentation] Outcome of poor responders at 6 weeks of induction therapy : JLSG-02protocol study for multisystem Langerhans cell histiocytosis in Japan. (2010)
  • Author(s): Kudo K.
  • Organizer: 26th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Boston
  • Year and Date: 2010-10-19
• [Presentation] Comprehensive analysis of serum cytokine/ chemokine and growth factors in pediatric patients with Langerhans cell histiocytosis. (2010)
  • Author(s): Morimoto A.
  • Organizer: 26th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Boston
  • Year and Date: 2010-10-18
• [Presentation] A case of case of Langerhans cell histiocytosis with a recurrent CNS masslesion successfully treated with 2-chlorodeoxyadenosine. (2010)
  • Author(s): Oh Y.
  • Organizer: 26th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Boston
  • Year and Date: 2010-10-18
• [Presentation] JLSG-02に登録された成人例の多病変型ランゲルハンス細胞組織球症(LCH)の中間報告 (2010)
  • Author(s): 森本哲
  • Organizer: 第72回日本血液学会
  • Place of Presentation: 横浜
  • Year and Date: 2010-09-24
• [Presentation] JLSG-02 に登録された成人例の多病変型ランゲルハンス細胞組織球症(LCH)の中間報告 (2010)
  • Author(s): 森本 哲, 島崎千尋, 高橋 聡, 吉川浩平, 黒田梨恵, 脇田 久, 小林 裕, 金兼弘和, 秋山聖子, 今宿晋作
  • Organizer: 第72回日本血液学会
  • Place of Presentation: 横浜
• [Presentation] Comprehensive analysis of serum cytokine/ chemokine and growth factors in pediatric patients with Langerhans cell histiocytosis. (2010)
  • Author(s): Morimoto A, Nakamura S, Shioda Y, Imamura T, Oh Y, Imashuku S, Momoi M
  • Organizer: 26th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Boston USA
• [Presentation] A case of case of Langerhans cell histiocytosis with a recurrent CNS mass lesion successfully treated with 2-chlorodeoxyadenosine. (2010)
  • Author(s): Oh Y, Morimoto A, Itoh T, Kashii Y, Gunji Y, Momoi M.
  • Organizer: 26th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Boston USA
• [Presentation] Outcome of poor responders at 6 weeks of induction therapy: JLSG-02 protocol study for multisystem Langerhans cell histiocytosis in Japan. (2010)
  • Author(s): Kudo K, Morimoto A, Imamura T, Shioda Y, Sato T, Ishii E, Horibe K, Kinugawa N, Tsunematsu Y, Imashuku S.
  • Organizer: 26th Annual Meeting of the Histiocyte Society
  • Place of Presentation: Boston USA
• [Presentation] LCH の病態解明と治療の進歩 (2010)
  • Author(s): 森本 哲
  • Organizer: 第52回日本小児血液学会
  • Place of Presentation: 大阪
• [Book] Management of Langerhans cell histiocytosis (LCH) -induced central diabetes insipidus and its associated endocrinological/ neurological sequelae (2011)
  • Author(s): Imashuku S, Morimoto A
  • Publisher: Diabetes Insipidus, Croatia: InTech - Open Access Publisher
• [Book] ランゲルハンス細胞組織球症 (2011)
  • Author(s): 翁 由紀子, 森本 哲
  • Publisher: 医薬ジャーナル社
• [Book] 小児がん診療ハンドブック(各論7.ランゲルハンス細胞組織球症) (2011)
  • Author(s): 翁由紀子、森本哲(分担執筆)
  • Publisher: 医薬ジャーナル社,大阪
• [Book] Diabetes Insipidus (Chapter 1. Management of Langerhans Cell Histiocytosis (LCH)-induced central diabetes insipidus and its associated endocrinological/neurological sequelae) (2011)
  • Author(s): Imashuku S, Morimoto A
  • Publisher: InTech-Open Access Publisher, Croatia
• [Book] ランゲルハンス細胞組織球増殖症 (2010)
  • Author(s): 村上一郎, 林 一彦, 森本 哲
  • Publisher: 中山書店
• [Book] リンパ球増殖疾患 癌診療指針のための病理診断プラクティス(第3章 リンパ球増殖疾患の鑑別診断 ランゲルハンス細胞組織球増殖症) (2010)
  • Author(s): 村上一郎、林一彦、森本哲(分担執筆)
  • Publisher: 中山書店
• [Remarks]
  • URL: http://www.jlsg.jp/
• [Remarks]
  • URL: http://www.jlsg.jp/