Valliyammai Natarajan, Nancy Nirmala K, Sagar Tenali G, Rajkumar Thangarajan
Departments of Molecular Oncology.
Medical Oncology, Cancer Institute (WIA), Chennai, Tamil Nadu, India.
J Pediatr Hematol Oncol. 2018 Jan;40(1):e1-e8. doi: 10.1097/MPH.0000000000001006.
NOTCH1/FBXW7 mutations trigger oncogenic NOTCH1 signaling and its downstream target genes play crucial roles in the molecular pathogenesis of T-cell acute lymphoblastic leukemia (T-ALL). In the present study, NOTCH1 and FBXW7 mutations were studied in 25 primary T-ALL samples. All 34 exons of NOTCH1 and hotspot exons (exon 9 and exon 10) of FBXW7 were polymerase chain reaction amplified and sequenced for mutations. Our results showed that 13/25 (52%) were NOTCH1-mutated, of which 11 patients (44%) showed mutation in the hotspot exons. Four patients (16%) had mutations in non-hotspot exons of NOTCH1. Notably, 2 T-ALL patients (8%) harbored mutations in both hotspot and non-hotspot exons of NOTCH1, whereas 2 patients (8%) had mutations in the hotspot exons of FBXW7. In all, 7 mutations were identified which were not previously reported. The real-time polymerase chain reaction study in 15 patients revealed that increased expression of activated NOTCH1 was found in NOTCH1/FBXW7 hotspot exon-mutated cases. In addition, NOTCH1/FBXW7-mutated patients had showed upregulated HES1, c-MYC, NOTCH3 gene expression. When survival analysis was performed including samples (n=50) from our previous study, an early treatment response and better survival was observed in NOTCH1/FBXW7 hotspot-mutated patients. Our study suggests that NOTCH1/FBXW7 hotspot-mutated T-ALL cases had better response to ALL BFM-95 protocol.
NOTCH1/FBXW7突变引发致癌性NOTCH1信号传导,其下游靶基因在T细胞急性淋巴细胞白血病(T-ALL)的分子发病机制中起关键作用。在本研究中,对25例原发性T-ALL样本进行了NOTCH1和FBXW7突变研究。对NOTCH1的所有34个外显子和FBXW7的热点外显子(外显子9和外显子10)进行聚合酶链反应扩增并测序以检测突变。我们的结果显示,13/25(52%)为NOTCH1突变,其中11例患者(44%)在热点外显子中出现突变。4例患者(16%)在NOTCH1的非热点外显子中有突变。值得注意的是,2例T-ALL患者(8%)在NOTCH1的热点和非热点外显子中均有突变,而2例患者(8%)在FBXW7的热点外显子中有突变。总共鉴定出7种先前未报道的突变。对15例患者的实时聚合酶链反应研究表明,在NOTCH1/FBXW7热点外显子突变的病例中发现活化NOTCH1的表达增加。此外,NOTCH1/FBXW7突变的患者显示HES1、c-MYC、NOTCH3基因表达上调。当对包括我们先前研究中的样本(n = 50)进行生存分析时,观察到NOTCH1/FBXW7热点突变的患者有早期治疗反应和更好的生存率。我们的研究表明,NOTCH1/FBXW7热点突变的T-ALL病例对ALL BFM-95方案反应更好。