Chronic myeloid leukemia could be treated by tyrosine kinase inhibitor successfully. However, a few rare but lethal adverse reactions have been reported. In cases of dasatinib-induced pulmonary arterial hypertension (PAH), the majority generally recovers incompletely and needs alternative TKIs for CML treatment. A 34-year-old woman diagnosed with chronic myeloid leukemia (CML) had taken dasatinib for more than two years. Before dasatinib, she had been under nilotinib treatment for two years, achieving complete molecular response at sixth months, but with persistent hyperbilirubinemia and epigastric discomfort. Twenty months after shifting to dasatinib, she began to experience shortness of breath. Severe pulmonary arterial hypertension was subsequently diagnosed. After withdrawal of dasatinib, the PAH thoroughly resolved. Moreover, complete molecular response was maintained for more than two years without additional tyrosine kinase inhibitor (TKIs) administration. The strength of our case is that the patient fully recovered from the dasatinibinduced severe PAH and kept complete molecular response without taking other TKIs. Early detection of the adverse effect of dasatinib is important for better outcomes.