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Etiology and pathology of Kimura disease (eosinophilic granuloma of soft tissue) with a predilection for Japanese young individuals

Research Project

Project/Area Number 20K08258
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Review Section Basic Section 52050:Embryonic medicine and pediatrics-related
Research InstitutionKochi University

Principal Investigator

FUJIEDA Mikiya  高知大学, 教育研究部医療学系臨床医学部門, 教授 (60209020)

Co-Investigator(Kenkyū-buntansha) 樋口 智紀  高知大学, 教育研究部医療学系基礎医学部門, 講師 (00448771)
橋田 裕美子  高知大学, 教育研究部医療学系基礎医学部門, 助教 (00767999)
大畑 雅典  高知大学, 教育研究部医療学系基礎医学部門, 教授 (50263976)
Project Period (FY) 2020-04-01 – 2023-03-31
Project Status Completed (Fiscal Year 2022)
Budget Amount *help
¥4,290,000 (Direct Cost: ¥3,300,000、Indirect Cost: ¥990,000)
Fiscal Year 2022: ¥1,300,000 (Direct Cost: ¥1,000,000、Indirect Cost: ¥300,000)
Fiscal Year 2021: ¥1,300,000 (Direct Cost: ¥1,000,000、Indirect Cost: ¥300,000)
Fiscal Year 2020: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
Keywordsウイルス / 炎症 / 皮下腫瘤
Outline of Research at the Start

木村病は頭頸部軟部組織に無痛性腫瘤を形成し、日本人の若年者に好発するという特徴をもつ。Ⅰ型アレルギーの関与が示唆されるが、原因はいまだ明らかにされていない。申請者らはこれまでに、木村病の病理組織からヒトポリオーマウイルス6型(HPyV6)のDNAとそのウイルス由来蛋白を高率に検出した。しかも、そのウイルス株は日本人特有の遺伝子型を有することを突き止めた。
本研究では、HPyV6と木村病の関連性を詳細に追及し、何故日本人に好発し、どのような個体に発生し易いのか、という問題の解決に挑む。また、HPyV6感染と木村病患者の臨床的特徴との相関性を明らかにし、木村病の病態の解明に資する研究を推進する。

Outline of Final Research Achievements

The prevalence of eight cutaneous human polyomaviruses (HPyVs) in Kimura disease was investigated, using biopsy specimens obtained from Japanese patients. Human polyomavirus 6 (HPyV6) DNA with a relatively high viral load was frequently found in specimens from patients with Kimura disease. Immunohistochemistry revealed the expression of the HPyV6-derived antigen. HPyV6 was detected consistently even when the disease recurred after treatment. The HPyV6 strains studied here were all of the Asian/Japanese genotypes.
The presence of high HPyV6 DNA load, expression of viral antigen, and persistent infection during clinical course suggest that HPyV6 is associated with the pathogenesis of Kimura disease in a subject of patients. Worldwide studies are expected to understand whether the Asian/Japanese HPyV6 genotype is related preferentially to the incidence of the disease with an ethnic predilection for Asian populations.

Academic Significance and Societal Importance of the Research Achievements

本研究において、ウイルス抗原の発現を伴う高ウイルス量のHPyV6が木村病から検出され、これまで不明であった木村病の病因との関連性が示唆された。さらに、日本人発症の木村病から検出されたHPyV6株は全て日本/アジア型変異株あることが示された。このウイルス遺伝子型が、日本人に好発し欧米人では発生頻度が低いという木村病発症率の地理的相違に関与する可能性が示された。これらの研究結果を基に、木村病の病勢診断や新規治療法開発に向けた研究へと発展させることが期待できる。

Report

(4 results)
  • 2022 Annual Research Report   Final Research Report ( PDF )
  • 2021 Research-status Report
  • 2020 Research-status Report
  • Research Products

    (9 results)

All 2023 2022 2021 2020

All Journal Article (6 results) (of which Peer Reviewed: 6 results,  Open Access: 6 results) Presentation (3 results)

  • [Journal Article] Protease-digested egg-white products induce oral tolerance in mice but elicit little IgE production upon epicutaneous exposure2022

    • Author(s)
      Yamada Ayako、Hasegawa Takanori、Fujieda Mikiya、Morita Hideaki、Matsumoto Kenji
    • Journal Title

