Adenomyoepithelioma is a rare tumor characterized by proliferation of two different cell populations. These tumors have a variable biological behavior. Majority of them are benign but have a tendency to recur locally. Malignant transformation is rare in this disease and distant metastasis is rarer still. We report here an unusual case of bilateral adenomyoepithelioma at an unusual age and showing a remarkable response to an unconventional drug, tyrosine kinase inhibitor "imatinib".