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Oral submucous fibrosis (OSMF) is a chronic progressive debilitating disease of oral cavity leading to the stiffness of oral mucosa, burning and trismus resulting in marked rigidity, and an inability to open the mouth. As the medical treatment of OSMF is yet not standardized, the purpose of the present case study was to explore the effectiveness of osteopathic manipulative treatment (OMT) in patients with OSMF. The patient showed a significant increase in mouth opening from approximately 10 mm to 22 mm of mouth opening at the end of the treatment sessions.