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Biochemical study for deficient root resorption in the primary dentition and dental radiculomegaly formation in OFCD syndrome

Research Project

Project/Area Number 15K11369
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Research Field Orthodontics/Pediatric dentistry
Research InstitutionNagasaki University

Principal Investigator

NISHIGUCHI Miyuki  長崎大学, 医歯薬学総合研究科(歯学系), 助教 (10253676)

Co-Investigator(Kenkyū-buntansha) 藤原 卓  長崎大学, 医歯薬学総合研究科(歯学系), 教授 (00228975)
星野 倫範  長崎大学, 医歯薬学総合研究科(歯学系), 准教授 (00359960)
釜崎 陽子  長崎大学, 病院(歯学系), 講師 (30253678)
佐藤 恭子  長崎大学, 医歯薬学総合研究科(歯学系), 客員研究員 (70404499)
Research Collaborator YAMADA Aya  
Project Period (FY) 2015-04-01 – 2019-03-31
Project Status Completed (Fiscal Year 2018)
Budget Amount *help
¥4,810,000 (Direct Cost: ¥3,700,000、Indirect Cost: ¥1,110,000)
Fiscal Year 2017: ¥1,040,000 (Direct Cost: ¥800,000、Indirect Cost: ¥240,000)
Fiscal Year 2016: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
Fiscal Year 2015: ¥2,080,000 (Direct Cost: ¥1,600,000、Indirect Cost: ¥480,000)
KeywordsOFCD syndrome / radiculomegaly / 乳歯歯髄細胞 / 破骨細胞 / 細胞増殖 / BCOR遺伝子 / 乳歯脱落不全 / OFCDsyndrome / radiculomegary / caridiac disease / congenital cataracts / BCOR / dental anomaly / OFCD症候群 / 歯根吸収不全 / 歯髄細胞 / 間葉系幹細胞 / 歯根長根 / 歯髄冠細胞
Outline of Final Research Achievements

To confirm this diagnosis, genetic analysis was performed on genomic DNA isolated from a saliva sample using Orangene® DNA kit (GNA Genotek, Inc, Ottawa, ON, Canada). DNA sequencing showed a frameshift mutation in exon 12 of the BCOR gene of this patient (c.2559_2560insA).
Patient’s anterior primary teeth were extracted were markedly long compared with those in Japanese controls Dental anomalies of her include delayed eruption and prolonged retention of primary teeth, delayed eruption of permanent teeth, radiculomegaly of primary and permanent teeth, supernumerary teeth and major fused teeth. We extracted the pulp cell of the primary mandibular canine which extracted to promote eruption of the permanent tooth. We confirmed osteoclasts differentiation using the pulp cell, but did not show differentiation. Also, as a result of having examined the proliferation potency of the cultured cell, proliferation potency was loud in comparison with control.

Academic Significance and Societal Importance of the Research Achievements

OFCD症候群の患者では全身疾患以外に口腔内の異常が多く認められる。その中で長根と乳歯脱落遅延による歯列不正はほとんどの本疾患患者に認められる。しかし、乳歯列からの症例が少なく矯正の対応は遅れている。このことから本疾患患者に対する矯正治療を支援するための報告となったと考える。また長根や乳歯脱落不全となる原因は不明である。今回、患者の歯髄細胞の増殖が早かったことや破歯細胞分化を認めなかったという結果は歯の再生研究において重要な示唆を与える可能性を持つと考える。

Report

(5 results)
  • 2018 Annual Research Report   Final Research Report ( PDF )
  • 2017 Research-status Report
  • 2016 Research-status Report
  • 2015 Research-status Report
  • Research Products

    (2 results)

All 2018

All Journal Article (1 results) (of which Peer Reviewed: 1 results,  Open Access: 1 results) Presentation (1 results)

  • [Journal Article] Long-term observation of a case of oculo-facio-cardio-dental syndrome that showed remarkable radiculomegaly of primary teeth2018

    • Author(s)
      Nishiguchi M, Sasaki Y, Satoh K, Kamasaki Y, Kondo Y and Fujiwara T
    • Journal Title

      Journal of Translational Science

      Volume: 5 Issue: 2 Pages: 1-4

    • DOI

      10.15761/jts.1000257

    • Related Report
      2018 Annual Research Report
    • Peer Reviewed / Open Access
  • [Presentation] A case of a girl with oculofaciocardiodental(OFCD)syndrome2018

    • Author(s)
      Miyuki Nishiguchi, Keigo Imamura, Haruka Nishimata, Kyoko Satoh, Yasunori Sasaki, Taku Fujiwara
    • Organizer
      日本小児歯科学会
    • Related Report
      2018 Annual Research Report

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Published: 2015-04-16   Modified: 2020-03-30  

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