通过高通量分子诊断方法在两名罹患晚期婴幼儿变色性白质营养不良症的女孩身上发现新型 ARSA 基因突变

IF 4.3 3区 材料科学 Q1 ENGINEERING, ELECTRICAL & ELECTRONIC ACS Applied Electronic Materials Pub Date : 2023-12-01 Epub Date: 2023-09-08 DOI:10.1007/s12017-023-08757-y
Abolfazl Yari, Farzane Vafaeie, Zahra Miri Karam, Mahya Hosseini, Hassan Hashemzade, Maryam Sadat Rahimi, Alireza Ehsanbakhsh, Ebrahim Miri-Moghaddam
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引用次数: 0

摘要

变色性白质营养不良症(MLD)是一种罕见的白质脑病,由 ARSA 基因的致病突变引起。它表现为严重的运动症状、精神问题,有时还会出现癫痫发作。我们的目的是调查一个伊朗家庭中 MLD 的表型表现和遗传原因。本病例中,一名 3 岁女童表现为肌张力低下、肌肉萎缩和癫痫发作。我们对她进行了神经系统和神经肌肉检查,以评估其临床特征。全外显子组测序(WES)用于检测致病变异。为预测该变异的致病性,进行了硅学分析。利用GROMACS软件进行了分子动态模拟(MDS)。神经学研究显示,运动传导速度明显减慢,运动单位动作电位持续时间延长。脑磁共振成像扫描显示白质异常。通过应用 WES,我们在 ARSA 中发现了一个新的同源错义变异(NM_000487.6,c.938G > C,p.R313P)。直接测序在她无症状的妹妹身上发现了这一同源变异,而父母双方都携带杂合变异。这种变异在遗传数据库或文献中均未见报道。硅学分析预测,该 DNA 位置的任何变异都会导致疾病,因为它具有高度保守性。根据 ACMG/AMP 指南,c.938G>C 变异被归类为致病变异。MDS 分析表明,c.938G > C 对 ARSA 的结构和稳定性都有重大影响,最终导致蛋白质功能受损。该变异的发现扩大了与 MLD 相关的 ARSA 基因突变的范围,并强调了基因检测对 MLD 诊断的重要性。
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Identification of a Novel ARSA Gene Mutation Through High-Throughput Molecular Diagnosis Method in Two Girls with Late Infantile Metachromatic Leukodystrophy.

Metachromatic leukodystrophy (MLD) is a rare leukoencephalopathy caused by pathogenic mutations in the ARSA gene. It manifests as severe motor symptoms, mental problems, and sometimes, seizures. We aimed to investigate the phenotypic manifestations and genetic causes of MLD in an Iranian family. We present the case of a 3-year-old girl who presented with hypotonia, muscular atrophy, and seizures. Neurological and neuromuscular examinations were performed to evaluate clinical characteristics. Whole exome sequencing (WES) was used to detect disease-causing variants. In silico analysis was performed to predict the pathogenicity of this variant. GROMACS software was utilized for molecular dynamic simulation (MDS). Neurological studies revealed marked slowing of motor conduction velocities and an increased motor unit action potential duration. Brain MRI scan revealed white matter abnormalities. By applying WES, we identified a novel homozygous missense variant (NM_000487.6, c.938G > C, p.R313P) in ARSA. Direct sequencing identified this homozygous variant in her asymptomatic younger sister, whereas both parents carried a heterozygous variant. This mutation has not been reported in genetic databases or in literature. In silico analysis predicted that any variation in this DNA position would cause disease, as it is highly conserved. The c.938G > C variant was classified as a pathogenic variant according to ACMG/AMP guidelines. MDS analysis indicated that c.938G > C had a significant impact on both the structure and stabilization of ARSA, ultimately resulting in impaired protein function. The identification of this variant expands the spectrum of ARSA gene mutations associated with MLD and highlights the importance of genetic testing for the diagnosis of MLD.

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