Friedman Eitan, Moran Daniel S, Ben-Avraham Danny, Yanovich Ran, Atzmon Gil
Susanne Levy Gertner Oncogenetics Unit,The Danek Gertner Institute of Human Genetics,Chaim Sheba Medical Center, 52621 Tel-Hashomer,Israel.
Ariel University,Ariel, Samaria,Israel.
Genet Res (Camb). 2014;96:e004. doi: 10.1017/S001667231400007X.
While genetic factors in all likelihood contribute to stress fracture (SF) pathogenesis, a few studies focusing on candidate genes have previously been reported. The objective of this study is to gain better understanding on the genetic basis of SF in a gene-naive manner. Exome sequence capture followed by massive parallel sequencing of two pooled DNA samples from Israeli combat soldiers was employed: cases with high grade SF and ethnically matched healthy controls. The resulting sequence variants were individually verified using the Sequenom™ platform and the contribution of the genetic alterations was validated in a second cohort of cases and controls. In the discovery set that included DNA pool of cases (n = 34) and controls (n = 60), a total of 1174 variants with >600 reads/variant/DNA pool were identified, and 146 (in 127 genes) of these exhibited statistically significant (P < 0·05) different rates between SF cases and controls after multiple comparisons correction. Subsequent validation of these 146 sequence variants individually in a total of 136 SF cases and 127 controls using the Sequenom™ platform validated 20/146 variants. Of these, three missense mutations (rs7426114, rs4073918, rs3752135 in the NEB, SLC6A18 and SIGLEC12 genes, respectively) and three synonymous mutations (rs2071856, rs2515941, rs716745 in the ELFN2, GRK4, LRRC55 genes) displayed significant different rates in SF cases compared with controls. Exome sequencing seemingly unravelled novel candidate genes as involved in SF pathogenesis and predisposition.
虽然遗传因素很可能在应力性骨折(SF)的发病机制中起作用,但此前仅有少数针对候选基因的研究报道。本研究的目的是以一种不依赖已知基因的方式,更好地了解SF的遗传基础。我们采用外显子序列捕获技术,随后对来自以色列战斗士兵的两个混合DNA样本进行大规模平行测序:重度SF患者和种族匹配的健康对照。使用Sequenom™平台对所得序列变异进行个体验证,并在第二组病例和对照中验证基因改变的作用。在发现集中,包括病例(n = 34)和对照(n = 60)的DNA池,共鉴定出1174个变异(每个变异/DNA池>600条读数),其中146个(在127个基因中)在多重比较校正后,SF病例和对照之间显示出统计学显著(P < 0·05)的不同频率。随后使用Sequenom™平台在总共136例SF病例和127例对照中对这146个序列变异进行个体验证,验证了20/146个变异。其中,三个错义突变(分别在NEB、SLC6A18和SIGLEC12基因中的rs7426114、rs4073918、rs3752135)和三个同义突变(在ELFN2、GRK4、LRRC55基因中的rs2071856、rs2515941、rs716745)与对照相比,在SF病例中显示出显著不同的频率。外显子测序似乎揭示了与SF发病机制和易感性相关的新候选基因。