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在一个土耳其家族中发现了一种(c.869-1G>A)PSEN1 基因剪接变异,该家族存在一种罕见的情况:伴有痉挛性截瘫的变异型阿尔茨海默病。该家族具有临床和分子学发现。

Clinical and Molecular Findings in a Turkish Family Who Had a (c.869- 1G>A) Splicing Variant in PSEN1 Gene with A Rare Condition: The Variant Alzheimer's Disease with Spastic Paraparesis.

机构信息

Department of Medical Genetics, Malatya Turgut Ozal University Medical Faculty, Malatya, Turkey.

Department of Medical Genetics, Aksaray University Medical Faculty, Aksaray, Turkey.

出版信息

Curr Alzheimer Res. 2022;19(3):223-235. doi: 10.2174/1567205019666220414101251.

DOI:10.2174/1567205019666220414101251
PMID:35430993
Abstract

BACKGROUND

Early-onset Alzheimer's disease (EOAD) is commonly diagnosed with an onset age of earlier than 65 years and accounts for 5-10% of all Alzheimer's disease (AD) cases. To date, although only 10-15% of familial EOAD cases have been explained, the genetic cause of the vast proportion of cases has not been explained. The variant Alzheimer's disease with spastic paraparesis (var- AD) is defined as a rare clinical entity characterized by early-onset dementia, spasticity of the lower extremities, and gait disturbance. Although the disease was first associated with variants in exon 9 of the PSEN1 gene, it was later shown that variations in other exons were also responsible for the disease.

OBJECTIVE

The current study aims to raise awareness of varAD, which occurs as a rare phenotype due to pathogenic variants in PSEN1. In addition, we aimed to evaluate the spectrum of mutations in varAD patients identified to date.

METHODS

Detailed family histories and clinical data were recorded. Whole exome sequencing was performed and co-segregation analysis of the family was done by Sanger sequencing. Also, a review of the molecularly confirmed patients with (varAD) from the literature was evaluated.

RESULTS

We identified a heterozygous splicing variant (c.869-1G>A) in the PSEN1 gene, in a family with two affected individuals who present with varAD. We reported the clinical and genetic findings from the affected individuals.

CONCLUSION

We present the detailed clinical and genetic profiles of a Turkish patient with the diagnosis of varAD together with subjects from the literature. Together, we think that the clinical characteristics and the effect of the (c.869-1G>A) variant will facilitate our understanding of the PSEN1 gene in AD pathogenesis.

摘要

背景

早发性阿尔茨海默病(EOAD)通常以发病年龄早于 65 岁为诊断标准,占所有阿尔茨海默病(AD)病例的 5-10%。迄今为止,尽管只有 10-15%的家族性 EOAD 病例得到了解释,但绝大多数病例的遗传原因尚未得到解释。以痉挛性截瘫为特征的变异型阿尔茨海默病(var-AD)定义为一种罕见的临床实体,其特征为早发性痴呆、下肢痉挛和步态障碍。虽然该疾病最初与 PSEN1 基因外显子 9 的变异有关,但后来表明其他外显子的变异也与该疾病有关。

目的

本研究旨在提高对 varAD 的认识,varAD 是由于 PSEN1 中的致病变异而导致的罕见表型。此外,我们旨在评估迄今为止发现的 varAD 患者的突变谱。

方法

详细记录家族史和临床数据。进行全外显子组测序,并通过 Sanger 测序对家系进行共分离分析。还对文献中分子确诊的(varAD)患者进行了综述。

结果

我们在一个有两个受影响个体的家族中发现了 PSEN1 基因中的杂合剪接变异(c.869-1G>A),该家族患有 varAD。我们报告了受影响个体的临床和遗传发现。

结论

我们一起呈现了一名土耳其患者与文献中主题的详细临床和遗传特征,诊断为 varAD。我们认为,(c.869-1G>A)变异的临床特征和影响将有助于我们理解 PSEN1 基因在 AD 发病机制中的作用。

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