A three-dimensional analysis of primary failure of eruption in humans and mice.
A three-dimensional analysis of primary failure of eruption in humans and mice.
复制标题
DOI：
10.1111/odi.13249
复制
发表时间：
2020-03
期刊：
影响因子：
3.8
通讯作者：
Ono W
中科院分区：
文献类型：
--
作者：
Tokavanich N;Gupta A;Nagata M;Takahashi A;Matsushita Y;Yatabe M;Ruellas A;Cevidanes L;Maki K;Yamaguchi T;Ono N;Ono W
原发性萌出失败（PFE）是一种表现为牙齿萌出停止的遗传性疾病。据报道，甲状旁腺激素（PTH）/甲状旁腺激素相关肽（PTHrP）受体（PTH/PTHrP受体，PPR）的功能丧失突变是人类这种疾病的根本原因。我们在PFE小鼠模型中发现，PTHrP-PPR信号传导是正常牙囊细胞分化和牙齿萌出的原因。然而，PFE喷发缺陷的机制尚不清楚。在这项描述性研究中，我们的目的是通过3D microCT分析按时间顺序观察小鼠PFE磨牙的牙齿萌出和牙根形成。昭和大学招募了两名PFE患者。通过在表达PTHrP的牙囊中特异性缺失PPR来产生小鼠PFE模型，并将其分为三组，PPRfl/fl; R26 RtdTomato/+（对照），PTHrP-creER;PPRfl/+; R26 RtdTomato/+（cHet）和PTHrP-creER;PRRfl/fl; R26 RtdTomato/+（cKO）。通过CBCT获取来自人类PFE受试者的图像。在出生后第14天、第25天、第91天和第182天，在P3时三苯氧胺脉冲后研究所有组的小鼠样本，并呈现三组之间的3D microCT图像的叠加。小鼠和人类PFE磨牙在三维CT分析中表现出相似的表现。小鼠的定量分析表明，出生后第25天，与其他组相比，cKO第一和第二磨牙的萌出高度在统计学上显著降低。此外，cKO磨牙表现出明显缩短的牙根，并伴有与根间骨高度降低相关的撕裂。与正常磨牙相比，小鼠PFE磨牙的萌出速度要慢得多，这与牙根缩短和撕裂以及根间骨缺陷有关。
Primary failure of eruption (PFE) is a genetic disorder exhibiting the cessation of tooth eruption. Loss-of-function mutations in parathyroid hormone (PTH)/parathyroid hormone-related peptide (PTHrP) receptor (PTH/PTHrP receptor, PPR) were reported as the underlying cause of this disorder in humans. We showed in a PFE mouse model that PTHrP-PPR signaling is responsible for normal dental follicle cell differentiation and tooth eruption. However, the mechanism underlying the eruption defect in PFE remains undefined. In this descriptive study, we aim to chronologically observe tooth eruption and root formation of mouse PFE molars through 3D microCT analyses. Two individuals with PFE were recruited at Showa University. A mouse PFE model was generated by deleting PPR specifically in PTHrP-expressing dental follicle and divided into three groups, PPRfl/fl;R26RtdTomato/+(Control), PTHrP-creER;PPRfl/+;R26RtdTomato/+(cHet), and PTHrP-creER;PRRfl/fl;R26RtdTomato/+(cKO). Images from human PFE subjects were acquired by CBCT. All groups of mouse samples were studied at postnatal days 14, 25, 91, and 182 after a tamoxifen pulse at P3, and superimposition of 3D microCT images among three groups was rendered. Mouse and human PFE molars exhibited a similar presentation in the 3D CT analyses. The quantitative analysis in mice demonstrated a statistically significant decrease in the eruption height of cKO first and second molars compared to other groups after postnatal day 25. Additionally, cKO molars demonstrated significantly shortened roots with dilacerations associated with the reduced interradicular bone height. Mouse PFE molars erupt at a much slower rate compared to normal molars, associated with shortened and dilacerated roots and defective interradicular bones.
登录
查看更多内容
DOI：
10.1007/s00056-014-0215-y
发表时间：
2014-05-01
影响因子：
1.7
作者：
Pilz, Peter;Meyer-Marcotty, Philipp;Stellzig-Eisenhauer, Angelika
通讯作者：
Stellzig-Eisenhauer, Angelika
DOI：
10.1016/j.ajodo.2006.09.038
发表时间：
2007-05-01
影响因子：
3
作者：
Frazier-Bowers, Sylvia A.;Koehler, Karen E.;Proffit, William R.
通讯作者：
Proffit, William R.
DOI：
10.1016/s0096-6347(41)90413-1
发表时间：
1941-01-01
期刊：
AMER JOUR ORTHODONT AND ORAL SURG
影响因子：
--
作者：
MASSLER, MAURY;SHOUR, I.
通讯作者：
SHOUR, I.
影响因子：
64.8
作者：
Mizuhashi K;Ono W;Matsushita Y;Sakagami N;Takahashi A;Saunders TL;Nagasawa T;Kronenberg HM;Ono N
通讯作者：
Ono N
影响因子：
3.4
作者：
Grippaudo, Cristina;Cafiero, Concetta;Frazier-Bowers, Sylvia A.
通讯作者：
Frazier-Bowers, Sylvia A.