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利用多模态成像和下一代测序技术对一大群视网膜色素变性患者的临床特征和遗传变异进行分析。

Clinical Characteristics and Genetic Variants of a Large Cohort of Patients with Retinitis Pigmentosa Using Multimodal Imaging and Next Generation Sequencing.

机构信息

Department of Ophthalmology and Visual Neurosciences, University of Minnesota, Minneapolis, MN 55455, USA.

出版信息

Int J Mol Sci. 2023 Jun 30;24(13):10895. doi: 10.3390/ijms241310895.

Abstract

This retrospective study identifies patients with RP at the Inherited Retinal Disease Clinic at the University of Minnesota (UMN)/M Health System who had genetic testing via next generation sequencing. A database was curated to record history and examination, genetic findings, and ocular imaging. Causative pathogenic and likely pathogenic variants were recorded. Disease status was further characterized by ocular coherence tomography (OCT) and fundus autofluorescence (AF). Our study cohort included a total of 199 patients evaluated between 1 May 2015-5 August 2022. The cohort included 151 patients with non-syndromic RP and 48 with syndromic RP. Presenting symptoms included nyctalopia (85.4%) photosensitivity/hemeralopia (60.5%), and decreased color vision (55.8%). On average, 38.9% had visual acuity of worse than 20/80. Ellipsoid zone band width on OCT scan of less than 1500 μm was noted in 73.6%. Ninety-nine percent had fundus autofluorescence (AF) findings of a hypo- or hyper-fluorescent ring within the macula and/or peripheral hypo-AF. Of the 127 subjects who underwent genetic testing, a diagnostic pathogenic and/or likely pathogenic variant was identified in 67 (52.8%) patients-33.3% of syndromic RP and 66.6% of non-syndromic RP patients had a diagnostic gene variant identified. It was found that 23.6% of the cohort had negative genetic testing results or only variants of uncertain significance identified, which were deemed as non-diagnostic. We concluded that patients with RP often present with advanced disease. In our population, next generation sequencing panels identified a genotype consistent with the exam in just over half the patients. Additional work will be needed to identify the underlying genetic etiology for the remainder.

摘要

这项回顾性研究在明尼苏达大学(UMN)/M 健康系统的遗传性视网膜疾病诊所识别了接受下一代测序基因检测的 RP 患者。建立了一个数据库来记录病史和检查、遗传发现和眼部成像。记录了致病和可能致病的变体。通过眼部相干断层扫描(OCT)和眼底自发荧光(AF)进一步确定疾病状态。我们的研究队列包括 2015 年 5 月 1 日至 2022 年 8 月 5 日期间评估的总共 199 名患者。该队列包括 151 名非综合征性 RP 患者和 48 名综合征性 RP 患者。首发症状包括夜盲症(85.4%)、光敏感/昼盲(60.5%)和色觉减退(55.8%)。平均而言,38.9%的患者视力低于 20/80。OCT 扫描的椭圆体带宽度小于 1500μm 占 73.6%。99%的患者眼底自发荧光(AF)显示黄斑区和/或周边低自发荧光呈低荧光或高荧光环。在接受基因检测的 127 名受试者中,67 名(52.8%)患者发现了诊断性致病性和/或可能致病性变异-33.3%的综合征性 RP 和 66.6%的非综合征性 RP 患者发现了诊断性基因变异。发现 23.6%的患者基因检测结果为阴性或仅发现意义不明的变异,被认为是非诊断性的。我们得出结论,RP 患者常出现晚期疾病。在我们的人群中,下一代测序面板仅在超过一半的患者中识别出与检查一致的基因型。需要进一步的工作来确定其余患者的潜在遗传病因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4f00/10341662/7fca2422a2ea/ijms-24-10895-g001.jpg

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