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TEK 基因相关性原发性先天性青光眼:10 个无关家系的墨西哥队列的表型特征和突变谱。

TEK gene-related primary congenital glaucoma: Phenotypic features and mutational spectrum in a Mexican cohort of 10 unrelated families.

机构信息

Department of Genetics, Institute of Ophthalmology "Conde de Valenciana", Mexico City, Mexico.

Laboratorio 5 Edificio A-4, Carrera de Médico Cirujano, Facultad de Estudios Superiores Iztacala, Universidad Nacional Autónoma de, Mexico City, Mexico.

出版信息

Am J Med Genet A. 2024 Oct;194(10):e63716. doi: 10.1002/ajmg.a.63716. Epub 2024 Jun 7.

Abstract

Primary congenital glaucoma (PCG) is one of the leading causes of visual damage and blindness, severely affecting the quality of life of affected children. It is characterized by cupping of the optic disc and loss of ganglion cells due to elevated intraocular pressure. While most PCG patients exhibit epiphora, photophobia, and buphthalmos with corneal opacity, variability in phenotypic manifestations is not uncommon. Prompt diagnosis and treatment of PCG affected individuals becomes relevant to preserve visual function throughout their lives. Most PCG cases are sporadic or autosomal recessive; however, an incompletely dominant autosomal dominant form arising from mutations in the TEK gene has recently been demonstrated. Here, we describe the clinical and mutational features of a cohort of Mexican patients with TEK-related PCG. Our results support the involvement of the TEK gene as an important cause of the disease in our ethnic group and expand the mutational spectrum causing PCG by reporting 10 novel disease-causing variants.

摘要

原发性先天性青光眼(PCG)是导致视觉损伤和失明的主要原因之一,严重影响了受影响儿童的生活质量。其特征是由于眼内压升高导致视盘凹陷和神经节细胞丧失。虽然大多数 PCG 患者表现出溢泪、畏光和角膜混浊的牛眼,但表型表现的变异性并不少见。及时诊断和治疗 PCG 患者对于在其一生中保持视力功能至关重要。大多数 PCG 病例是散发性或常染色体隐性遗传的;然而,最近已经证明了一种来自 TEK 基因突变的不完全显性常染色体显性形式。在这里,我们描述了一组墨西哥 TEK 相关性 PCG 患者的临床和突变特征。我们的结果支持 TEK 基因作为我们族群中该疾病的重要原因,并通过报告 10 种新的致病变体扩展了导致 PCG 的突变谱。

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