MacPherson David, Sage Julien, Kim Teresa, Ho Dennis, McLaughlin Margaret E, Jacks Tyler
Department of Biology, Massachusetts Institute of Technology, Cambridge, Massachusetts 02139, USA.
Genes Dev. 2004 Jul 15;18(14):1681-94. doi: 10.1101/gad.1203304. Epub 2004 Jul 1.
Certain cells of the human retina are extremely sensitive to loss of function of the retinoblastoma tumor suppressor gene RB. Retinoblastomas develop early in life and at high frequency in individuals heterozygous for a germ-line RB mutation, and sporadic retinoblastomas invariably have somatic mutation in the RB gene. In contrast, retinoblastomas do not develop in Rb+/- mice. Although retinoblastoma is thought to have developmental origins, the function of Rb in retinal development has not been fully characterized. Here we studied the role of Rb in normal retinal development and in retinoblastoma using conditional Rb mutations in the mouse. In late embryogenesis, Rb-deficient retinas exhibited ectopic S-phase and high levels of p53-independent apoptosis, particularly in the differentiating retinal ganglion cell layer. During postnatal retinal development, loss of Rb led to more widespread retinal apoptosis, and adults showed loss of photoreceptors and bipolar cells. Conditional Rb mutation in the retina did not result in retinoblastoma formation even in a p53-mutant background. However, on a p107- or p130-deficient background, Rb mutation in the retina caused retinal dysplasia or retinoblastoma.
人类视网膜的某些细胞对视网膜母细胞瘤肿瘤抑制基因RB的功能丧失极为敏感。视网膜母细胞瘤在生命早期发生,并且在种系RB突变的杂合个体中具有高发性,散发性视网膜母细胞瘤总是在RB基因中发生体细胞突变。相比之下,Rb+/-小鼠不会发生视网膜母细胞瘤。尽管视网膜母细胞瘤被认为起源于发育过程,但Rb在视网膜发育中的功能尚未完全阐明。在这里,我们利用小鼠中的条件性Rb突变研究了Rb在正常视网膜发育和视网膜母细胞瘤中的作用。在胚胎发育后期,Rb缺陷型视网膜表现出异位S期和高水平的不依赖p53的细胞凋亡,特别是在分化中的视网膜神经节细胞层。在出生后视网膜发育过程中,Rb的缺失导致更广泛的视网膜细胞凋亡,成年小鼠表现出光感受器和双极细胞的缺失。即使在p53突变背景下,视网膜中的条件性Rb突变也不会导致视网膜母细胞瘤的形成。然而,在p107或p130缺陷背景下,视网膜中的Rb突变会导致视网膜发育异常或视网膜母细胞瘤。