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The aim of the study is to describe the clinical characteristics and prognosis of malignant transformation of adenomyosis in patients with endometrial cancer.In this retrospective descriptive study, the clinical data of patients with endometrial cancer (n = 127) who were admitted at our hospital between January 2006 and December 2013 were evaluated.Among the 127 patients with endometrial cancer, 24 patients had endometrial cancer concurrently with adenomyosis. Among these 24 patients, 3 were diagnosed with malignant transformation of adenomyosis. Postoperative pathological investigations in the cancer+adenomyosis group revealed endometrial adenocarcinoma of Grade I (n = 21) and II (n = 3). The patients with malignant transformation of adenomyosis were relatively younger than the other patients. In those 3 patients, both the estrogen and progesterone receptors were strongly expressed in eutopic endometrium and were weakly positive in ectopic endometrium.Although adenomyosis is usually benign, it might also be a precursor of malignant disease. As the incidence of adenomyosis malignant transformation is low, and its clinical manifestations are nonspecific, it may only be confirmed by postoperative pathological examination. Further investigations on larger sample size may provide additional data about prognosis of adenomyosis malignant transformation.