McNamee Shannon M, Akula Monica, Love Zoe, Nasraty Neelaab, Nystuen Kaden, Singh Pushpendra, Upadhyay Arun K, DeAngelis Margaret M, Haider Neena B
Schepens Eye Research Institute, Massachusetts Eye and Ear, Department of Ophthalmology, Harvard Medical School, 20 Staniford Street, Boston, MA, 02114, USA.
University of Massachusetts Amherst, Amherst, MA, USA.
Sci Rep. 2024 Jul 17;14(1):16490. doi: 10.1038/s41598-024-67095-6.
Retinitis Pigmentosa is a leading cause of severe vision loss. Retinitis Pigmentosa can present with a broad range of phenotypes impacted by disease age of onset, severity, and progression. This variation is influenced both by different gene mutations as well as unique variants within the same gene. Mutations in the nuclear hormone receptor 2 family e, member 3 are associated with several forms of retinal degeneration, including Retinitis Pigmentosa. In our previous studies we demonstrated that subretinal administration of one Nr2e3 dose attenuated retinal degeneration in rd7 mice for at least 3 months. Here we expand the studies to evaluate the efficacy and longitudinal impact of the NR2E3 therapeutic by examining three different doses administered at early or intermediate stages of retinal degeneration in the rd7 mice. Our study revealed retinal morphology was significantly improved 6 months post for all doses in the early-stage treatment groups and for the low and mid doses in the intermediate stage treatment groups. Similarly, photoreceptor function was significantly improved in the early stage for all doses and intermediate stage low and mid dose groups 6 months post treatment. This study demonstrated efficacy in multiple doses of NR2E3 therapy.
视网膜色素变性是严重视力丧失的主要原因。视网膜色素变性可表现出广泛的表型,受疾病发病年龄、严重程度和进展的影响。这种变异既受不同基因突变的影响,也受同一基因内独特变异的影响。核激素受体2家族e成员3的突变与多种形式的视网膜变性有关,包括视网膜色素变性。在我们之前的研究中,我们证明了视网膜下给予一剂Nr2e3可使rd7小鼠的视网膜变性至少减轻3个月。在此,我们扩展研究,通过检查在rd7小鼠视网膜变性早期或中期给予的三种不同剂量,来评估NR2E3治疗的疗效和长期影响。我们的研究表明,早期治疗组的所有剂量以及中期治疗组的低剂量和中剂量在治疗6个月后,视网膜形态均有显著改善。同样,治疗6个月后,早期所有剂量组以及中期低剂量和中剂量组的光感受器功能均有显著改善。这项研究证明了多剂量NR2E3治疗的疗效。