A benign lymphoepithelial lesion (BLEL) is an idiopathic inflammation of the parotid gland, characterized by hyperplasia, lymphocyte infiltration and the formation of epimyoepithelial islands, as well as atrophy of the gland parenchyma. Common treatment methods include immunosuppression and glucocorticoid therapies, in addition to surgical dissections. Cytokine-induced killer (CIK) cells sensitized to specific antigens by dendritic cells (DCs) are used in DC-CIK biotherapy. The present study reports the case of a 22-year-old female suffering from a postoperative recurrent BLEL on the left parotid gland, which was gradually increasing. Following initial unsuccessful conservative treatment attempts, a 10-day course of DC-CIK therapy was initiated, after which the lesion in the gland area was reduced in size and local infection and skin ulcerations were improved. DC-CIK biotherapy was continued for three months (four sessions of 10-day treatments with a 10-day break in between) until the lesion disappeared and the skin ulceration was healed. Computerized tomography scans of the parotid gland revealed complete remission of the primary lesion and recovery of the bone destruction. The patient was discharged and remained stable with no sign of recurrence during a 10-month follow-up period. In the present case report, a successful DC-CIK adoptive cellular immunotherapy treatment for a BLEL was described for the first time.