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黄斑营养不良:临床和影像学特征、分子遗传学和治疗选择。

Macular dystrophies: clinical and imaging features, molecular genetics and therapeutic options.

机构信息

Moorfields Eye Hospital, London, UK.

Institute of Ophthalmology, UCL, London, UK.

出版信息

Br J Ophthalmol. 2020 Apr;104(4):451-460. doi: 10.1136/bjophthalmol-2019-315086. Epub 2019 Nov 8.

DOI:10.1136/bjophthalmol-2019-315086
PMID:31704701
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7147237/
Abstract

Macular dystrophies (MDs) consist of a heterogeneous group of disorders that are characterised by bilateral symmetrical central visual loss. Advances in genetic testing over the last decade have led to improved knowledge of the underlying molecular basis. The developments in high-resolution multimodal retinal imaging have also transformed our ability to make accurate and more timely diagnoses and more sensitive quantitative assessment of disease progression, and allowed the design of optimised clinical trial endpoints for novel therapeutic interventions. The aim of this review was to provide an update on MDs, including Stargardt disease, Best disease, X-linked r etinoschisis, pattern dystrophy, Sorsby fundus dystrophy and autosomal dominant drusen. It highlights the range of innovations in retinal imaging, genotype-phenotype and structure-function associations, animal models of disease and the multiple treatment strategies that are currently in clinical trial or planned in the near future, which are anticipated to lead to significant changes in the management of patients with MDs.

摘要

黄斑营养不良(MDs)是一组异质性疾病,其特征是双侧对称的中心视力丧失。过去十年中,遗传检测技术的进步使我们对潜在的分子基础有了更好的了解。高分辨率多模态视网膜成像的发展也改变了我们进行准确和及时诊断以及更敏感地评估疾病进展的能力,并允许为新型治疗干预设计优化的临床试验终点。本文旨在对 MDs 进行更新,包括 Stargardt 病、Best 病、X 连锁性视网膜劈裂症、图形性营养不良、Sorsby 眼底营养不良和常染色体显性玻璃膜疣病。本文强调了视网膜成像、基因型-表型和结构-功能关联、疾病动物模型以及目前正在临床试验或计划在不久的将来进行的多种治疗策略方面的一系列创新,预计这些创新将对 MDs 患者的治疗产生重大影响。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/9053a89694ef/bjophthalmol-2019-315086f05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/04dfed3d1db5/bjophthalmol-2019-315086f01.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/ade283295e28/bjophthalmol-2019-315086f03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/58150dda4f92/bjophthalmol-2019-315086f04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/9053a89694ef/bjophthalmol-2019-315086f05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/04dfed3d1db5/bjophthalmol-2019-315086f01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/0e15a25990b8/bjophthalmol-2019-315086f02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/ade283295e28/bjophthalmol-2019-315086f03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/58150dda4f92/bjophthalmol-2019-315086f04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11ff/7147237/9053a89694ef/bjophthalmol-2019-315086f05.jpg

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A Quantitative Chloride Channel Conductance Assay for Efficacy Testing of AAV.BEST1.用于AAV.BEST1功效测试的定量氯离子通道电导测定法
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