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通过全外显子组测序破译儿科患者未确诊的眼部异常及全身改变的病因。

Deciphering the etiology of undiagnosed ocular anomalies along with systemic alterations in pediatric patients through whole exome sequencing.

机构信息

Laboratorio de Biología Molecular, Subdirección de Investigación Médica, Instituto Nacional de Pediatría, Mexico City, Mexico.

Laboratorio de Biomoléculas y Salud Infantil, Instituto Nacional de Pediatría, Mexico City, México.

出版信息

Sci Rep. 2024 Jun 22;14(1):14380. doi: 10.1038/s41598-024-65227-6.

Abstract

Inherited and developmental eye diseases are quite diverse and numerous, and determining their genetic cause is challenging due to their high allelic and locus heterogeneity. New molecular approaches, such as whole exome sequencing (WES), have proven to be powerful molecular tools for addressing these cases. The present study used WES to identify the genetic etiology in ten unrelated Mexican pediatric patients with complex ocular anomalies and other systemic alterations of unknown etiology. The WES approach allowed us to identify five clinically relevant variants in the GZF1, NFIX, TRRAP, FGFR2 and PAX2 genes associated with Larsen, Malan, developmental delay with or without dysmorphic facies and autism, LADD1 and papillorenal syndromes. Mutations located in GZF1 and NFIX were classified as pathogenic, those in TRRAP and FGFR2 were classified as likely pathogenic variants, and those in PAX2 were classified as variants of unknown significance. Protein modeling of the two missense FGFR2 p.(Arg210Gln) and PAX2 p.(Met3Thr) variants showed that these changes could induce potential structural alterations in important functional regions of the proteins. Notably, four out of the five variants were not previously reported, except for the TRRAP gene. Consequently, WES enabled the identification of the genetic cause in 40% of the cases reported. All the syndromes reported herein are very rare, with phenotypes that may overlap with other genetic entities.

摘要

遗传性和发育性眼病种类繁多,由于其等位基因和基因座异质性高,确定其遗传原因具有挑战性。新的分子方法,如外显子组测序(WES),已被证明是解决这些病例的有力分子工具。本研究使用 WES 鉴定了 10 名无亲缘关系的墨西哥儿科患者的遗传病因,这些患者具有复杂的眼部异常和其他未知病因的全身改变。WES 方法使我们能够在与 Larsen、Malan、伴有或不伴有畸形面容和自闭症、LADD1 和乳头状肾综合征相关的 GZF1、NFIX、TRRAP、FGFR2 和 PAX2 基因中发现五个与临床相关的变体。位于 GZF1 和 NFIX 中的突变被归类为致病性,位于 TRRAP 和 FGFR2 中的突变为可能致病的变体,位于 PAX2 中的突变为意义不明的变体。对两种错义 FGFR2 p.(Arg210Gln)和 PAX2 p.(Met3Thr)变体的蛋白质建模表明,这些变化可能会导致蛋白质重要功能区域的潜在结构改变。值得注意的是,除了 TRRAP 基因外,这五个变体中有四个以前没有报道过。因此,WES 能够在 40%的报告病例中确定遗传原因。本文报道的所有综合征都非常罕见,其表型可能与其他遗传实体重叠。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/658f/11193775/22a9334e0f57/41598_2024_65227_Fig1_HTML.jpg

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