In this paper an unusual case of lymphadenopathy associated with Actinomyces infection is reported. In the literature, there are only two cases presented with histopathologic features. A forty-year-old male patient was admitted to the hospital with painless mass on the neck. Clinical examination revealed a firm mass on the right submandibular region. The patient had no malignancy history. A cystic mass radiologically compatible with abscess was detected adjacent to the submandibular gland. Sialadenitis and lymphadenitis were considered but the mass was refractory to the antibiotherapy. The mass and the right submandibular gland were excised. Histopathologic evaluation of the lymph node revealed characteristic sulfur granules of Actinomyces.