沙特家族DHX38基因新错义变异C.2571 (P.Ala857=)导致常染色体隐性视网膜色素变性。

IF 0.5 Q4 OPHTHALMOLOGY
Middle East African Journal of Ophthalmology Pub Date : 2022-04-30 eCollection Date: 2021-10-01 DOI:10.4103/meajo.meajo_40_21
Saud Al-Johani, Abdulelah Alabdullah, Sawsan R Nowilaty
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引用次数: 1

摘要

我们提出了两例新的错义变异突变的DHX38基因,这与常染色体隐性视网膜色素变性(RP)有关的两个沙特姐妹谁自童年以来表现为视力差。经初步检查,两姐妹双眼的最佳矫正视力为20/300。眼底检查显示广泛的视网膜色素改变,线性周围色素沉着团块,骨刺,双侧视神经结节伴双侧黄斑色素沉着。光谱域光学相干断层扫描显示视网膜外层的损失和视网膜下纤维化和脉络膜变薄的存在。DHX38外显子组的分子测序分析发现了纯合子变异c. 2571 (p. Ala857=)的一个新的错义突变,该突变与编码premRNA剪接因子PRP16的常染色体隐性RP基因共分离。本报告的目的是描述与该变异相关的临床特征,并提供DHX38参与RP的额外证据。据我们所知,文献中没有描述过这种变体。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

A Novel Missense Variant C.2571 (P.Ala857=) of the DHX38 Gene in a Saudi Family Causes an Autosomal Recessive Retinitis Pigmentosa.

A Novel Missense Variant C.2571 (P.Ala857=) of the DHX38 Gene in a Saudi Family Causes an Autosomal Recessive Retinitis Pigmentosa.

A Novel Missense Variant C.2571 (P.Ala857=) of the DHX38 Gene in a Saudi Family Causes an Autosomal Recessive Retinitis Pigmentosa.

We present two cases of a novel missense variant mutation in the DHX38 gene, which is associated with autosomal recessive retinitis pigmentosa (RP) in two Saudi sisters who presented with poor visual acuity since childhood. On initial examination, the best-corrected visual acuity was 20/300 in both eyes for the two sisters. Fundus examination revealed widespread retinal pigmentary changes, linear peripheral hyperpigmentation clumps, bone spicules, and bilateral optic nerve drusen with bilateral macular hyperpigmentation. Spectral-domain optical coherence tomography scans reveal losses of the outer retinal layer and the presence of subretinal fibrosis and thinning of the choroid. Molecular sequencing analysis of the DHX38 exome identified a novel missense mutation of the homozygous variant c. 2571 (p. Ala857=), which co-segregates with the autosomal recessive RP gene that encodes the premRNA splicing factor, PRP16. The aim of this report is to describe the clinical feature associated with this variant and to provide additional evidence that DHX38 is involved in RP. To the best of our knowledge, this variant has not been described in the literature.

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来源期刊
CiteScore
1.40
自引率
0.00%
发文量
1
期刊介绍: The Middle East African Journal of Ophthalmology (MEAJO), published four times per year in print and online, is an official journal of the Middle East African Council of Ophthalmology (MEACO). It is an international, peer-reviewed journal whose mission includes publication of original research of interest to ophthalmologists in the Middle East and Africa, and to provide readers with high quality educational review articles from world-renown experts. MEAJO, previously known as Middle East Journal of Ophthalmology (MEJO) was founded by Dr Akef El Maghraby in 1993.
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