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具有假性玻璃膜疣样外观的广泛黄斑萎缩(EMAP)的临床特征、诊断标准以及来自相关遗传性视网膜疾病和年龄相关性黄斑变性的见解

Extensive macular atrophy with pseudodrusen-like appearance (EMAP) clinical characteristics, diagnostic criteria, and insights from allied inherited retinal diseases and age-related macular degeneration.

作者信息

Antropoli Alessio, Bianco Lorenzo, Romano Francesco, Trinco Andrea, Arrigo Alessandro, Benadji Amine, Atia Raphaël, Palacci Oana, Dagostinoz Dorothée, Devisme Céline, Condroyer Christel, Antonio Aline, Bosello Francesca, Casati Stefano, Salvetti Anna Paola, Zaffalon Chiara, Gaudric Alain, Sahel José-Alain, Staurenghi Giovanni, Bandello Francesco, Sennlaub Florian, Zeitz Christina, Meunier Isabelle, Battaglia Parodi Maurizio, Audo Isabelle

机构信息

Sorbonne Université, INSERM, CNRS, Institut de la Vision, Paris, France; CHNO des Quinze-Vingts, Centre de Référence Maladies Rares REFERET and DHU Sight Restore, INSERM-DGOS CIC1423, Paris, France; Department of Ophthalmology, IRCCS San Raffaele Scientific Institute, Milan, Italy.

Eye Repair Unit, Division of Neuroscience, IRCCS San Raffaele Scientific Institute, Milan, Italy.

出版信息

Prog Retin Eye Res. 2025 Jan;104:101320. doi: 10.1016/j.preteyeres.2024.101320. Epub 2024 Nov 25.

Abstract

Extensive macular atrophy with pseudodrusen-like appearance (EMAP) was first described in France in 2009 as a symmetric and rapidly progressive form of macular atrophy primarily affecting middle-aged individuals. Despite the recent identification of a significant number of cases in Italy and worldwide, EMAP remains an underrecognized condition. The clinical triad typical of EMAP consists of vertically oriented macular atrophy with multilobular borders, pseudodrusen-like deposits across the posterior pole and mid-periphery, and peripheral pavingstone degeneration. Nonetheless, recent research has portrayed EMAP as a highly stage-dependent condition, allowing the identification of novel disease hallmarks, including a diffuse separation between the Bruch's membrane and the retinal pigment epithelium, along with consistent sparing of a region temporal to the macula. Additionally, retinal electrophysiology is particularly useful in distinguishing EMAP from age-related macular degeneration (AMD). Supported by unpublished data from the largest EMAP cohorts worldwide, this review aims to provide a comprehensive and updated description of EMAP, now recognized as a severely blinding disease characterized by diffuse chorioretinal atrophy and photoreceptor dysfunction. Furthermore, we propose a set of diagnostic criteria that incorporate clinical, imaging, and functional tests, to facilitate the recognition of this clinical entity. Lastly, we aim to shed light on its pathogenesis by comparing it with AMD and monogenic retinal disorders exhibiting similar phenotypes.

摘要

广泛黄斑萎缩伴假性玻璃膜疣样外观(EMAP)于2009年在法国首次被描述,是一种主要影响中年人的对称性且快速进展的黄斑萎缩形式。尽管最近在意大利和全球范围内发现了大量病例,但EMAP仍然是一种未被充分认识的疾病。EMAP典型的临床三联征包括边界呈多叶状的垂直性黄斑萎缩、后极部和中周边部的假性玻璃膜疣样沉积物以及周边铺路石样变性。然而,最近的研究将EMAP描述为一种高度依赖分期的疾病,这使得能够识别新的疾病特征,包括布鲁赫膜与视网膜色素上皮之间的弥漫性分离,以及黄斑颞侧区域的持续 spared 。此外,视网膜电生理在区分EMAP与年龄相关性黄斑变性(AMD)方面特别有用。在全球最大的EMAP队列的未发表数据支持下,本综述旨在对EMAP进行全面且更新的描述,EMAP现在被认为是一种以弥漫性脉络膜视网膜萎缩和光感受器功能障碍为特征的严重致盲疾病。此外,我们提出了一套结合临床、影像学和功能测试的诊断标准,以促进对这一临床实体的识别。最后,我们旨在通过将其与表现出相似表型的AMD和单基因视网膜疾病进行比较,来阐明其发病机制。

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