      Allergology International

      Volume: 71 Issue: 4 Pages: 528-535

    • DOI

      10.1016/j.alit.2022.03.006

    • ISSN
      1323-8930, 1440-1592
    • Related Report
      2022 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Dyskeratosis, a characteristic histopathological feature, seen in a patient with systemic juvenile idiopathic arthritis2022

    • Author(s)
      Nakajima Kimiko、Nakajima Hideki、Araki Mariko、Fujieda Mikiya、Sano Shigetoshi
    • Journal Title

      Journal of Cutaneous Immunology and Allergy

      Volume: 5 Issue: 4 Pages: 136-138

    • DOI

      10.1002/cia2.12227

    • Related Report
      2022 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Cancer After Pediatric Kidney Transplantation: A Long-term Single-center Experience in Japan2021

    • Author(s)
      Yabuuchi Tomoo, Miura Kenichiro, Shimizu Satoru, Kaneko Naoto, Ishizuka Kiyonobu, Kanda Sshoichiro, Chikamoto Hiroko, Akioka Yuko, Fujieda Mikiya, Hattori Motoshi
    • Journal Title

      Transplantation Direct

      Volume: 7 Issue: 4 Pages: e687-e687

    • DOI

      10.1097/txd.0000000000001137

    • NAID

      120007183297

    • Related Report
      2021 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Human polyomavirus 6 with the Asian/Japanese genotype in cases of Kimura disease and angiolymphoid hyperplasia with eosinophilia.2020

    • Author(s)
      Yumiko Hashida, Tomonori Higuchi, Kimiko Nakajima, Takako Ujihara, Ichiro Murakami, Mikiya Fujieda, Shigetoshi Sano, Masanori Daibata.
    • Journal Title

      Journal of Investigative Dermatology

      Volume: 印刷中 Issue: 8 Pages: 1650-1653.e4

    • DOI

      10.1016/j.jid.2019.12.027

    • Related Report
      2020 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Respiratory Syncytial Virus Infection Exacerbates Pneumococcal Pneumonia via Gas6/Axl-mediated Macrophage Polarization2020

    • Author(s)
      Shibata T, Makino A, Ogata R, Nakamura S, Ito T, Nagata K, Terauchi Y, Oishi T, Fujieda M, Takahashi Y, Ato M.
    • Journal Title

      J Clin Invest .

      Volume: 6 Issue: 6 Pages: 3021-3037

    • DOI

      10.1172/jci125505

    • NAID

      120007017546

    • Related Report
      2020 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Transcriptome analysis reveals two distinct endotypes and putative immune pathways in tonsils from children with periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis syndrome2020

    • Author(s)
      Hara Mariko、Morimoto Noriko、Suzuki Noriomi、Tsuchihashi Nana、Komori Manabu、Yoshihama Keisuke、Fujii Kae、Yamaguchi Sota、Tsunoda Mayumi、Tomisato Syuta、Takahashi Nozomi、Oyake Koichiro、Okuba Yuzuru、Fujieda Mikiya、Matsumoto Kenji
    • Journal Title

      Allergy

      Volume: 76 Issue: 1 Pages: 359-363

    • DOI

      10.1111/all.14477

    • Related Report
      2020 Research-status Report
    • Peer Reviewed / Open Access
  • [Presentation] EBウイルス (EBV) とPTLD EBV動態と監視を中心に2023

    • Author(s)
      藤枝幹也
    • Organizer
      第56回日本臨牀腎移植学会
    • Related Report
      2022 Annual Research Report
  • [Presentation] 最近の腎移植後感染症-予防と治療- EBウイルス 移植後リンパ増殖性疾患2020

    • Author(s)
      三浦健一郎、薮内智朗、藤枝幹也、服部元史
    • Organizer
      第53回日本臨床腎移植学会
    • Related Report
      2020 Research-status Report
  • [Presentation] human metapneumovirus感染症で入院した児の中耳炎合併率の評価2020

    • Author(s)
      前田美咲、長尾佳樹、藤枝幹也
    • Organizer
      第15回日本小児耳鼻咽喉科学会総会・学術講演会
    • Related Report
      2020 Research-status Report

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Published: 2020-04-28   Modified: 2024-01-30  

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