Small cell neuroendocrine carcinoma (SmCC) is rarely found in the head and neck region. With most cases having been reported in the larynx, SmCC of the salivary glands remains a rare clinical entity. Similar to that in the lung and larynx, SmCC of the salivary glands has an aggressive nature associated with strong metastatic potential, although its prognosis is relatively better. The authors experienced a case of a 50-year-old male patient who presented with a submandibular mass. After surgical excision, histopathologic and immunohistochemical examination confirmed the mass to be primary small cell neuroendocrine carcinoma of the submandibular gland. The patient received postoperative chemoradiation and has remained disease-free during 36 months of follow-up.