Abstract
PURPOSE: Carcinoma Cuniculatum (CC) is an exceptionally rare variant of squamous cell carcinoma with limited description in the literature. Oral Carcinoma Cuniculatum (OCC) continues to be a diagnostic dilemma as it generally presents as an indolent infection, often undergoing multiple biopsies and debridements prior to definitive diagnosis. This study aims to review the diagnostic challenges, clinical features, histopathology, and management of OCC. We specifically present a unique case of OCC in a patient with human immunodeficiency virus (HIV). METHODS: A review of the current literature on OCC was conducted, focusing on diagnostic delays, clinical presentation, histopathologic characteristics, and treatment approaches. Additionally, a case report of a 47-year-old male with OCC is presented, including clinical course, diagnostic workup, and management. RESULTS: The patient underwent multiple biopsies and surgical debridements over a 3-year period prior to definitive diagnosis. He was ultimately treated with subtotal maxillectomy and radial forearm free flap reconstruction. Final pathology demonstrated pT4a OCC. His clinical course was further complicated by previously undiagnosed HIV infection. Multidisciplinary tumor board recommendations included bilateral elective neck dissections and adjuvant radiation therapy. CONCLUSION: OCC remains a diagnostic challenge due to its indolent behavior and nonspecific presentation, often resulting in delayed diagnosis and advanced disease at treatment. This case highlights the importance of maintaining clinical suspicion for malignancy in persistent oral lesions and may represent the first reported case of OCC in a patient with HIV, thus raising new questions regarding the potential role of immunosuppression while propagating prior inquiries.