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