Sheng Guangying, Zhang Jian, Zeng Zhao, Pan Jinlan, Wang Qinrong, Wen Lijun, Xu Yang, Wu Depei, Chen Suning
Key Laboratory of Thrombosis and Hemostasis of Ministry of Health, Jiangsu Institute of Hematology, The First Affiliated Hospital of Soochow University, 188 Shizi Street, Suzhou 215006, China.
Key Laboratory of Thrombosis and Hemostasis of Ministry of Health, Jiangsu Institute of Hematology, The First Affiliated Hospital of Soochow University, 188 Shizi Street, Suzhou 215006, China; Collaborative Innovation Center of Hematology, Soochow University, Suzhou, China.
Cancer Genet. 2017 Oct;216-217:16-19. doi: 10.1016/j.cancergen.2017.05.002. Epub 2017 May 24.
Membrane-proximal and truncated mutations of colony-stimulating factor 3 receptor (CSF3R) are frequently found in chronic neutrophilic leukemia (CNL) and atypical chronic myeloid leukemia (aCML). However, rearrangement involving CSF3R in hematological neoplasms has not been reported. Here, we report a case of a 21-year-old female diagnosed as aCML with t(1;9)(p34;q34) who presented a CSF3R rearrangement. First, RNA sequencing identified a novel fusion transcript involving exon 17 of CSF3R and exon 50 of non-erythrocytic-1-spectrin-alpha (SPTAN1). Subsequent reverse transcription-polymerase chain reaction (RT-PCR) and bidirectional Sanger sequencing confirmed the in-frame fusion. The breakpoint was located at the C-terminus of CSF3R, suggesting a pattern of truncation mutation of CSF3R. Unexpectedly, the patient failed to achieve a complete hematological response following the SRC kinase inhibitor dasatinib therapy, which has been reported to effectively inhibit truncated forms of CSF3R. The patient accepted allogeneic hematopoietic stem cell transplantation (HSCT) and currently remains in a good state. In conclusion, this report is the first to identify a fusion involving CSF3R and SPTAN1 in aCML with t(1;9)(p34;q34).
集落刺激因子3受体(CSF3R)的膜近端和截短突变在慢性嗜中性粒细胞白血病(CNL)和非典型慢性髓性白血病(aCML)中经常被发现。然而,血液肿瘤中涉及CSF3R的重排尚未见报道。在此,我们报告一例21岁女性,诊断为aCML伴t(1;9)(p34;q34),其存在CSF3R重排。首先,RNA测序鉴定出一种新的融合转录本,涉及CSF3R的第17外显子和非红细胞1 - 血影蛋白α(SPTAN1)的第50外显子。随后的逆转录 - 聚合酶链反应(RT-PCR)和双向桑格测序证实了读码框内融合。断点位于CSF3R的C末端,提示CSF3R的截短突变模式。出乎意料的是,该患者在接受SRC激酶抑制剂达沙替尼治疗后未能实现完全血液学缓解,据报道该药物可有效抑制CSF3R的截短形式。患者接受了异基因造血干细胞移植(HSCT),目前状态良好。总之,本报告首次在伴t(1;9)(p34;q34)的aCML中鉴定出涉及CSF3R和SPTAN1的融合。