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2017 Fiscal Year Final Research Report

Analysis of Gorlin syndrome derived benign or malignant tumor

Research Project

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Project/Area Number 15K11098
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Research Field Pathobiological dentistry/Dental radiology
Research InstitutionHyogo Medical University

Principal Investigator

Nakano Yoshiro  兵庫医科大学, 医学部, 教授 (30360267)

Co-Investigator(Kenkyū-buntansha) 玉置 知子 (橋本知子)  兵庫医科大学, 医学部, 名誉教授 (10172868)
山西 清文  兵庫医科大学, 医学部, 教授 (10182586)
岸本 裕充  兵庫医科大学, 医学部, 教授 (30291818)
野口 一馬  兵庫医科大学, 医学部, 准教授 (50309473)
Project Period (FY) 2015-04-01 – 2018-03-31
KeywordsGorlin症候群 / 角化嚢胞性歯原性腫瘍 / hedgehog情報伝達 / PTCH1 / SMO / 基底細胞癌
Outline of Final Research Achievements

Gorlin syndrome is a dominantly inherited autosomal disease with a high frequency of tumor induction caused by the hedgehog receptor PTCH1. We analyzed PTCH1 and SMO alternation in Gorlin syndrome-associated tumors, such as malignant basal cell carcinoma, benign keratocystic odontogenic tumor (KCOT), and ovarian fibroma, revealing that the mutational status of PTCH1 or SMO itself does not account for differences in malignancy and tissue-specificity of these tumors.
KCOT is the most frequent benign tumor in Gorlin syndrome patients. We established KCOT cell lines from sporadic and Gorlin syndrome-derived KCOTs. Both lines showed stem and mesenchymal cell characteristics. However, in the presence of calcium, cells showed parakeratosis and epithelial or keratinocyte-related proteins were induced. These features were similar to those of in vivo KCOTs. Using these cell lines and 3-D hydrogel culture, we may be able to advance understanding of the nature and origin of KCOTs.

Free Research Field

腫瘍遺伝学

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Published: 2019-03-29  

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