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早产儿视网膜病变患者的眼部表型与基因分析。

Ocular phenotype and genetical analysis in patients with retinopathy of prematurity.

机构信息

Department of Ophthalmology, Peking University People's Hospital, Eye Diseases and Optometry Institute, Beijing, China.

Beijing Key Laboratory of Diagnosis and Therapy of Retinal and Choroid Diseases, Beijing, China.

出版信息

BMC Ophthalmol. 2022 Jan 12;22(1):22. doi: 10.1186/s12886-022-02252-x.

Abstract

BACKGROUND

Retinopathy of prematurity (ROP) is a multifactorial retinal disease, involving both environmental and genetic factors; The purpose of this study is to evaluate the clinical presentations and genetic variants in Chinese patients with ROP.

METHODS

A total of 36 patients diagnosed with ROP were enrolled in this study, their medical and ophthalmic histories were obtained, and comprehensive clinical examinations were performed. Genomic DNA was isolated from peripheral blood of ROP patients, polymerase chain reaction and direct sequencing of the associated pathogenic genes (FZD4, TSPAN12, and NDP) were performed.

RESULTS

All patients exhibited the clinical manifestations of ROP. No mutations were detected in the TSPAN12 and NDP genes in all patients; Interestingly, three novel missense mutations were identified in the FZD4 gene (p.A2P, p.L79M, and p.Y378C) in four patients, for a detection rate of 11.1% (4/36).

CONCLUSIONS

This study expands the genotypic spectrum of FZD4 gene in ROP patients, and our findings underscore the importance of obtaining molecular analyses and comprehensive health screening for this retinal disease.

摘要

背景

早产儿视网膜病变(ROP)是一种多因素视网膜疾病,涉及环境和遗传因素;本研究旨在评估中国 ROP 患者的临床表型和遗传变异。

方法

本研究共纳入 36 例诊断为 ROP 的患者,获取其病史和眼科病史,并进行全面的临床检查。从 ROP 患者外周血中提取基因组 DNA,对相关致病基因(FZD4、TSPAN12 和 NDP)进行聚合酶链反应和直接测序。

结果

所有患者均表现出 ROP 的临床表现。在所有患者中均未检测到 TSPAN12 和 NDP 基因的突变;有趣的是,在 4 名患者中发现了 FZD4 基因的三个新的错义突变(p.A2P、p.L79M 和 p.Y378C),检出率为 11.1%(4/36)。

结论

本研究扩展了 ROP 患者 FZD4 基因突变的基因型谱,我们的研究结果强调了对这种视网膜疾病进行分子分析和全面健康筛查的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6b3e/8753894/279772f06949/12886_2022_2252_Fig1_HTML.jpg

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