DDR markers' expression is variable in PPOELs, showing a tendency to increase along with dysplasia. Activated DDR mechanisms may play an important protective role at early stages of oral carcinogenesis, but probably suffer progressive deregulation, eventually failing to suppress malignant transformation.
Chronic sclerosing sialadenitis (CSS) is a chronic inflammatory condition of uncertain etiology, primarily affecting the submandibular glands. It has recently been proposed that CSS belongs to the immunoglobulin G4 (IgG4)-related disease spectrum, although the exact relationship between these two entities requires further elucidation. Our aim is to present a case of bilateral CSS of the sublingual glands and discuss the possible underlying pathogenetic mechanisms. A 66-year-old woman presented with a 7-month history of an asymptomatic firm swelling of the floor of the mouth. Clinically, a diffuse, multinodular swelling of elastic to firm consistency was observed across the sublingual folds bilaterally. An incisional biopsy revealed a lymphoplasmacytic infiltrate with lymphoid follicle formation, along with acinar degeneration, fibrosclerosis and ductal dilation. Immunohistochemical investigation ruled out lymphoproliferative diseases revealing a polyclonal lymphoplasmacytic population with only a few scattered IgG4-positive plasma cells. IgG4 serum levels were also normal. No treatment was administered with progressive spontaneous resolution of the swelling and no evidence of recurrence at 2 years follow-up. CSS is a benign salivary gland disorder, which, at least in a subset of cases, may represent an IgG4-associated disorder. Therefore, immunohistochemical evaluation of the IgG4/IgG plasma cell ratio and the IgG4 serum levels is a recommended investigation in the diagnostic work-up of CSS cases.
Ossifying fibromyxoid tumor (OFMT) is a mesenchymal neoplasm of uncertain lineage and intermediate biologic behavior. Involvement of the head and neck area is infrequent (10% to 15%) and intraoral presentation is very rare. An OFMT developing in the retromolar trigone of a 13-year-old male is presented, along with a comprehensive review of oral OFMT cases. Among 12 oral OFMTs (including the present case), most patients were male (72.7%), with a mean age of 30.3 (13-67) years. The tumors generally appeared as painless masses of firm or hard consistency (mean diameter 27.7 mm), most commonly located in the soft tissues of the mandible (50%). Common microscopic features included ossification, lack of atypia or high mitotic activity, and immunohistochemical positivity for S100 (5/7), vimentin (6/6), GFAP (3/6), and SMA (2/6). Recurrence was reported only in one case. Further characterization of this rare entity is needed to increase our understanding of its distinct clinical and histopathologic features.
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