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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.

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This article describes the first published case of coexistence in a child of a rare hybrid odontogenic ghost cell tumor and a solitary cutaneous pilomatrixoma. An 11-year-old boy presented with a large well-defined unilocular radiolucent lesion in the right posterior mandible. Marsupialization followed by enucleation of the remaining lesion at a later period was the treatment of choice. Histopathologic analysis revealed a hybrid tumor demonstrating areas identical to calcifying cystic odontogenic tumor, ameloblastoma, ameloblastic fibro-odontoma, ameloblastic fibromyxoma, and adenoid odontogenic tumor. A cutaneous nodule was also removed from the facial area and demonstrated classic features of pilomatrixoma on histopathology. Sixteen cases of hybrid calcifying cystic odontogenic tumor associated with odontogenic tumors other than ameloblastomas and odontomas are referred in the literature to date. Young males are frequently affected, and the mandible is the most common site of involvement. The occurrence in the same patient of 2 distinctive entities, which both demonstrate ghost/shadow cells, may be a coincidental finding or suggest a common origin regarding the histogenesis of these cells. Alternatively, future molecular studies may clarify possible genetic or/and predisposing factors for the development of these lesions.