Metachronous occurrence of Epstein-Barr virus-positive mucocutaneous ulcers suspected to be related to periodontitis: A case report

Epstein-Barr virus-positive mucocutaneous ulcer (EBVMCU) is an Epstein-Barr virus-associated B-cell lymphoproliferative disorder that usually occurs in immunosuppressed patients. In this report, we describe the case of a 75-year-old man who repeatedly developed EBVMCUs at different gingival sites. He had a history of B-cell lymphoma and had achieved remission with chemotherapy 2 years before visiting our hospital. In all three EBVMCUs, radiographic examination revealed alveolar bone resorption beneath the EBVMCUs, and the ulcers regressed after spontaneous loss or extraction of adjacent teeth. Four years after the third appearance, no new lesions emerged, and the periodontal disease was controlled. Histopathological features showed that ulcers in the first and third instances were classical Hodgkin lymphoma-like and that in the second instance was a diffuse large B-cell lymphoma-like EBVMCU. Programmed death-ligand 1 (PD-L1) expression was examined using a PD-L1 antibody (SP142); only the ulcer in the third instance was positive. This case suggests a possible association between EBVMCU and periodontitis and that the past history of chemotherapy can be a risk factor for systemic immunosuppression causing EBVMCU.