Department of Hematopathology, The University of Texas MD Anderson Cancer Center, Houston, TX, USA.
Department of Pathology and Laboratory Medicine, Weill Cornell Medical College, New York, NY, USA.
Mod Pathol. 2021 Sep;34(9):1673-1685. doi: 10.1038/s41379-021-00817-7. Epub 2021 May 14.
Myeloid/lymphoid neoplasms (M/LN) with 13q12/FLT3 rearrangement have been suggested as candidates for possible inclusion in the World Health Organization classification group of M/LN with eosinophilia (M/LN-eo). We report 12 patients with confirmed FLT3 rearrangement, six with t(12;13)/ETV6-FLT3; one with ins(13;22)/BCR-FLT3; and five with an unconfirmed partner gene located on chromosome bands 2p16, 3q27, 5q15, 5q35, and 7q36. Disease presentations were heterogeneous, including lymphoblastic leukemia/lymphoma, myeloid sarcoma, chronic eosinophilic leukemia, chronic myelomonocytic leukemia, and myelodysplastic syndrome. However, some common features were observed, such as extramedullary involvement (n = 7, 58%), associated eosinophilia in blood, bone marrow, or tissue (n = 8, 67%), multilineage involvement, either as biphasic myeloid/lymphoid neoplasms (n = 2) or mixed phenotype acute leukemia (n = 2). Mutations were detected in 4/8 (50%) patients by next-generation sequencing. None (0/10) had FLT3 or KIT mutations. Eleven patients received disease-based chemotherapy or hypomethylating agents, three received FLT3 inhibitors, and five patients proceeded to hematopoietic stem cell transplant. Together with a review of 16 cases published in the literature, it is apparent that M/LNs with FLT3 rearrangement show disease features reminiscent of members in the category of M/LN-eo with PDGFRA, PDGFRB, FGFR1, and PCM1/JAK2 rearrangement, characterized by a specific gene rearrangement, frequent eosinophilia, multi-lineage involvement and therapeutic benefit from kinase inhibitors.
具有 13q12/FLT3 重排的髓系/淋系肿瘤(M/LN)已被提议作为可能包含在伴有嗜酸性粒细胞增多的 M/LN(M/LN-eo)的世界卫生组织分类组中的候选者。我们报告了 12 例确诊的 FLT3 重排患者,其中 6 例为 t(12;13)/ETV6-FLT3;1 例为 ins(13;22)/BCR-FLT3;5 例未确认的伙伴基因位于染色体 2p16、3q27、5q15、5q35 和 7q36。疾病表现呈异质性,包括淋巴母细胞白血病/淋巴瘤、髓系肉瘤、慢性嗜酸性粒细胞白血病、慢性髓单核细胞白血病和骨髓增生异常综合征。然而,观察到一些共同特征,例如髓外累及(n=7,58%),伴有血液、骨髓或组织中的嗜酸性粒细胞增多(n=8,67%),多谱系受累,表现为双相髓系/淋系肿瘤(n=2)或混合表型急性白血病(n=2)。通过下一代测序检测到 4/8(50%)例患者存在突变。10 例患者均未发现 FLT3 或 KIT 突变。11 例患者接受了基于疾病的化疗或低甲基化剂治疗,3 例患者接受了 FLT3 抑制剂治疗,5 例患者进行了造血干细胞移植。结合文献中报道的 16 例病例回顾,显然具有 FLT3 重排的 M/LN 具有与 PDGFRA、PDGFRB、FGFR1 和 PCM1/JAK2 重排的 M/LN-eo 成员相似的疾病特征,表现为特定的基因重排、频繁的嗜酸性粒细胞增多、多谱系受累以及激酶抑制剂的治疗获益。