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OBJECTIVE: This systematic review seeks to examine and synthesize the available literature on the clinical and pathological features of leishmaniasis affecting the oral cavity. STUDY DESIGN: A systematic review was conducted in accordance with PRISMA guidelines and registered in PROSPERO (CRD420251070738). PubMed, Web of Science, and Scopus were searched up to January 11, 2025. Inclusion criteria covered case reports and series with histopathological confirmation. Clinical, pathological, diagnostic, and treatment data were extracted. Study quality was assessed using the Joanna Briggs Institute (JBI) tools. RESULTS: Fifty-one studies (40 case reports, 11 case series) involving 132 patients were included. The mean age was 41.2 years, with 87.8% male. Leishmania infantum (16.6%) and L. braziliensis (6.1%) were the most reported species. Ulcerative lesions (37.8%) and nodules (8.3%) predominated, commonly affecting the upper lip, soft palate, and buccal mucosa. Histopathology was the primary diagnostic method (78.7%). Meglumine antimoniate (37.1%) was the most used treatment. Recurrence occurred in 25% of cases after an average of 16.4 months of follow-up. CONCLUSION: Oral leishmaniasis is an uncommon clinical entity with varied presentation, requiring a high index of suspicion for accurate diagnosis. This review offers essential information to support early recognition and appropriate management.