Literature DB >> 25542771

Case of mild Schmid-type metaphyseal chondrodysplasia with novel sequence variation involving an unusual mutational site of the COL10A1 gene.

Hyunwoong Park1, Susie Hong1, Sung Im Cho1, Tae-Joon Cho2, In Ho Choi2, Dong-Kyu Jin3, Young Bae Sohn4, Sung Won Park3, Hyun-Hae Cho5, Jung-Eun Cheon5, So Yeon Kim6, Ji Yeon Kim7, Sung Sup Park8, Moon-Woo Seong9.   

Abstract

Schmid-type metaphyseal chondrodysplasia (MCDS) is characterized by short stature with short legs, bowing of the long bones, coxa vara, and waddling gait. MCDS is a relatively common form of MCD. Most mutations that cause MCDS occur within the carboxyl-terminal non-collagenous domain (NC1) of the COL10A1 gene. We performed mutational analysis of the COL10A1 genes in 4 unrelated Korean patients with diagnosed MCDS. Mutational analysis of COL10A1 identified c.1904_1915delinsT (p.Gln635LeufsX10) and c.1969dupG (p.Ala657GlyfsX10), 2 novel frameshift mutations, and c.2030T>A (p.Val677Glu) and c.862G>C (p.Gly288Arg) at unusual mutational sites, which could be pathogenic. We present the first report of the molecular characteristics of MCDS in 4 Korean patients. Our findings suggest that a novel sequence variation involving an unusual mutational site of the COL10A1 gene can cause mild MCDS.
Copyright © 2014 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  COL10A1; Collagen type X; Schmid metaphyseal chondrodysplasia

Mesh:

Substances:

Year:  2014        PMID: 25542771     DOI: 10.1016/j.ejmg.2014.12.011

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  6 in total

1.  A Venezuelan Case of Schmid-Type Metaphyseal Chondrodysplasia with a Novel Mutation in COL10A1.

Authors:  Francisco Cammarata-Scalisi; Uta Matysiak; Tanja Velten; Michele Callea; Dianora Araque; Colin E Willoughby; Angela Galeotti; Andrea Avendaño
Journal:  Mol Syndromol       Date:  2019-02-09

Review 2.  Characterization of a novel COL10A1 variant associated with Schmid-type metaphyseal chondrodysplasia and a literature review.

Authors:  Huixiao Wu; Shuping Wang; Guimei Li; Yangyang Yao; Ning Wang; Xiaoqing Sun; Li Fang; Xiuyun Jiang; Jiajun Zhao; Yanzhou Wang; Chao Xu
Journal:  Mol Genet Genomic Med       Date:  2021-03-25       Impact factor: 2.183

3.  Identification of key genes associated with Schmid-type metaphyseal chondrodysplasia based on microarray data.

Authors:  Bing Wang; Li He; Wusheng Miao; Ge Wu; Hai Jiang; Yongtao Wu; Jining Qu; Min Li
Journal:  Int J Mol Med       Date:  2017-04-19       Impact factor: 4.101

4.  A Novel Presentation of Metaphyseal Chondrodysplasia, Schmid Type with Factor VII Deficiency.

Authors:  Mushtaq Ahmed; Saad Nasir; Syeda Shaheera Riaz Hashmi; Zia Iqbal; Ayesha Saleem
Journal:  Cureus       Date:  2020-03-23

5.  Identification of two novel COL10A1 heterozygous mutations in two Chinese pedigrees with Schmid-type metaphyseal chondrodysplasia.

Authors:  Lingchi Kong; Li Shi; Wenbo Wang; Rongtai Zuo; Mengwei Wang; Qinglin Kang
Journal:  BMC Med Genet       Date:  2019-12-19       Impact factor: 2.103

6.  A novel missense COL10A1 mutation: c.2020G>A; p. Gly674Arg linked with the bowed legs stature in the Schmid metaphyseal chondrodysplasia-affected Chinese lineage.

Authors:  Qiong Chen; Sheng-Nan Wu; Yong-Xing Chen; Selvaa Kumar C; Lu Zhang; Hai-Yan Wei; Senthil Arun Kumar
Journal:  Bone Rep       Date:  2019-12-13
  6 in total

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