The C-terminal region including the MH6 domain of Msx1 regulates skeletal development

• Material Type: 博士論文
• Title: The C-terminal region including the MH6 domain of Msx1 regulates skeletal development
• Author/Editor: Ichihara, Aki; Yasue, Akihiro; Mitsui, Silvia Naomi; Arai, Daishi; Minegishi, Yoshiyuki; Oyadomari, Seiichi; Imoto, Issei; Tanaka, Eiji
• Author Heading: Ichihara, Aki; Yasue, Akihiro; Mitsui, Silvia Naomi; Arai, Daishi; Minegishi, Yoshiyuki; Oyadomari, Seiichi; Imoto, Issei; Tanaka, Eiji
• Publication, Distribution, etc.: Elsevier
• Publication Date: 2020-03-16
• Publication Date (W3CDTF): 2020-03-16
• Alternative Title: 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
• Periodical title: Biochemical and Biophysical Research Communications
• No. or year of volume/issue: 526 1
• Volume: 526
• Issue: 1
• Pages: 62-69
• ISSN (Periodical Title): ISSN : 0006-291X
• Degree grantor/type: 徳島大学
• Date Granted: 2020-09-10
• Dissertation Number: 甲第3443号
• Degree Type: 博士（歯学）
• Text Language Code: eng
• Subject Heading: Msx1; MH6 domain; Bone; Development; Genome editing technology
• Target Audience: 一般
• Note (General): 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.
• Format (IMT): application/pdf
• Access Restrictions: インターネット公開
• Rights (production): © 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/).
• Related Material (DOI): 10.1016/j.bbrc.2020.03.068
• Data Provider (Database): 国立情報学研究所 : 学術機関リポジトリデータベース（IRDB）（機関リポジトリ）; https://irdb.nii.ac.jp
• Original Data Provider (Database): 徳島大学 : 徳島大学機関リポジトリ

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http://repo.lib.tokushima-u.ac.jp/files/public/11/115012/2020092514493429859/k3443_abstract.pdf fulltext

http://repo.lib.tokushima-u.ac.jp/files/public/11/115012/20200925144934523383/k3443_review.pdf fulltext

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