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Oral submucous fibrosis (OSF) is a chronic condition of the oral mucosa, first described among five East African women of Indian origin under the term "atrophia idiopathica (tropica) mucosae oris." It is characterized by a generalized submucous fibrosis. The pathogenesis of the disease is not well-established, but epidemiological evidences strongly indicate the association of betel quid habit. It is logical to hypothesize that the increased collagen production or reduced collagen degradation is the possible mechanism in the development of the disease. The malignant potentiality and also the origin of cancers in different intraoral locations in OSF patients is attributable to the generalized epithelial atrophy. Here, a clinical case of OSF in a 36-year-old male patient is being discussed, encompassing the pathogenesis and malignant potentiality of the disease.