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描绘与RNU4ATAC相关的剪接体病的表型。

Delineating the phenotype of RNU4ATAC-related spliceosomopathy.

作者信息

Tabib Amanda, Richmond Christopher M, McGaughran Julie

机构信息

Paediatrics, John Hunter Children's Hospital, Newcastle, New South Wales, Australia.

Genetic Health QLD, Royal Brisbane & Women's Hospital, Herston, Queensland, Australia.

出版信息

Am J Med Genet A. 2023 Apr;191(4):1094-1100. doi: 10.1002/ajmg.a.63110. Epub 2023 Jan 9.

Abstract

Biallelic pathogenic variants in RNU4ATAC cause microcephalic osteodysplastic primordial dwarfism type I (MOPD1), Roifman syndrome (RS) and Lowry-Wood syndrome (LWS). These conditions demonstrate significant phenotypic heterogeneity yet have overlapping features. Although historically described as discrete conditions they appear to represent a phenotypic spectrum with clinical features not always aligning with diagnostic categories. Clinical variability and ambiguity in diagnostic criteria exist among each disorder. Here we report an individual with a novel genotype and phenotype spanning all three disorders, expanding the phenotypic spectrum of RNU4ATAC-related spliceosomeopathies.

摘要

RNU4ATAC基因的双等位基因致病变异可导致I型小头骨发育异常原发性侏儒症(MOPD1)、罗夫曼综合征(RS)和劳里-伍德综合征(LWS)。这些病症表现出显著的表型异质性,但具有重叠特征。尽管历史上被描述为不同的病症,但它们似乎代表了一个表型谱,其临床特征并不总是与诊断类别一致。每种疾病在临床变异性和诊断标准的模糊性方面都存在问题。在此,我们报告了一名具有跨越所有三种疾病的新型基因型和表型的个体,扩展了RNU4ATAC相关剪接体病的表型谱。

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