Objectives: Oral Squamous cell carcinoma (OSCC) is primarily a disease that mainly occurs in males in their sixth and seventh decades of life and is rare in young adults. Study Design: In this retrospective study, records of patients under the age of 40, with the diagnosis of OSCC in the Oral Medicine Department of Mashhad Dental Faculty during the past 13 years were analyzed. Their socioeconomic data, demographic, clinical and histopathological characteristics, risk factors, familial history were assessed and applicable studies and case reports in the literatures were reviewed. PCR (Polymerase chain reaction) analysis was also done for detection of human papilloma virus (HPV). Results: From 158 cases of OSCC diagnosed in our centre, 21 patients were younger than 40 years. Most of them were young men (12 cases). There was no significant risk factor in this group. The most common site of involvement was the tongue. The most common clinical presentation was exophytic lesion with ulcer. No HPV DNA was detected in these patients. Conclusion: Characteristics of OSCC in young patients are different from older age group. Major risk factors (smoking and alcohol consumption and HPV) were not etiologic factors for OSCC in young patients in our province. © Medicina Oral S. L.