Division of Cancer Genetics, Department of Basic Oncology, Oncology Institute, Istanbul University, Istanbul, Turkey.
Division of Pediatric Hematology-Oncology, Oncology Institute, Istanbul University, Istanbul, Turkey.
Mol Genet Genomic Med. 2019 Aug;7(8):e785. doi: 10.1002/mgg3.785. Epub 2019 Jun 17.
Retinoblastoma (Rb) is the most prevalent intraocular pediatric malignancy of the retina. Significant genetic factors are known to have a role in the development of Rb.
Here, we report the mutation status of 4813 clinically significant genes in six patients with noncarrier of RB1 gene mutation and having normal RB1 promoter methylation from three families having higher risk for developing Rb in the study.
A total of 27 variants were detected in the study. Heterozygous missense variants c.1162G > A (p.Gly388Arg) in the FGFR4 gene; c.559C > T (p.Pro187Ser) in the NQO1 gene were identified. The family based evaluation of the variants showed that the variant, c.714T > G (p.Tyr238Ter), in the CLEC7A gene in first family; the variant, c.55C > T (p.Arg19Ter), in the APOC3 gene and the variant, c.1171C > T (p.Gln391Ter), in the MUTYH gene in second family; and the variant, c.211G > A (p.Gly71Arg), in the UGT1A1 gene in the third family, were found statistically significant (p < 0.05).
This study might be an important report on emphazing the mutational status of other genes in patients without RB1 gene mutations and having high risk for developing Rb. The study also indicates the interaction between the retinoic acid pathway and Rb oncogenesis for the first time.
视网膜母细胞瘤(Rb)是最常见的视网膜儿童眼内恶性肿瘤。已知有一些重要的遗传因素在 Rb 的发展中起作用。
在这里,我们报告了来自三个具有较高 Rb 发生风险的家族的六名非 RB1 基因突变且 RB1 启动子甲基化正常的患者中 4813 个临床重要基因的突变状态。
研究中共检测到 27 种变异。在 FGFR4 基因中发现了杂合错义变异 c.1162G > A(p.Gly388Arg);在 NQO1 基因中发现了 c.559C > T(p.Pro187Ser)。基于家族的变异评估表明,在第一个家族中,CLEC7A 基因中的变异 c.714T > G(p.Tyr238Ter);在 APOC3 基因中的变异 c.55C > T(p.Arg19Ter)和在 MUTYH 基因中的变异 c.1171C > T(p.Gln391Ter),在第二个家族中;在第三个家族中的 UGT1A1 基因中的变异 c.211G > A(p.Gly71Arg),具有统计学意义(p < 0.05)。
本研究可能是强调无 RB1 基因突变且具有较高 Rb 发生风险的患者其他基因突变状态的重要报告。该研究还首次表明了视黄酸途径与 Rb 致癌作用之间的相互作用。