Case report: Oral ulceration of buccal mucosa

A 72-year-old woman referred to pain and burning on the right oral buccal mucosa due to an ulceration present for about 1 week, which caused her feeding difficulties (Figure 1). The patient, who had never smoked, was affected by diabetes mellitus type II, treated with oral hypoglycemic agents, hypertension, and osteoporosis on therapy with oral bisphosphonates (alendronate once a day) for 5 years. In 2005, she underwent quadrantectomy for breast cancer. Intraoral examination revealed oral ulceration of about 4 cm in diameter on the right buccal mucosa, with regular edges. The ulcer had a whitish/yellowish color and it had not a hard consistency. The patient was edentulous on the right mandibular side. Blood chemistry tests (blood count, plasma protein electrophoresis, and immunoglobulin dosage) performed showed no abnormalities. A biopsy was performed in two different areas of the lesion. The histologic examination showed a dense infiltrate of small CD3+ lymphocytes, predominantly mixed with large elements. That at the immunohistochemical examinations revealed to be CD20+, CD79a+, MUM1+ as well as CD30+ and in situ hybridization for Epstein–Barr virus (EBV/EBER+) (Figure 2a,b). Molecular biology investigation for lymphoproliferative lesions identified a clonal B and polyclonal pattern. A maxillofacial CT scan was also performed, which showed no sign of disease. Single necrotic ulcers of the oral cavity are lesions that can occur anywhere, with different etiology and whose clinical classification is not always easy to get. The histopathological examination of the lesion revealed an immunomorphological pattern that is characterized by an atypical CD30+ lymphoproliferative process, consistent with traumatic ulcerative granuloma associated with stromal eosinophilia (Sarangarajan et al., 2015). The examination also showed an atypical clonal proliferation of type B cells, described in the last few years in the literature in ulcerative granulomas. Given the histopathological features and the persistence of the lesion, the patient underwent hematological investigations, CT of the head and neck and bone marrow biopsy (Roberts et al., 2016). All the investigations performed were negative. Clonality B and positivity for EBV could better frame this lesion as an EBV+ mucocutaneous ulcer (EBVMCU). These lesions, recently described in the updated WHO classification of lymphoid neoplasms as a newly recognized entity associated with iatrogenic immunosuppression or age-related immunosenescence, do not fall into the neoplastic or lymphomatous forms and are characterized by spontaneous resolution in most of the cases (Swerdlow et al., 2016). The patient was referred to the Hematological Center of University Hospital of Parma, Italy: a bone marrow biopsy was performed, which showed no blood cell abnormalities. She underwent a close follow-up of the oral lesion which was treated with topical application of hyaluronic acid twice a day, with slow healing process of the lesion lasting several months. She did not develop any other lesion (Figure 3). Maddalena Manfredi: Conceptualization; writing – original draft; writing – review and editing; data curation; supervision; methodology. Maura Mureddu: Conceptualization; investigation; data curation. Cristina Mancini: Data curation; visualization; validation; methodology. Paolo Vescovi: Writing – review and editing. Open Access Funding provided by Universita degli Studi di Parma within the CRUI-CARE Agreement. Open Access Funding provided by Universita degli Studi di Parma within the CRUI-CARE Agreement. All authors have no conflicts of interest to disclose. The patient reported in this manuscript provided written informed consent for the publication of the case details. The data that support the findings of this study are openly available in wiley online library at https://onlinelibrary.wiley.com.