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一名患有德桑托-希纳维综合征患者的黄斑病变

Torpedo maculopathy in a patient with DeSanto-Shinawi syndrome.

作者信息

Petroni Sergio, Catena Gino, Iarossi Giancarlo, Federici Matteo, Zinzanella Gaetano, De Sanctis Carlo Maria, Valente Paola, Buzzonetti Luca

机构信息

Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.

出版信息

Eur J Ophthalmol. 2025 May;35(3):NP7-NP9. doi: 10.1177/11206721251313840. Epub 2025 Feb 12.

Abstract

IntroductionTorpedo maculopathy (TM) is a rare, typically benign and congenital anomaly of the retinal pigment epithelium (RPE) characterized by a torpedo-shaped lesion in the macula. DeSanto-Shinawi syndrome (DESS) is an equally rare genetic disorder caused by mutations in the WAC gene, presenting with intellectual disability, dysmorphic features, and growth retardation.Case descriptionThis report illustrates the case of monolateral TM in a patient with DESS. The genetic and phenotypic characteristic were described and correlated with retinal features, assessed by means of retinography, spectral domain OCT (SD-OCT) and fluorescein angiography (FA).ConclusionThis case report discusses the unusual co-occurrence of TM in a patient diagnosed with DESS, contributing to the sparse literature on this association and expanding the phenotypic spectrum of both conditions.

摘要

引言

黄斑部鱼雷样病变(TM)是一种罕见的、通常为良性的先天性视网膜色素上皮(RPE)异常,其特征为黄斑区出现鱼雷样病变。德桑托 - 希纳维综合征(DESS)是一种同样罕见的由WAC基因突变引起的遗传性疾病,表现为智力障碍、畸形特征和生长发育迟缓。

病例描述

本报告阐述了一名患有DESS的患者单侧TM的病例。描述了其遗传和表型特征,并将其与通过视网膜造影、光谱域光学相干断层扫描(SD - OCT)和荧光素血管造影(FA)评估的视网膜特征进行了关联。

结论

本病例报告讨论了在一名被诊断为DESS的患者中TM的不寻常同时出现,这为关于这种关联的稀少文献做出了贡献,并扩展了这两种病症的表型谱。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8b89/12012276/884da2057d26/10.1177_11206721251313840-fig1.jpg

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