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Analysis of PSMB8 mutations in cases with skin damage by a proteasome inhibitor

Research Project

Project/Area Number 26860890
Research Category

Grant-in-Aid for Young Scientists (B)

Allocation TypeMulti-year Fund
Research Field Dermatology
Research InstitutionWakayama Medical University

Principal Investigator

KAYO KUNIMOTO  和歌山県立医科大学, 医学部, 助教 (10438278)

Project Period (FY) 2014-04-01 – 2016-03-31
Project Status Completed (Fiscal Year 2015)
Budget Amount *help
¥3,900,000 (Direct Cost: ¥3,000,000、Indirect Cost: ¥900,000)
Fiscal Year 2015: ¥1,950,000 (Direct Cost: ¥1,500,000、Indirect Cost: ¥450,000)
Fiscal Year 2014: ¥1,950,000 (Direct Cost: ¥1,500,000、Indirect Cost: ¥450,000)
Keywordsproteasome / Nakajo-Nishimura / protesome inhibitor / skin eruption / プロテアソーム / 中條-西村症候群 / PSMB8遺伝子 / 自己炎症疾患 / 多発性骨髄腫 / プロテアソ-ム / PSMB8遺伝子
Outline of Final Research Achievements

It is known that a proteasome inhibitor induces cutaneous adverse reaction with a high probability. The histopathology of the skin eruption is similar to that of Nakajo-Nishimura syndrome(NNS). PSMB8 mutation was not detected by direct sequence method using the genome DNA extracted from 8 skin biopsy specimens of protesome inhibitor-induced cutaneous eruption because the PSMB8 mutation might present as genetic background. Slides were obtained from the 9 reported cases of proteasome inhibitor-induced to compare the histology. The hematoxylin-eosin and immunohistochemical findings of both cases were similar. In both cases, the histology showed mononuclear infiltrate in the dermis to adipose tissue and around adnexal tissue. The dermal infiltrate was positive for myeloperoxidase, CD68, ubiquitin. In 9 patients with proteasome inhibitor-induced cutaneous eruption, the dermal infiltrate was positive for CD4. On the other hand, the infiltrate was positive for CD8 in NNS.

Report

(3 results)
  • 2015 Annual Research Report   Final Research Report ( PDF )
  • 2014 Research-status Report
  • Research Products

    (9 results)

All 2015 2014

All Journal Article (1 results) (of which Peer Reviewed: 1 results) Presentation (8 results) (of which Int'l Joint Research: 3 results,  Invited: 1 results)

  • [Journal Article] Is CANDLE the best nomenclature?2014

    • Author(s)
      Kanazawa N, Kunimoto K, Ishii N, et al
    • Journal Title

      Br J Dermatol

      Volume: (in press) Issue: 3 Pages: 659-660

    • DOI

      10.1111/bjd.12962

    • Related Report
      2014 Research-status Report
    • Peer Reviewed
  • [Presentation] Analysis of the ROS production in neutrophils of Nakajo-Nishimura syndrome patients.2015

    • Author(s)
      Kayo Kunimoto, Fumiko Ozaki, Fukumi Furukawa, Nobuo Kanazawa
    • Organizer
      The 40th Annual Meeting of the Japanese Society for lnvestigative Dermatology
    • Place of Presentation
      Okayama, Japan
    • Year and Date
      2015-12-11
    • Related Report
      2015 Annual Research Report
    • Int'l Joint Research
  • [Presentation] Beneficial effect of methotrexate on a case of Nakajo-Nishimura syndrome.2015

    • Author(s)
      Kayo Kunimoto, Fumiko Ozaki, Fukumi Furukawa, Nobuo Kanazawa
    • Organizer
      The 8th International Congress of Familial Mediterranean Fever and Systemic Autoinflammatory Diseases
    • Place of Presentation
      Dresden, Germany
    • Year and Date
      2015-09-30
    • Related Report
      2015 Annual Research Report
    • Int'l Joint Research
  • [Presentation] Temporal changes of serum cytokine/chemokine levels in patients with Nakajo-Nishimra syndrome treated with tocilizumab.2015

    • Author(s)
      Nobuo Kanazawa, Yumi Nakatani, Yutaka Inaba, Kayo Kunimoto, Fukumi Furukawa
    • Organizer
      The 8th International Congress of Familial Mediterranean Fever and Systemic Autoinflammatory Diseases
    • Place of Presentation
      Dresden, Germany
    • Year and Date
      2015-09-30
    • Related Report
      2015 Annual Research Report
    • Int'l Joint Research
  • [Presentation] メトトレキサートの追加投与が有効であった中條-西村症候群の小児例2015

    • Author(s)
      国本佳代、稲葉豊、古川福実、金澤伸雄
    • Organizer
      第39回日本小児皮膚科学会学術大会
    • Place of Presentation
      鹿児島
    • Year and Date
      2015-07-18
    • Related Report
      2015 Annual Research Report
  • [Presentation] プロテアゾーム機能不全症~中條-西村症候群とその類症~2015

    • Author(s)
      国本佳代、稲葉 豊、古川福実、金澤伸雄
    • Organizer
      第114回日本皮膚科学会総会
    • Place of Presentation
      横浜
    • Year and Date
      2015-05-29
    • Related Report
      2015 Annual Research Report
    • Invited
  • [Presentation] プロテアソーム機能不全症~中條‐西村症候群とその類症~2015

    • Author(s)
      国本佳代、稲葉豊、古川福実、金澤伸雄
    • Organizer
      第114回日本皮膚科学会総会
    • Place of Presentation
      横浜
    • Year and Date
      2015-05-29
    • Related Report
      2014 Research-status Report
  • [Presentation] 中條―西村症候群 患者血中サイトカンンの経時的解析2014

    • Author(s)
      金澤伸雄、中谷友美、稲葉 豊、国本佳代、古川福実
    • Organizer
      第438回日本皮膚科学会京滋地方会
    • Place of Presentation
      京都
    • Year and Date
      2014-12-19
    • Related Report
      2014 Research-status Report
  • [Presentation] Nakajo-Nishimura syndrome: a hereditary proteasome disability syndrome sharing the genetic origin with JMP and CANDLE syndrome.2014

    • Author(s)
      Nobuo Kanazawa, Yutaka Inaba, Kayo Kunimoto, Fukumi Furukawa
    • Organizer
      The 11th Meeting of the GJSD
    • Place of Presentation
      Heiderberg, Germany
    • Year and Date
      2014-06-11 – 2014-06-14
    • Related Report
      2014 Research-status Report

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Published: 2014-04-04   Modified: 2017-05-10  

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