ID | 115012 |
Title Alternative | 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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Author |
Ichihara, Aki
Tokushima University
Mitsui, Silvia Naomi
Tokushima University
Arai, Daishi
Tokushima University
Minegishi, Yoshiyuki
Tokushima University
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Oyadomari, Seiichi
Tokushima University
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Imoto, Issei
Tokushima University|Aichi Cancer Center Research Institute|Nagoya University
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Tanaka, Eiji
Tokushima University
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Keywords | Msx1
MH6 domain
Bone
Development
Genome editing technology
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Content Type |
Thesis or Dissertation
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Description | 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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Journal Title |
Biochemical and Biophysical Research Communications
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ISSN | 0006291X
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NCID | AA00564395
AA11542044
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Publisher | Elsevier
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Volume | 526
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Issue | 1
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Start Page | 62
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End Page | 69
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Published Date | 2020-03-16
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Remark | 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。 |
Rights | © 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 | |
DOI (Published Version) | |
URL ( Publisher's Version ) | |
FullText File | |
language |
eng
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TextVersion |
ETD
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MEXT report number | 甲第3443号
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Diploma Number | 甲口第464号
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Granted Date | 2020-09-10
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Degree Name |
Doctor of Dental Science
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Grantor |
Tokushima University
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departments |
University Hospital
Oral Sciences
Institute of Advanced Medical Sciences
Medical Sciences
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