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阿尔及利亚患者队列中综合征性身材矮小的基因组基础。

Genomic basis of syndromic short stature in an Algerian patient cohort.

机构信息

Clinical Genetics, Tygerberg Hospital and Division of Molecular Biology and Human Genetics, Stellenbosch University, Cape Town, South Africa.

Institute of Human Genetics, University Medical Center Göttingen, Göttingen, Germany.

出版信息

Am J Med Genet A. 2022 Feb;188(2):606-612. doi: 10.1002/ajmg.a.62532. Epub 2021 Oct 13.

DOI:10.1002/ajmg.a.62532
PMID:34644002
Abstract

Short stature is one of the most common reasons for a referral to the pediatric endocrinology clinic. Thousands of patients with short stature are assessed annually at the Department of Endocrine and Metabolic Diseases (DEMD) at Bab el Oued University Hospital in Algiers, Algeria. However, diagnostic rates in patients with syndromic short stature are not optimal due to the unavailability of next generation sequencing (NGS) technology. Here, we enrolled 10 Algerian patients with syndromic short stature in a pilot study to test the impact of genetic and genomic approaches in the DEMD. Using a combination of two different NGS modalities, namely exome sequencing and the Mendeliome (TruSight™ One sequencing panel) along with single gene testing, we were able to establish a confirmed molecular diagnosis in 7/10 patients (70%) and to identify strong likely disease-causing variants in a further two patients. Novel variants in NPR2 and VPS13B were identified. Using copy number variation analysis on the exome data, we also identified a de novo deletion of the short arm of chromosome X. These definitive diagnoses have made a substantial impact on patient treatment, management and genetic counseling. Genomic testing has the ability to transform clinical practice, and is an essential diagnostic tool in any tertiary pediatric clinic, particularly in resource limited settings.

摘要

身材矮小是转诊儿科内分泌科的最常见原因之一。在阿尔及利亚阿尔及尔 Bab el Oued 大学医院的内分泌和代谢疾病科(DEMD),每年都会评估数千名身材矮小的患者。然而,由于缺乏下一代测序(NGS)技术,综合征性身材矮小患者的诊断率并不理想。在这里,我们在一项试点研究中招募了 10 名患有综合征性身材矮小的阿尔及利亚患者,以测试遗传和基因组方法在 DEMD 中的影响。使用两种不同的 NGS 方式(外显子组测序和 Mendeliome(TruSight One 测序面板)以及单基因测试相结合,我们能够在 7/10 名患者(70%)中建立明确的分子诊断,并在另外两名患者中鉴定出强烈的可能致病变异。鉴定到 NPR2 和 VPS13B 的新变异。我们还通过外显子组数据的拷贝数变异分析,鉴定到 X 染色体短臂的新生缺失。这些明确的诊断对患者的治疗、管理和遗传咨询产生了重大影响。基因组测试有能力改变临床实践,并且是任何三级儿科诊所的重要诊断工具,特别是在资源有限的环境中。

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