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

IF 1.5 4区 心理学 Q4 NEUROSCIENCES Vision Research Pub Date : 2024-03-08 DOI:10.1016/j.visres.2024.108379
Vivian Wu , Malgorzata Swider , Alexander Sumaroka , Valerie L. Dufour , Joseph E. Vance , Tomas S. Aleman , Gustavo D. Aguirre , William A. Beltran , Artur V. Cideciyan
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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.

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利用超高分辨率光学视网膜成像技术评估 BEST1 突变体狗视网膜对光照射的反应
BEST1 基因突变会导致狗患一种常染色体隐性遗传病,最早的变化发生在感光体-RPE 界面,并表现出视网膜范围内的微脱离,这种脱离受光线照射的影响。本研究的目的是通过超高分辨率 OCT 确定不同年龄段和不同 BEST1 突变的外层视网膜的空间和时间细节及其对光的反应。每只眼睛选择了三个视网膜区域:近眼窝区、视神经附近(均位于视锥区)和非视锥区视神经下侧。OS+ 板厚度定义为内节和外节交界处附近的峰值(IS/OS)与基底 RPE、布鲁氏膜、绒毛膜和近端视网膜之间的过渡(RPE/T)。在野生型(WT)狗中,两个锥体区域在 OS+ 板块内显示出额外的超散射 OCT 峰,可能代表锥体和杆外节尖端(COST 和 ROST)。WT 狗的下非锥体区域只有一个这样的峰,可能是 ROST。在 BEST1 基因突变的狗中,所有三个位置都显示一个峰值,这可能表明 COST 的光学沉默。在所有年龄段的狗和 BEST1 基因突变狗的视网膜上,都能检测到微脱离随光扩展约 10 um。
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来源期刊
Vision Research
Vision Research 医学-神经科学
CiteScore
3.70
自引率
16.70%
发文量
111
审稿时长
66 days
期刊介绍: Vision Research is a journal devoted to the functional aspects of human, vertebrate and invertebrate vision and publishes experimental and observational studies, reviews, and theoretical and computational analyses. Vision Research also publishes clinical studies relevant to normal visual function and basic research relevant to visual dysfunction or its clinical investigation. Functional aspects of vision is interpreted broadly, ranging from molecular and cellular function to perception and behavior. Detailed descriptions are encouraged but enough introductory background should be included for non-specialists. Theoretical and computational papers should give a sense of order to the facts or point to new verifiable observations. Papers dealing with questions in the history of vision science should stress the development of ideas in the field.
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