ID | 115012 |
タイトル別表記 | Msx1のMH6ドメインを含むC末端領域は骨格形成に関与する
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著者 |
市原, 亜起
徳島大学大学院口腔科学教育部(口腔科学専攻)
ミツイ, シルビア ナオミ
Tokushima University
荒井, 大志
Tokushima University
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キーワード | Msx1
MH6 domain
Bone
Development
Genome editing technology
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資料タイプ |
学位論文
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抄録 | MSX1 is a causative gene for oligodontia in humans. Although conventional Msx1-deficient mice die neonatally, a mutant mouse lacking the C-terminus MH6 domain of MSX1 (Msx1ΔMH6/ΔMH6) showed two different phenotypes; newborn homozygotes with cleft palates died neonatally, whereas those with thin palates remained alive and had craniofacial dysplasia and growth retardation compared with wild-type mice, with most mice dying by the age of 4–5 weeks. In a previously reported case of human oligodontia caused by a heterozygous defect of the Msx1 MH6 domain, a small foramen was observed on the occipital bone. The aim of this study was to test the hypothesis that the Msx1 MH6 domain is involved in bone formation in vivo. In Msx1ΔMH6/ΔMH6 mice, cranial suture fusion was delayed at embryonic day 18.5, and the anteroposterior cranial diameter was smaller and long bone length was decreased at 3 weeks of age. The femoral epiphysis showed no change in the trabecular number, but decreased bone mass, bone density, and trabecular width in Msx1ΔMH6/ΔMH6 mice. In addition, cancellous bone mass was reduced and the cartilage layer in the growth plate was thinner in Msx1ΔMH6/ΔMH6 mice. The mRNA expression levels of major osteoblast and chondrocyte differentiation marker genes were decreased in Msx1ΔMH6/ΔMH6 mice compared with wild-type mice. These findings suggest that the C-terminal region including the MH6 domain of MSX1 plays important roles not only in tooth development and palatal fusion, but also in postnatal bone formation.
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掲載誌名 |
Biochemical and Biophysical Research Communications
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ISSN | 0006291X
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cat書誌ID | AA00564395
AA11542044
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出版者 | Elsevier
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巻 | 526
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号 | 1
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開始ページ | 62
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終了ページ | 69
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発行日 | 2020-03-16
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備考 | 内容要旨・審査要旨・論文本文の公開
本論文は,著者Aki Ichiharaの学位論文として提出され,学位審査・授与の対象となっている。 |
権利情報 | © 2020 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
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EDB ID | |
出版社版DOI | |
出版社版URL | |
フルテキストファイル | |
言語 |
eng
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著者版フラグ |
博士論文全文を含む
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文科省報告番号 | 甲第3443号
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学位記番号 | 甲口第464号
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学位授与年月日 | 2020-09-10
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学位名 |
博士(歯学)
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学位授与機関 |
徳島大学
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部局 |
病院
歯学系
先端酵素学研究所
医学系
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