Follicular lymphoma manifests as multiple erosive and proliferative lesions of the oral mucosa: case report and brief literature review

Abstract

Background: Erosion is one of the most common and basic lesions of oral mucosal diseases. Long-term refractory oral erosions, induced by autoimmune blistering diseases, infectious diseases, malignant diseases, and some rare conditions, may substantially reduce the quality of life of patients or even constitute a life-threatening condition, resulting in a clinical dilemma regarding the accurate diagnosis and precise management of these diseases. As a special type of malignant lymphoma, most lesions of follicular lymphoma (FL) in the oral mucosa present as masses or swelling of the oral mucosa, while emerging novel presentations lead to intractable diagnoses. Hence, diagnostic algorithms for such diseases are clinically required. CASE PRESENTATION: A 55-year-old female patient presented to the clinic with long-lasting oral mucosal erosions and proliferative lesions. Blood tests, pathological examinations of oral lesions including haematoxylin-eosin (HE) staining, and direct immunofluorescence precluded all of the potential diagnoses described previously. Unexpectedly, positron emission tomography/computed tomography (PET/CT) and abdominal CT of the patient revealed a dense mass in the retroperitoneal area, and the final diagnosis of the retroperitoneal mass was FL. After three courses of chemotherapy conducted by the haematologist, the erosion and proliferative lesions in the patient's oral mucosa had significantly improved. HE and immunohistochemical staining results of intraoral lesions also confirmed it as oral FL. The successful diagnosis of FL in this case is of great clinical significance, as the oral and abdominal FL were treated in a timely manner to avoid unfavourable outcomes.

Conclusions: To the best of our knowledge, this is the first case of FL that exhibited widespread erosions interspersed with proliferative lesions. Clinicians should be aware of oral FL or seek systemic factors in the presence of similar refractory oral erosions when treatment is non-responsive and the diagnosis is intractable.

Keywords: Case report; Erosive and proliferative lesions; Follicular lymphoma; Oral manifestation.

Fig. 1

Oral mucosal manifestations of the patient. Widespread erosive lesions on the dorsum of tongue, interspersed with several mucosal proliferative or nodule-like lesions (A). Linear or reticular white striae with erosions and hyperemia could be observed on bilateral tongue margins (B), right angle of the mouth (C), and the lower part of right and left buccal mucosa (C and D). Black arrows: proliferative lesions with erosive surface. Blue arrows: reticular white striae. White arrows: erosions. All images were captured at the resolution of 300 dpi

Fig. 2

Pathological examination of the oral lesion. A HE staining of the right upper lip mucosa showed nodular aggregation of lymphocytes and plasma cells in the subepithelial connective tissue and around the blood vessels. B-D IHC result of the right upper lip mucosa showed follicular-like structures presenting as CD20 ( +), BCL-2 ( +), CD21 ( +), respectively. Black arrow: nodular aggregation of lymphocytes. All stained sections were imaged using Aperio Versa (Leica, Germany) at 100 × and 200 × magnification, and the images of stained slides were acquired at the resolution of 300 dpi. Abbreviations: HE: hematoxylin–eosin; IHC: immunohistochemistry; BCL-2: B-cell lymphoma-2

Fig. 3

Recent follow-up imaging of the patient’s dorsum of tongue shows significant remission of the erosions and proliferative lesions after undergoing standardized chemotherapy against the follicular lymphoma. The image was captured at the resolution of 300 dpi

Fig. 4

Diagnostic algorithms for diseases characterized by refractory erosion of the oral mucosa proposed by the authors of this work. Abbreviations: HIV: human immunodeficiency virus; HE: hematoxylin–eosin; OPMD: oral potentially malignant disorders; OLP: oral lichen planus; OLK: oral leukoplakia; OE: oral erythroplakia; DLE: discoid lupus erythematosus; DIF: direct immunofluorescence; CT: computed tomography; PAMS: paraneoplastic autoimmune multiorgan syndrome; PNP: paraneoplastic pemphigus; PSV: pyostomatitis vegetans; OSCC: oral squamous cell carcinoma; qPCR: quantitative polymerase chain reaction; TB: tuberculosis; LCH: Langerhans cell histiocytosis; IHC: immunohistochemistry; FL: follicular lymphoma