Compared with FLT3-ITD, Y572C, V592A and V592A conferred high sensitivity to gilteritinib (Fig...Notably, JMD muts were also sensitive to quizartinib and sorafenib, whereas TKD muts are not...In vitro, the most common FLT3-JMD point muts confer a transforming potential to hematopoietic cells as predicted, and our data suggest that pts carrying FLT3-JMD point muts could respond to treatment with available FLT3 inhibitors. Finally, in contrast to TKD, JMD mutant cells are sensitive to type 2 FLT3 inhibitors, expanding the scope of treatment options for these pts.