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导致具有家族内异质性的肌病的新型剪接 dysferlin 突变。

Novel splicing dysferlin mutation causing myopathy with intra-familial heterogeneity.

机构信息

Laboratory of Neurogenetics, Parkinson's Disease and Cerebrovascular Disease (LR-12-SP-19), Habib Bourguiba University Hospital, University of Sfax, Sfax, Tunisia.

Clinical Investigation Center (CIC), Habib Bourguiba University Hospital, Sfax, Tunisia.

出版信息

Mol Biol Rep. 2020 Aug;47(8):5755-5761. doi: 10.1007/s11033-020-05643-9. Epub 2020 Jul 14.

DOI:10.1007/s11033-020-05643-9
PMID:32666437
Abstract

Dysferlinopathies belong to the heterogeneous group of autosomal recessive muscular disorders, caused by mutations in the dysferlin gene and characterized by a high degree of clinical variability even though within the same family. This study aims to describe three cases, belonging to a consanguineous Tunisian family, sharing a new splicing mutation in the dysferlin gene and presenting intra-familial variability of dysferlinopathies: Proximal-distal weakness and distal myopathy with anterior tibial onset. We performed the next generation sequencing for mutation screening and reverse transcriptase-PCR for gene expression analysis. Routine muscle histology was used for muscle biopsy processing. The clinical presentation demonstrated heterogeneous phenotypes between the three cases: Two presented intermediate phenotypes of dysferlinopathy with proximal-distal weakness and the third had a distal myopathy with anterior tibial onset. Genetic analysis yielded a homozygous splicing mutation (c.4597-2A>G) in the dysferlin gene, giving rise to the suppression of 28 bp of the exon 43. The splicing mutation found in our family (c.4597-2A>G) is responsible for the suppression of 28 bp of the exon 43 and a wide clinical intra-familial variability.

摘要

肌营养不良蛋白病属于常染色体隐性遗传性肌肉疾病的异质性群体,由肌营养不良蛋白基因突变引起,即使在同一家庭中,也具有高度的临床变异性。本研究旨在描述三个病例,属于一个近亲结婚的突尼斯家庭,携带肌营养不良蛋白基因的新剪接突变,并表现出肌营养不良蛋白病的家族内变异性:近端-远端无力和胫骨前起始的远端肌病。我们进行了下一代测序以进行突变筛查和逆转录聚合酶链式反应以进行基因表达分析。常规肌肉组织学用于肌肉活检处理。临床表现显示三个病例之间存在异质性表型:两个表现出肌营养不良蛋白病的中间表型,具有近端-远端无力,第三个表现出胫骨前起始的远端肌病。基因分析显示肌营养不良蛋白基因中的纯合剪接突变(c.4597-2A>G),导致外显子 43 缺失 28bp。我们在家族中发现的剪接突变(c.4597-2A>G)导致外显子 43 缺失 28bp 和广泛的家族内临床变异性。

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