Zheng Jie, Xu Xiaowei, Zhang Xinjie, Wang Xuetao, Shu Jianbo, Cai Chunquan
Graduate College, Tianjin Medical University, Tianjin 300070, P.R. China.
Tianjin Pediatric Research Institute, Tianjin Children's Hospital (Children's Hospital of Tianjin University), Tianjin 300134, P.R. China.
Exp Ther Med. 2021 Feb;21(2):104. doi: 10.3892/etm.2020.9536. Epub 2020 Nov 27.
Limb-girdle muscular dystrophies (LGMDs) are a group of neuromuscular diseases that are characterized by progressive muscle weakness. LGMD type 2A (LGMD2A), caused by variants in the calpain-3 (CAPN3) gene, is the most prevalent type. The present study aimed to analyze pathogenic CAPN3 gene variants in two pedigrees affected by LGMD2A. Each family contains three patients who are siblings and sought genetic counseling. Genomic DNA was extracted from the peripheral blood samples collected from the probands and family members and whole-exome sequencing (WES) was used to detect the pathogenic genes in the probands. Suspected variants were subsequently validated by Sanger sequencing. In family 1, WES revealed that the proband carried the compound heterogeneous variants c.1194-9A>G and c.1437C>T (p.Ser479=) in CAPN3 (NM_000070.2). In family 2, WES identified that the proband carried the compound heterogeneous variants c.632+4A>G and c.1468C>T (p.Arg490Trp) in CAPN3 (NM_000070.2). In conclusion, the present study indicated that the compound heterogeneous variants of the CAPN3 gene were most likely responsible for LGMD2A in the two Chinese families.
肢带型肌营养不良症(LGMDs)是一组以进行性肌肉无力为特征的神经肌肉疾病。由钙蛋白酶3(CAPN3)基因变异引起的2A型肢带型肌营养不良症(LGMD2A)是最常见的类型。本研究旨在分析两个受LGMD2A影响的家系中的致病性CAPN3基因变异。每个家系包含三名患者,他们是兄弟姐妹,并寻求遗传咨询。从先证者和家庭成员采集的外周血样本中提取基因组DNA,采用全外显子组测序(WES)检测先证者中的致病基因。随后通过桑格测序验证可疑变异。在家族1中,WES显示先证者在CAPN3(NM_000070.2)中携带复合杂合变异c.1194-9A>G和c.1437C>T(p.Ser479=)。在家族2中,WES鉴定出先证者在CAPN3(NM_000070.2)中携带复合杂合变异c.632+4A>G和c.1468C>T(p.Arg490Trp)。总之,本研究表明,CAPN3基因的复合杂合变异很可能是这两个中国家系中LGMD2A的病因。