Treatment of recurrent oral carcinoma cuniculatum with immune checkpoint blockade: A case report and literature review

Background

Carcinoma cuniculatum (CC) is a rare variant of carcinoma that can arise in the oral cavity. CC can be locally aggressive but rarely metastasizes, and there are few reports of treatment with systemic therapy.

Methods

We present a case of a 44-year-old man with T4bN0 oral carcinoma cuniculatum of the retromolar trigone.

Results

Despite resection with wide margins and adjuvant radiation, the disease recurred 14 months later and was extensive. Following positive margins on a repeat resection, the patient opted for systemic therapy. Despite a PD-L1 combined positive score of 15, the patient did not respond to immunotherapy (pembrolizumab).

Conclusion

Though it has a low propensity for metastasis, oral CC is a locally aggressive variant of squamous cell carcinoma, and recurrences are difficult to treat. Although reports of systemic therapy for CC are limited, this case suggests that immunotherapy may have limited activity in this oral cancer variant.