Uterine leiomyoma with massive lymphocytic infiltration is known to be associated with Gona-dotropin releasing hormone (GnRH) agonist treatment. The lymphocytic cells in those cases were composed predominantly of T-lymphocytes. We report an unusual case of uterine leiomyoma with massive lymphocytic infiltration, composed predominantly of B-lymphocytes, without a history of GnRH agonist treatment. A 59-year-old woman underwent a transvaginal hysterectomy for uterine leiomyomas. Microscopically, the leiomyoma showed a massive infiltration of the lymphocytes, histiocytes, and also showed scattered plasma cells and many lymphoid follicles. The lymphocytic infiltrates were confined to the leiomyoma. These lymphocytic cells mainly represented the B-cell phenotype. She had no history of GnRH agonist treatment. To the best of our knowledge, This is the first reported case in Korea.