Phex mutation causes the reduction of Npt2b mRNA in teeth

Phex mutation causes the reduction of Npt2b mRNA in teeth
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DOI:
10.1177/154405910708600210
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发表时间:
2007-02-01
影响因子:
7.6
通讯作者:
Ooshima, T.
Ooshima, T.
中科院分区:
医学1区
文献类型:
--
作者:
Onishi, T.;Okawa, R.;Ooshima, T.

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Hyp小鼠(人类X-连锁低磷酸盐血症的鼠同源物)具有磷酸盐稳态障碍,并且在骨骼和牙齿中显示低矿化。我们研究了Phex(X染色体上的内肽酶磷酸调节基因同源性)突变是否对Hyp小鼠发育中牙齿中II型钠依赖性磷酸盐协同转运蛋白(Npt 2)的表达水平有影响。定量RT-PCR分析显示,在体内和体外实验中,Hyp小鼠牙胚中Npt 2的亚型Npt 2b mRNA的量显著低于野生型小鼠。此外,在添加Phex反义寡核苷酸的培养基中培养的野生型小鼠牙胚也显示Npt 2b mRNA表达减少。这些结果表明,Phex功能的丧失与牙齿中Npt 2b表达的缺陷有关,Npt 2b减少是Hyp小鼠牙齿的内在缺陷。
Hyp mice (murine homologue of human X-linked hypophosphatemia) have a disorder in phosphate homeostasis, and display hypomineralization in bones and teeth. We investigated whether a mutation of Phex (phosphate regulating gene homologies to endopeptidase on the X chromosome) has an effect on the expression level of type II sodium-dependent phosphate cotransporter (Npt2) in the developing teeth of the Hyp mouse. Quantitative RT-PCR analyses revealed that the amount of Npt2b mRNA, an isoform of Npt2, in Hyp mouse tooth germs was significantly lower than that in wild-type mice, in both in vivo and in vitro experiments. In addition, tooth germs from wild-type mice cultured in medium supplemented with antisense oligo-deoxynucleotide for Phex also showed a reduction of Npt2b mRNA expression. These findings suggest that the loss of Phex function is related to the defect of Npt2b expression in teeth, and Npt2b reduction is an intrinsic defect of Hyp murine teeth.