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斯洛文尼亚眼皮肤白化病患者队列的遗传变异性。

Genetic Variability in Slovenian Cohort of Patients with Oculocutaneous Albinism.

出版信息

Acta Chim Slov. 2021 Sep;68(3):683-692.

Abstract

Oculocutaneous albinism (OCA) is an inherited disorder affecting the visual system and skin pigmentation. Our aim was to evaluate genetic and clinical heterogeneity in a cohort of Slovenian paediatric patients with clinically suspected OCA using advanced molecular-genetics approach. In as much as 20 out of 25 patients, genetic variants explaining their clinical phenotype were identified. The great majority of patients (15/25) had genetic variants in TYR gene associated with OCA type 1, followed by variants in TYRP1, SLC45A2 and HPS1 genes causative for OCA3, OCA4 and Hermansky-Pudlak syndrome type 1, respectively. We concluded that OCA phenotype could not predict genotype and vice versa. Nevertheless, the diagnostic yield after targeted next generation sequencing (NGS) was 80% and proved to be affective in our paediatric cohort of patients with various degree of OCA. Even in 16 patients with normal complexion the diagnostic yield was 62,5%. Interestingly, we have identified a patient of white European ancestry with OCA3, which is an extremely rare report, and one patient with OCA due to the Hermansky-Pudlak syndrome type 1.

摘要

眼皮肤白化病(OCA)是一种影响视觉系统和皮肤色素沉着的遗传性疾病。我们的目的是使用先进的分子遗传学方法评估一组疑似 OCA 的斯洛文尼亚儿科患者的遗传和临床异质性。在 25 名患者中的 20 名中,鉴定出了导致其临床表型的遗传变异。绝大多数患者(15/25)的 TYR 基因遗传变异与 OCA 1 型相关,其次是 TYRP1、SLC45A2 和 HPS1 基因的变异,分别导致 OCA3、OCA4 和 Hermansky-Pudlak 综合征 1 型。我们得出的结论是,OCA 表型不能预测基因型,反之亦然。尽管如此,靶向下一代测序(NGS)的诊断率仍达到 80%,并在我们的具有不同程度 OCA 的儿科患者群体中证明是有效的。即使在 16 名肤色正常的患者中,诊断率也达到了 62.5%。有趣的是,我们鉴定出一名具有 OCA3 的白人欧洲血统患者,这是一个极其罕见的报告,以及一名患有 Hermansky-Pudlak 综合征 1 型的 OCA 患者。

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