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用超高分辨率 OCT 评估 BEST1 突变犬的光暴露视网膜反应。

Retinal response to light exposure in BEST1-mutant dogs evaluated with ultra-high resolution OCT.

机构信息

Center for Hereditary Retinal Degenerations, Scheie Eye Institute, Department of Ophthalmology, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, United States.

Division of Experimental Retinal Therapies, Department of Clinical Sciences & Advanced Medicine, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA, United States.

出版信息

Vision Res. 2024 May;218:108379. doi: 10.1016/j.visres.2024.108379. Epub 2024 Mar 8.

Abstract

Mutations in BEST1 cause an autosomal recessive disease in dogs where the earliest changes localize to the photoreceptor-RPE interface and show a retina-wide micro-detachment that is modulated by light exposure. The purpose of this study was to define the spatial and temporal details of the outer retina and its response to light with ultra-high resolution OCT across a range of ages and with different BEST1 mutations. Three retinal regions were selected in each eye: near the fovea-like area, near the optic nerve, both in the tapetal area, and inferior to the optic nerve in the non-tapetal area. The OS+ slab thickness was defined between the peak near the junction of inner and outer segments (IS/OS) and the transition between basal RPE, Bruch membrane, choriocapillaris and proximal tapetum (RPE/T). In wildtype (WT) dogs, two tapetal regions showed additional hyperscattering OCT peaks within the OS+ slab likely representing cone and rod outer segment tips (COST and ROST). The inferior non-tapetal region of WT dogs had only one of these peaks, likely ROST. In dogs with BEST1 mutations, all three locations showed a single peak, likely suggesting optical silence of COST. Light-dependent expansion of the micro-detachment by about 10 um was detectable in both tapetal and non-tapetal retina across all ages and BEST1 mutations.

摘要

BEST1 基因突变会导致犬类常染色体隐性疾病,最早的变化定位于光感受器-RPE 界面,并表现出受光暴露调节的全视网膜微脱离。本研究的目的是通过超高速 OCT 定义不同 BEST1 突变的不同年龄的外视网膜及其对光的反应的时空细节。在每只眼睛中选择三个视网膜区域:靠近类黄斑区域、靠近视神经、均位于绒毡层区域以及在非绒毡层区域低于视神经。OS+ 板层厚度定义为靠近内、外节交界处(IS/OS)的峰值和基底 RPE、Bruch 膜、脉络膜毛细血管和近端绒毡层(RPE/T)之间的过渡之间。在野生型(WT)犬中,两个绒毡层区域在 OS+ 板层内显示出额外的超散射 OCT 峰,可能代表圆锥和杆状细胞外节尖端(COST 和 ROST)。WT 犬的下非绒毡层区域只有一个这样的峰,可能是 ROST。在具有 BEST1 突变的犬中,所有三个位置均显示单个峰,可能表明 COST 光学沉默。在所有年龄和 BEST1 突变中,在绒毡层和非绒毡层视网膜中都可以检测到微脱离受光依赖性扩张约 10 µm。

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