Literature DB >> 24535484

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

Tingting Yu1, Yongguo Yu2, Jian Wang3, Lei Yin4, Yunfang Zhou5, Daming Ying5, Rongkui Huang5, Huijin Chen5, Shenmei Wu5, Yongnian Shen4, Qihua Fu3, Fuxiang Chen1.   

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.

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Year:  2014        PMID: 24535484     DOI: 10.3892/mmr.2014.1955

Source DB:  PubMed          Journal:  Mol Med Rep        ISSN: 1791-2997            Impact factor:   2.952


  5 in total

1.  Novel mutations of TCIRG1 cause a malignant and mild phenotype of autosomal recessive osteopetrosis (ARO) in four Chinese families.

Authors:  Xiao-Ya Zhang; Jin-Wei He; Wen-Zhen Fu; Chun Wang; Zhen-Lin Zhang
Journal:  Acta Pharmacol Sin       Date:  2017-08-17       Impact factor: 6.150

2.  99Tcm-MDP Imaging of Osteopetrosis: Case Report.

Authors:  Li-Chun Zheng; Xiang-Liu OuYang; Gui-Chao Liu; Wen-Jun Zhang; Xiao-Ming Zhang
Journal:  Medicine (Baltimore)       Date:  2015-06       Impact factor: 1.889

3.  Identification of two novel mutations on CLCN7 gene in a patient with malignant ostopetrosis.

Authors:  Giuseppe Bonapace; Maria Teresa Moricca; Valentina Talarico; Francesca Graziano; Licia Pensabene; Roberto Miniero
Journal:  Ital J Pediatr       Date:  2014-11-20       Impact factor: 2.638

4.  Novel CLCN7 mutation identified in a Han Chinese family with autosomal dominant osteopetrosis-2.

Authors:  Hao Deng; Dan He; Pengfei Rong; Hongbo Xu; Lamei Yuan; Liu Li; Qian Lu; Yi Guo
Journal:  Mol Pain       Date:  2016-06-20       Impact factor: 3.395

5.  Clinical and molecular characterization of five Chinese patients with autosomal recessive osteopetrosis.

Authors:  Huanhuan Liang; Niu Li; Ru-En Yao; Tingting Yu; Lixia Ding; Jing Chen; Jian Wang
Journal:  Mol Genet Genomic Med       Date:  2021-09-21       Impact factor: 2.183

  5 in total

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