Moser Markus, Li Yong, Vaupel Kristina, Kretzschmar Doris, Kluge Reinhart, Glynn Paul, Buettner Reinhard
Max Planck Institute for Biochemistry, D-82152 Martinsried, Germany.
Mol Cell Biol. 2004 Feb;24(4):1667-79. doi: 10.1128/MCB.24.4.1667-1679.2004.
Age-dependent neurodegeneration resulting from widespread apoptosis of neurons and glia characterize the Drosophila Swiss Cheese (SWS) mutant. Neuropathy target esterase (NTE), the vertebrate homologue of SWS, reacts with organophosphates which initiate a syndrome of axonal degeneration. NTE is expressed in neurons and a variety of nonneuronal cell types in adults and fetal mice. To investigate the physiological functions of NTE, we inactivated its gene by targeted mutagenesis in embryonic stem cells. Heterozygous NTE(+/-) mice displayed a 50% reduction in NTE activity but underwent normal organ development. Complete inactivation of the NTE gene resulted in embryonic lethality, which became evident after gastrulation at embryonic day 9 postcoitum (E9). As early as E7.5, mutant embryos revealed growth retardation which did not reflect impaired cell proliferation but rather resulted from failed placental development; as a consequence, massive apoptosis within the developing embryo preceded its resorption. Histological analysis indicated that NTE is essential for the formation of the labyrinth layer and survival and differentiation of secondary giant cells. Additionally, impairment of vasculogenesis in the yolk sacs and embryos of null mutant conceptuses suggested that NTE is also required for normal blood vessel development.
果蝇瑞士奶酪(SWS)突变体的特征是神经元和神经胶质细胞广泛凋亡导致的年龄依赖性神经退行性变。神经病变靶标酯酶(NTE)是SWS的脊椎动物同源物,可与引发轴突变性综合征的有机磷酸酯发生反应。NTE在成年和胎儿小鼠的神经元以及多种非神经元细胞类型中表达。为了研究NTE的生理功能,我们通过胚胎干细胞中的靶向诱变使其基因失活。杂合NTE(+/-)小鼠的NTE活性降低了50%,但器官发育正常。NTE基因的完全失活导致胚胎致死,这在妊娠后第9天(E9)的原肠胚形成后变得明显。早在E7.5时,突变胚胎就显示出生长迟缓,这并非反映细胞增殖受损,而是由于胎盘发育失败所致;因此,发育中的胚胎在被吸收之前就出现了大量凋亡。组织学分析表明,NTE对于迷路层的形成以及次级巨细胞的存活和分化至关重要。此外,空突变体胚胎的卵黄囊和胚胎中血管生成的受损表明,正常血管发育也需要NTE。