Basit Sulman, Naqvi Syed Kamran-ul-Hassan, Wasif Naveed, Ali Ghazanfar, Ansar Muhammad, Ahmad Wasim
Department of Biochemistry, Faculty of Biological Sciences, Quaid-i-Azam University Islamabad, Pakistan.
BMC Med Genet. 2008 Nov 27;9:102. doi: 10.1186/1471-2350-9-102.
Grebe-type chondrodysplasia (GCD) is a rare autosomal recessive syndrome characterized by severe acromesomelic limb shortness with non-functional knob like fingers resembling toes. Mutations in the cartilage-derived morphogenetic protein 1 (CDMP1) gene cause Grebe-type chondrodysplasia.
Genotyping of six members of a Pakistani family with Grebe-type chondrodysplasia, including two affected and four unaffected individuals, was carried out by using polymorphic microsatellite markers, which are closely linked to CDMP1 locus on chromosome 20q11.22. To screen for a mutation in CDMP1 gene, all of its coding exons and splice junction sites were PCR amplified from genomic DNA of affected and unaffected individuals of the family and sequenced directly in an ABI Prism 310 automated DNA sequencer.
Genotyping results showed linkage of the family to CDMP1 locus. Sequence analysis of the CDMP1 gene identified a novel four bases insertion mutation (1114insGAGT) in exon 2 of the gene causing frameshift and premature termination of the polypeptide.
We describe a 4 bp novel insertion mutation in CDMP1 gene in a Pakistani family with Grebe-type chondrodysplasia. Our findings extend the body of evidence that supports the importance of CDMP1 in the development of limbs.
格莱贝型软骨发育不全(GCD)是一种罕见的常染色体隐性综合征,其特征为严重的肢端中间型肢体短小,手指呈无功能的瘤状,类似脚趾。软骨衍生形态发生蛋白1(CDMP1)基因突变导致格莱贝型软骨发育不全。
利用与20q11.22染色体上CDMP1基因座紧密连锁的多态性微卫星标记,对一个患有格莱贝型软骨发育不全的巴基斯坦家庭的6名成员进行基因分型,其中包括2名患者和4名未受影响的个体。为了筛查CDMP1基因中的突变,从该家庭受影响和未受影响个体的基因组DNA中PCR扩增其所有编码外显子和剪接连接位点,并在ABI Prism 310自动DNA测序仪中直接测序。
基因分型结果显示该家庭与CDMP1基因座连锁。CDMP1基因的序列分析在该基因的外显子2中鉴定出一个新的四个碱基插入突变(1114insGAGT),导致多肽移码和提前终止。
我们描述了一个患有格莱贝型软骨发育不全的巴基斯坦家庭中CDMP1基因的一个4 bp新插入突变。我们的发现扩展了支持CDMP1在肢体发育中重要性的证据。