Department of Pediatrics, Madigan Army Medical Center, Tacoma, Washington.
Am J Med Genet A. 2020 Jan;182(1):195-200. doi: 10.1002/ajmg.a.61395. Epub 2019 Nov 7.
RASopathies are a group of phenotypically overlapping disorders that arise from dysregulation of the RAS/MAPK pathway. These disorders include Noonan syndrome, Costello syndrome, cardiofaciocutaneous syndrome, and neurofibromatosis-Type 1. While somatic mutations in the three human Ras genes (KRAS, HRAS, and NRAS) are a common finding in a variety of cancers, germline mutations in each of the these genes cause developmental RASopathy phenotypes with mutations in specific genes typically correlating with specific phenotypes. We present the case of a germline heterozygous NRAS mutation producing a severe phenotype involving embryonal rhabdomyosarcoma, severe intellectual disability, and numerous melanocytic nevi in addition to more typical manifestations of Noonan syndrome. Additionally, the specific p.G12R NRAS mutation in this case is a common somatic mutation in cancer cells, and analysis of previously reported NRAS-RASopathy cases suggests that mutations at traditionally oncogenic codons are associated with elevated cancer risk not present with mutations at other sites.
RAS 病是一组表型重叠的疾病,源于 RAS/MAPK 通路的失调。这些疾病包括努南综合征、科斯塔尔综合征、心面综合征和神经纤维瘤病 1 型。虽然人类 Ras 基因(KRAS、HRAS 和 NRAS)的体细胞突变是各种癌症中的常见发现,但这些基因的种系突变会导致发育性 RAS 病表型,特定基因的突变通常与特定表型相关。我们介绍了一例种系杂合性 NRAS 突变导致的严重表型,涉及胚胎性横纹肌肉瘤、严重智力障碍和大量黑素细胞痣,此外还有更典型的努南综合征表现。此外,该病例中的特定 p.G12R NRAS 突变是癌细胞中的常见体细胞突变,对先前报道的 NRAS-RAS 病病例的分析表明,传统致癌密码子的突变与存在于其他部位的突变相比,与癌症风险升高有关。