Oral Carcinoma Cuniculatum: A Rare, Clinically Challenging Entity-A Case Report and Updated Systematic Review.

Abstract

Oral carcinoma cuniculatum (OCC) is a rare, well-differentiated variant of oral squamous cell carcinoma (SCC) that often mimics benign lesions, resulting in delayed diagnosis. Evidence on its clinical behavior and risk of recurrence remains limited. We report a representative OCC case and conducted a PRISMA-based systematic review of PubMed, Embase, Cochrane Library, Google Scholar, and Airiti Library, including studies with patient-level data. Forty-six studies comprising 115 patients were included. The gingiva was the most common primary site. Nodal metastasis was reported in 9.0% (8/89), and the overall recurrence rate was 15.8%, predominantly local. Higher recurrences were found among patients receiving neck dissection or adjuvant therapy, which likely reflected selection bias rather than a treatment effect. OCC shows a low nodal metastatic rate and mainly local recurrence. Routine elective neck dissection may not be required in clinically N0 patients, but this hypothesis requires validation in larger, multi-institutional cohorts.