Zhang Lixin, Xu Xiaowen, Chen Yufan, Li Lianyong, Zhang Lijun, Li Qiwei
Department of Pediatric Orthopaedics, Shengjing Hospital of China Medical University, Shenyang, Liaoning 110004, P.R. China.
Exp Ther Med. 2020 Apr;19(4):2799-2803. doi: 10.3892/etm.2020.8513. Epub 2020 Feb 11.
Developmental dysplasia of the hip (DDH), previously known as congenital hip dislocation, is a frequently disabling condition characterized by premature arthritis later in life. Genetic factors play a key role in the aetiology of DDH. In the present study, a genome-wide linkage scan with the Affymetrix 10K GeneChip was performed on a four-generation Chinese family, which included 19 healthy members and 5 patients. Parametric and non-parametric multipoint linkage analyses were carried out with Genespring GT v.2.0 software, and the logarithm of odds (LOD) score and nonparametric linkage (NPL) score were calculated. Parametric linkage analysis was performed, assuming an autosomal recessive trait with full penetrance and Affymetrix 'Asian' allele frequencies. The strongest evidence for linkage was found on chromosome 8q23-24, with a peak LOD score of 2.658 (θ=0), covering 2.377 Mb from single nucleotide polymorphisms (SNPs) rs724717 to rs720132. This interval included nine additional successive SNPs: rs1566071, rs1902121, rs756404, rs702768, rs777813, rs2033995, rs147959, rs2884367 and rs1898287. The same region also yielded the highest NPL score of 2.883 (P=0.0156) from the non-parametric multipoint linkage analysis. Additionally, the second highest NPL score of 2.727 (P=0.0156) and LOD score of 2.528 (θ=0) were obtained on chromosome 12p12 for three consecutive markers (rs1919980, rs763853 and rs725124). This region overlapped a narrow distance of 0.642 Mb. Notably, in addition to these two regions; no significant linkage was identified for other chromosomal regions (with LOD and NPL scores >2.0). For the first time, at least for this pedigree, the evidence in the present study showed that DDH is mapped to two novel regions at 8q23-q24 and 12p12.
发育性髋关节发育不良(DDH),以前称为先天性髋关节脱位,是一种常见的致残性疾病,其特征是在生命后期出现过早的关节炎。遗传因素在DDH的病因中起关键作用。在本研究中,使用Affymetrix 10K基因芯片对一个四代中国家系进行了全基因组连锁扫描,该家系包括19名健康成员和5名患者。使用Genespring GT v.2.0软件进行参数化和非参数化多点连锁分析,并计算优势对数(LOD)得分和非参数连锁(NPL)得分。进行参数化连锁分析时,假设为具有完全外显率的常染色体隐性性状和Affymetrix“亚洲”等位基因频率。在8号染色体q23-24上发现了最强的连锁证据,峰值LOD得分为2.658(θ=0),覆盖从单核苷酸多态性(SNP)rs724717到rs720132的2.377 Mb。该区间还包括另外九个连续的SNP:rs1566071、rs1902121、rs756404、rs702768、rs777813、rs2033995、rs147959、rs2884367和rs1898287。在非参数化多点连锁分析中,同一区域的NPL得分也最高,为2.883(P=0.0156)。此外,在12号染色体p12上,三个连续标记(rs1919980、rs763853和rs725124)的第二高NPL得分为2.727(P=0.0156),LOD得分为2.528(θ=0)。该区域重叠了0.642 Mb的狭窄距离。值得注意的是,除了这两个区域外,未在其他染色体区域发现显著连锁(LOD和NPL得分>2.0)。本研究首次至少针对该家系表明,DDH定位于8q23-q24和12p12的两个新区域。