Wang Yanjie, Nusinowitz Steven, Yang Xian-Jie
University of California, Los Angeles.
Res Sq. 2025 Jul 11:rs.3.rs-7089882. doi: 10.21203/rs.3.rs-7089882/v1.
Retinitis pigmentosa (RP) is an inherited retinal disease in which the loss of rod photoreceptors precedes cone photoreceptor degeneration. The neurocytokine ciliary neurotrophic factor (CNTF) can provide potent neuroprotection for photoreceptors in various retinal degeneration models and has thus been tested in clinical trials aimed at treating blinding diseases. In a preclinical model of RP, exogenous CNTF signaling mediated by the cytokine receptor gp130 initially triggered STAT3 and ERK phosphorylation in Muller glial cells and subsequently activated STAT3 in rods to promote photoreceptor survival. However, despite enhancing photoreceptor viability, the constitutive expression of exogenous CNTF perturbs the retinal transcriptome and further suppresses visual function. Activated STAT3 upregulates suppressor of cytokine signaling 3 (SOCS3), which acts as a feedback inhibitor to dampen cytokine signaling. In this study, we investigated whether eliminating SOCS3 in rod cells is sufficient to increase endogenous STAT3 signaling and enhance photoreceptor viability without exogenous CNTF. We show that rod-specific SOCS3 deletion attenuates photoreceptor degeneration and improves cone cell morphology in both the Pde6b/rd10 and Prph2(P216L)/rds mouse models of RP. SOCS3 ablation in rods not only causes STAT3 activation in rod photoreceptors but also leads to the propagation of STAT3 and ERK signaling to inner retinal cell types. Furthermore, rod SOCS3 deficiency led to improved visual function in the Pde6b/rd10 model and sustained cone function in the Prph2(P216L)/rds retina. Together, these findings demonstrate that intercellular communication occurs among retinal cells and the modulation of endogenous cytokine signaling events can be leveraged as an efficacious treatment to attenuate neuronal loss and preserve visual function.
视网膜色素变性(RP)是一种遗传性视网膜疾病,其中视杆光感受器的丧失先于视锥光感受器的退化。神经细胞因子睫状神经营养因子(CNTF)可以为各种视网膜变性模型中的光感受器提供强大的神经保护作用,因此已经在旨在治疗致盲疾病的临床试验中进行了测试。在RP的临床前模型中,由细胞因子受体gp130介导的外源性CNTF信号最初触发了穆勒胶质细胞中的STAT3和ERK磷酸化,随后激活了视杆细胞中的STAT3以促进光感受器存活。然而,尽管增强了光感受器的活力,但外源性CNTF的组成型表达扰乱了视网膜转录组并进一步抑制了视觉功能。激活的STAT3上调细胞因子信号转导抑制因子3(SOCS3),其作为反馈抑制剂来减弱细胞因子信号。在本研究中,我们研究了在视杆细胞中消除SOCS3是否足以增加内源性STAT3信号并在没有外源性CNTF的情况下增强光感受器活力。我们表明,在Pde6b/rd10和Prph2(P216L)/rds RP小鼠模型中,视杆特异性SOCS3缺失可减轻光感受器退化并改善视锥细胞形态。视杆细胞中SOCS3的缺失不仅导致视杆光感受器中STAT3的激活,还导致STAT3和ERK信号向内视网膜细胞类型的传播。此外,视杆SOCS3缺乏导致Pde6b/rd10模型中的视觉功能改善以及Prph2(P216L)/rds视网膜中视锥功能的维持。总之,这些发现表明视网膜细胞之间存在细胞间通讯,并且内源性细胞因子信号事件的调节可以作为一种有效的治疗方法来减轻神经元损失并保留视觉功能。