Cizmarova M, Kostalova L, Pribilincova Z, Lasabova Z, Hlavata A, Kovacs L, Ilencikova D
Endocr Regul. 2013 Oct;47(4):217-22. doi: 10.4149/endo_2013_04_217.
The term ´Rasopathies´ represents a group of five neurodevelopmental syndromes (Noonan, LEOPARD, Costello, Cardio-facio-cutaneous, and Neurofibromatose-Noonan syndrome) caused by germline mutation in genes encoding proteins involved in RAS/MAPK (rat sarcoma/mitogen-activated protein kinase) signaling pathway. The RAS/MAPK signaling pathway participates in regulation of cell determination, proliferation, differentiation, migration, and senescence and dysregulation of this pathway can lead to the risk of tumorigenesis. In this review, we aim to summarize the current clinical and molecular genetic knowledge on Rasopathies with special attention for the risk of cancer. We propose also clinical and therapeutic approach for patients with malignancy.
We are reviewing the clinical and molecular basis of Rasopathies based on recent studies, clinical examination, and molecular diagnostics (mutation analysis of causal genes for Rasopathies) in Slovak pediatric patients.
Some clinical features, such as short stature, a specific facial dysmorphology and cardiac abnormalities are common to all of Rasopathy syndromes. However, there are unique signs by which the syndromes can differ from each other, especially multiple lentigo in LEOPARD syndrome, increased risk of malignancy in Costello syndrome, dry hyperkeratotic skin in patients with cardio-facio-cutaneous syndrome, and neurofibromas and cafe-au-lait spots in neurofibromatosis-Noonan syndrome.
Despite the overlapping clinical features, Rasopathy syndromes exhibit unique fenotypical features and the precise molecular diagnostics may lead to confirmation of each syndrome. The molecular diagnostics may allow the detection of pathogenic mutation associated with tumorigenesis.
“Rasopathies”一词代表一组由参与RAS/丝裂原活化蛋白激酶(MAPK)信号通路的蛋白质编码基因种系突变引起的五种神经发育综合征(努南综合征、豹皮综合征、科斯特洛综合征、心面皮肤综合征和神经纤维瘤病-努南综合征)。RAS/MAPK信号通路参与细胞决定、增殖、分化、迁移和衰老的调节,该通路失调会导致肿瘤发生风险增加。在本综述中,我们旨在总结目前关于Rasopathies的临床和分子遗传学知识,并特别关注癌症风险。我们还提出了针对恶性肿瘤患者的临床和治疗方法。
我们基于斯洛伐克儿科患者的近期研究、临床检查和分子诊断(Rasopathies致病基因的突变分析),对Rasopathies的临床和分子基础进行综述。
一些临床特征,如身材矮小、特定的面部畸形和心脏异常,在所有Rasopathy综合征中都很常见。然而,各综合征之间也存在独特的体征差异,尤其是豹皮综合征中的多发性雀斑样痣、科斯特洛综合征中恶性肿瘤风险增加、心面皮肤综合征患者的干性角化过度皮肤,以及神经纤维瘤病-努南综合征中的神经纤维瘤和咖啡斑。
尽管临床特征存在重叠,但Rasopathy综合征表现出独特的表型特征,精确的分子诊断可能有助于确诊每种综合征。分子诊断可能有助于检测与肿瘤发生相关的致病突变。