Zhu Xiaomei, Deng Xiaopeng, Huang Guangying, Wang Jing, Yang Jingwen, Chen Si, Ma Xu, Wang Binbin
Graduate School of Peking Union Medical College, Beijing, China ; National Research Institute for Family Planning, Beijing, China.
Department of Obstetrics and Gynecology, Shengjing Hospital of China Medical University, Shenyang, China.
PLoS One. 2014 Feb 18;9(2):e87437. doi: 10.1371/journal.pone.0087437. eCollection 2014.
As a major product of extracellular matrix (ECM), Hyaluronic acid (HA) is involved in early cardiac development and mainly synthesized by Hyaluronan synthase 2 (HAS2) during embryogenesis. Targeted deletion of HAS2 gene in mice led to obvious cardiac and vascular defects. To clarify the potential association of the mutation in HAS2 with the development of congenital heart disease (CHD), in this study, we sequenced the coding region of HAS2 and identified a novel non-synonymous variant c.A1496T (p.Glu499Val) in one of 100 non-syndromic Ventricular Septal Defect (VSD) patients. The variant was not observed in 250 controls. In addition, to determine the contribution of HAS2 variant in VSD, we compared HA content in supernatant using HA quantitative analysis and found that the mutation obviously affected the HA synthetic activity of HAS2. To our knowledge, this is the first time that the mutation in HAS2 was found in Chinese VSD patients, which suggested that HAS2 may be involved in the etiology of non-syndromic VSD and have the vital function in the development of heart septum.
作为细胞外基质(ECM)的主要产物,透明质酸(HA)参与心脏早期发育,在胚胎发生过程中主要由透明质酸合酶2(HAS2)合成。在小鼠中靶向缺失HAS2基因会导致明显的心脏和血管缺陷。为了阐明HAS2突变与先天性心脏病(CHD)发生发展之间的潜在关联,在本研究中,我们对100例非综合征性室间隔缺损(VSD)患者中的HAS2编码区进行了测序,并在其中1例患者中鉴定出一种新的非同义变体c.A1496T(p.Glu499Val)。在250名对照中未观察到该变体。此外,为了确定HAS2变体在VSD中的作用,我们使用HA定量分析比较了上清液中的HA含量,发现该突变明显影响了HAS2的HA合成活性。据我们所知,这是首次在中国VSD患者中发现HAS2突变,这表明HAS2可能参与非综合征性VSD的病因学,并且在心脏隔膜发育中具有重要作用。