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Introduction: Orofacial granulomatosis (OFG) is a rare idiopathic, non-necrotizing granulomatous lesion of soft tissue in the orofacial region. It is clinically characterized by persistent swelling of lips (one or both), oral ulceration, and gingival enlargement. Case Presentation: A 21-year-old female patient presented with diffuse persistent anterior maxillary and mandibular gingival enlargement with upper lip swelling for the last 2 years. Clinical presentation, along with a histologic examination, confirmed a diagnosis of OFG. In this case report, OFG is presented with a detailed differential diagnosis. Conclusions: OFG is a disorder with inconstant clinical presentations and can be related to other granulomatous diseases. It has an uncertain cause, which may be associated with systemic diseases and can cause significant esthetic and functional problems. It has an unpredictable response to treatment, so to yield better clinical results, correct diagnosis and early treatment are essential.