We present a case of a 38-year-old person living with HIV (PLHIV) diagnosed with refractory idiopathic hypereosinophilic syndrome (HES). His initial symptoms were pruritic skin rash, dry cough, and diarrhea. The patient was unresponsive to corticosteroid as a first-line therapy. Subsequent treatment with Imatinib mesylate 100 mg per day resulted in excellent and sustained hematological and clinical response, with a likely molecular response as well.