Chronic sclerosing sialadenitis of the bilateral submandibular glands in childhood - a diagnostic dilemma.

Chronic sclerosing sialadenitis (CSS), currently included in the group of immunoglobulin G4 (IgG4)-related diseases, is an under-recognized inflammatory lesion that afflicts mostly the submandibular gland of 40-60 years adults. To our knowledge, only one case of CSS located in the submandibular gland has been reported in childhood to date. We present a case of CSS in a 5-year-old male child. He presented with bilateral submandibular swellings that clinically resembled discrete lumps, suspected to be tumors. The completely resected tumors composed predominantly of dense lymphoplasmacytic inflammatory infiltrate rich in IgG4-positive cells [77-90 IgG(+) cells per high-power field; IgG4(+)∕IgG(+) cells ratio of 42.77%]. We discuss the peculiarities of this case, and we also review the literature on CSS.

Eosinophilic sialodochitis: a case report of a rare disease.

Eosinophilic sialodochitis (ES) is a rare clinical entity which presents as recurrent major salivary gland swelling and the presence of eosinophil-rich mucus plugs or histopathological evidence of eosinophilic infiltration around the larger salivary gland ducts. We present a case of ES not related with underwent a left submaxillectomy because recurrent episodes of submandibular gland swelling associated for the last three years. The laboratory workup demonstrated eosinophils slightly elevated and high levels of immunoglobulin E (IgE) in peripheral blood. Pathology slides confirmed the final diagnosis of ES, showing a dense periductal eosinophil-rich inflammatory infiltrate and periductal fibrosis. Although the entity is well described in the literature, diagnosis is often difficult due to its clinical presentation being similar to other conditions. Lesions that should be taken into consideration in differential diagnosis are mentioned. This will be the first ES case report in Eastern European literature.