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Background: Oral papilloma is benign lesion while there is concern about high-risk types of Human Papilloma Virus (HPV) for cancer. Case Presentation: The patient was a 30-year-old woman who presented to the School of Dentistry, Semnan University of Medical Sciences for orthodontic treatment. Intraoral examinations revealed a prominent white papule on the tip of the tongue. Histopathologic exanimation revealed the proliferation of hyper keratinized stratified squamous epithelium arranged in projections with fibrovascular connective tissue cores, cell irregularity with hyperchromatic nucleus and koilocytes within the epithelium which confirmed the diagnosis of squamous papilloma (SP) with mild to moderate dysplasia. The patient was instructed to take care of the sexual behaviors and the injection of Gardasil vaccine was recommended. A month later, there was no evidence of a lesion. Conclusion: Though SPs are common lesions in the oral cavity, occurrence of dysplasia on SPs on the anterior part of the mouth is rare. Surgical removal supplemented by administration of Gardasil vaccine can be the treatment of choice in these cases. The significance of close attention to the oral lesions and consultation with an Oral and Maxillofacial Medicine Specialist is highly recommended in order to accurate diagnosis and timely treatment.

Dysplasia in Papilloma at the Tip of the Tongue; A Rare Entity-Importance of on Time Prevention