Massive lymphocytic infiltration of the leiomyomas of the uterus is rare and causes diagnostic difficulties. The pathogenesis of this lesion is
not clear. The recognition of its distinct histological features is important to avoid possible confusion with differential diagnosis including malignant lymphoma. Only 20 cases have been reported in the literature. We present a case of leiomyoma of the uterus with massive lymphocytic infiltration, and we investigated the literature.