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一名常染色体隐性遗传性骨硬化症患者中TCIRG1和CLCN7基因突变的鉴定。

Identification of TCIRG1 and CLCN7 gene mutations in a patient with autosomal recessive osteopetrosis.

作者信息

Yu Tingting, Yu Yongguo, Wang Jian, Yin Lei, Zhou Yunfang, Ying Daming, Huang Rongkui, Chen Huijin, Wu Shenmei, Shen Yongnian, Fu Qihua, Chen Fuxiang

机构信息

Department of Laboratory Medicine, Shanghai Ninth People's Hospital, Shanghai Jiaotong University School of Medicine, Shanghai 200011, P.R. China.

Institute of Pediatric Translational Medicine, Shanghai Children's Medical Center, Shanghai Jiaotong University School of Medicine, Shanghai 200127, P.R. China.

出版信息

Mol Med Rep. 2014 Apr;9(4):1191-6. doi: 10.3892/mmr.2014.1955. Epub 2014 Feb 17.

DOI:10.3892/mmr.2014.1955
PMID:24535484
Abstract

Osteopetrosis is a heritable bone disorder that exhibits highly clinical and genetical heterogeneity, and is caused by defective osteoclastic resorption. The three main forms are the autosomal recessive severe (ARO), the intermediate autosomal and the autosomal dominant benign osteopetrosis forms. In the present study, the clinical, biochemical and radiological manifestations were described in a patient with osteopetrosis. Sequence analysis identified the compound heterozygous mutations, c.909C>A (p.Tyr303X) and c.2008C>T (p.Arg670X), in TCIRG1, and a heterozygous splicing mutation, c.1798‑1G>T, in the chloride channel 7 gene (CLCN7). Two aberrant forms of the CLCN7 transcripts, c.1798_1883 (exon 20) deletion predicted to cause p.Leu601GlyfsX13, and the c.1798_1821 deletion, the first 24 bp of the exon 20, predicted to cause p.Gly600_Gln607del, were detected by further analysis of the splicing patterns in the leukocytes. The patient's asymptomatic mother carried the TCIRG1 c.909C>A (p.Tyr303X) and CLCN7 c.1798‑1G>T mutations, while the asymptomatic father carried the TCIRG1 c.2008C>T (p.Arg670X) mutation only. The patient was finally diagnosed with ARO on the basis of clinical and biochemical parameters, radiological changes and genetic defects. To the best of our knowledge, this is the first reported case of a patient with osteopetrosis who carries TCIRG1 and CLCN7 mutations. In addition, among the three mutations, TCIRG1 c.909C>A and CLCN7 c.1798‑1G>T were novel mutations.

摘要

骨硬化症是一种遗传性骨疾病,具有高度的临床和遗传异质性,由破骨细胞吸收缺陷引起。主要有三种形式,即常染色体隐性重症(ARO)、中间型常染色体和常染色体显性良性骨硬化症形式。在本研究中,描述了一名骨硬化症患者的临床、生化和放射学表现。序列分析确定了在TCIRG1基因中存在复合杂合突变c.909C>A(p.Tyr303X)和c.2008C>T(p.Arg670X),以及在氯离子通道7基因(CLCN7)中存在杂合剪接突变c.1798-1G>T。通过对白细胞剪接模式的进一步分析,检测到CLCN7转录本的两种异常形式,c.1798_1883(外显子20)缺失,预计导致p.Leu601GlyfsX13,以及c.1798_1821缺失,即外显子20的前24 bp,预计导致p.Gly600_Gln607del。患者无症状的母亲携带TCIRG1基因的c.909C>A(p.Tyr303X)和CLCN7基因的c.1798-1G>T突变,而无症状的父亲仅携带TCIRG1基因的c.2008C>T(p.Arg670X)突变。根据临床和生化参数、放射学变化及基因缺陷,该患者最终被诊断为ARO。据我们所知,这是首例报道的携带TCIRG1和CLCN7突变的骨硬化症患者。此外,在这三个突变中,TCIRG1基因的c.909C>A和CLCN7基因的c.1798-1G>T是新的突变。

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