Chiang Wei-Chieh, Joseph Victory, Yasumura Douglas, Matthes Michael T, Lewin Alfred S, Gorbatyuk Marina S, Ahern Kelly, LaVail Matthew M, Lin Jonathan H
Department of Pathology, University of California, La Jolla, 92093, San Diego, CA, USA.
Department of Ophthalmology, University of California, 94143, San Francisco, CA, USA.
Adv Exp Med Biol. 2016;854:185-91. doi: 10.1007/978-3-319-17121-0_25.
RHO (Rod opsin) encodes a G-protein coupled receptor that is expressed exclusively by rod photoreceptors of the retina and forms the essential photopigment, rhodopsin, when coupled with 11-cis-retinal. Many rod opsin disease -mutations cause rod opsin protein misfolding and trigger endoplasmic reticulum (ER) stress, leading to activation of the Unfolded Protein Response (UPR) signal transduction network. Chop is a transcriptional activator that is induced by ER stress and promotes cell death in response to chronic ER stress. Here, we examined the role of Chop in transgenic mice expressing human P23H rhodopsin (hP23H Rho Tg) that undergo retinal degeneration. With the exception of one time point, we found no significant induction of Chop in these animals and no significant change in retinal degeneration by histology and electrophysiology when hP23H Rho Tg animals were bred into a Chop (-/-) background. Our results indicate that Chop does not play a significant causal role during retinal degeneration in these animals. We suggest that other modules of the ER stress-induced UPR signaling network may be involved photoreceptor disease induced by P23H rhodopsin.
视紫红质(RHO)编码一种G蛋白偶联受体,该受体仅由视网膜的视杆光感受器表达,并在与11-顺式视黄醛结合时形成必需的光色素视紫红质。许多视紫红质疾病突变会导致视紫红质蛋白错误折叠,并引发内质网(ER)应激,从而导致未折叠蛋白反应(UPR)信号转导网络的激活。Chop是一种转录激活因子,由ER应激诱导,并在慢性ER应激反应中促进细胞死亡。在这里,我们研究了Chop在表达人P23H视紫红质(hP23H Rho Tg)并发生视网膜变性的转基因小鼠中的作用。除了一个时间点外,我们在这些动物中未发现Chop有明显诱导,并且当将hP23H Rho Tg动物培育到Chop(-/-)背景中时,通过组织学和电生理学检测,视网膜变性没有明显变化。我们的结果表明,Chop在这些动物的视网膜变性过程中不发挥重要的因果作用。我们认为,ER应激诱导的UPR信号网络的其他模块可能参与了由P23H视紫红质诱导的光感受器疾病。