School of Biological Science and Technology, Central South University, Changsha, Hunan, China.
Gene. 2012 Jul 10;502(2):168-71. doi: 10.1016/j.gene.2012.04.023. Epub 2012 Apr 29.
Osteogenesis imperfect (OI) is a heritable connective tissue disorder with bone fragility as a cardinal manifestation, accompanied by short stature, dentinogenesis imperfecta, hyperlaxity of ligaments and skin, blue sclerae and hearing loss. Dominant form of OI is caused by mutations in the type I procollagen genes, COL1A1/A2. Here we identified a novel splicing mutation c.3207+1G>A (GenBank ID: JQ236861) in the COL1A1 gene that caused type I OI in a Chinese family. RNA splicing analysis proved that this mutation created a new splicing site at c.3200, and then led to frameshift. This result further enriched the mutation spectrum of type I procollagen genes.
成骨不全症(OI)是一种遗传性结缔组织疾病,以骨骼脆弱为主要表现,伴有身材矮小、牙本质发育不全、韧带和皮肤过度松弛、巩膜蓝色和听力损失。OI 的显性形式是由 I 型前胶原基因 COL1A1/A2 的突变引起的。在这里,我们在一个中国家庭的 COL1A1 基因中发现了一个新的剪接突变 c.3207+1G>A(GenBank ID:JQ236861),导致 I 型 OI。RNA 剪接分析证明该突变在 c.3200 处创建了一个新的剪接位点,然后导致移码。这一结果进一步丰富了 I 型前胶原基因的突变谱。