| Literature DB >> 25542771 |
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.Entities:
Keywords: COL10A1; Collagen type X; Schmid metaphyseal chondrodysplasia
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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