Chronic sclerosing sialadenitis: report of a rare and atypical case
Rafaela Savio MelzerCíntia MilaniFernanda Noguez SumRui Cesar de Bittencourt DruszczJuliana Lucena Schussel
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A Chronic Sclerosing Sialodenitis is a rare and benign lesion that preferentially affects a submandibular gland of male patients. Although its etiology is considered unknown, it is suggested that the secretion of IgG4 immunoglobulin may be useful for its manifestation. This pathology has specific clinical characteristics for malignant neoplasms of the salivary glands, where the recommended treatment is a surgical excision. The present study presents an atypical case of chronic sclerosing sialodenitis that involves a woman's soft palate, where there was a complete resolution of the clinical case, in which the conservative treatment was chosen by the use of medications.Keywords:
Etiology
Sialadenitis
Surgical excision
Sialadenitis
Salivary Gland Diseases
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Abstract Background Sjögren’s syndrome is a systemic autoimmune disease characterized by immune cells predominantly infiltrating the exocrine glands and frequently forming ectopic lymphoid structures. These structures drive a local functional immune response culminating in autoantibody production and tissue damage, associated with severe dryness of mucosal surfaces and salivary gland hypofunction. Cenerimod, a potent, selective and orally active sphingosine-1-phosphate receptor 1 modulator, inhibits the egress of lymphocytes into the circulation. Based on the mechanism of action of cenerimod, its efficacy was evaluated in two mouse models of Sjögren’s syndrome. Methods Cenerimod was administered in two established models of Sjögren’s syndrome; firstly, in an inducible acute viral sialadenitis model in C57BL/6 mice, and, secondly, in the spontaneous chronic sialadenitis MRL/lpr mouse model. The effects of cenerimod treatment were then evaluated by flow cytometry, immunohistochemistry, histopathology and immunoassays. Comparisons between groups were made using a Mann-Whitney test. Results In the viral sialadenitis model, cenerimod treatment reduced salivary gland immune infiltrates, leading to the disaggregation of ectopic lymphoid structures, reduced salivary gland inflammation and preserved organ function. In the MRL/lpr mouse model, cenerimod treatment decreased salivary gland inflammation and reduced T cells and proliferating plasma cells within salivary gland ectopic lymphoid structures, resulting in diminished disease-relevant autoantibodies within the salivary glands. Conclusions Taken together, these results suggest that cenerimod can reduce the overall autoimmune response and improve clinical parameters in the salivary glands in models of Sjögren’s syndrome and consequently may reduce histological and clinical parameters associated with the disease in patients.
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Background Among the complex of HTLV ‐associated diseases, Sjögren's syndrome ( SS ) is one of the most controversial. This work aims to detect morphological and inflammatory alterations, including clues of the presence of HTLV ‐1, in minor salivary glands of patients with dryness symptoms. Methods We have assessed HTLV ‐1‐seropositive patients ( HTLV ‐1 group) and patients with SS ( SS group). We used formalin‐fixed, paraffin‐embedded minor salivary gland tissue to evaluate the morphological aspects and, by means of immunohistochemistry, the presence of Tax protein, CD 4, CD 8 and CD 20 cells. Additionally, viral particles and proviral load were analysed by PCR . Results The HTLV ‐1 group had the highest prevalence of non‐specific chronic sialadenitis (85.71%; P = 0.017) and greater amount of T CD 8 + cells. In the SS group, focal lymphocytic sialadenitis (80%; P = 0.017) prevailed, with a greater amount of B CD 20 + . Both immunohistochemistry and PCR identified the Tax protein and its gene in the salivary glands of both groups and in similar proportions. Conclusion The results indicate that HTLV ‐1‐seropositive patients have different patterns of morphological/inflammatory alterations, suggesting a likely difference in the process of immune activation.
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Abstract Objective This study was undertaken to determine whether induction of systemic inflammation accelerates the development of Sjögren's syndrome (SS) in genetically susceptible mice. Methods Female (NZB × NZW)F 1 mice were treated with either Freund's incomplete adjuvant (IFA) or phosphate buffered saline (PBS) at monthly intervals. Salivary gland function was monitored by measuring pilocarpine‐induced saliva volume. Mice were killed at different time points and examined for sialadenitis and salivary gland–infiltrating cells. Sera were analyzed for autoantibodies to salivary gland antigens, nuclear antigens, and Ro60. Results While IFA‐treated mice had significantly decreased salivary secretion 7 weeks after the initial treatment, salivary secretion did not decrease in PBS‐treated controls until 17 weeks. At 7 weeks, the severity of sialadenitis and the number of T and B cells infiltrating the salivary glands did not differ between the 2 groups. However, at this time point IFA‐treated mice showed significantly higher frequencies of CD11c low , B220+, Ly6C+, mouse PDCA‐1+ dendritic cells (DCs) in the salivary glands. While levels of autoantibodies did not differ between the 2 groups at early time points, by late time points IFA‐treated mice had higher levels. The gland dysfunction observed in IFA‐treated mice at earlier time points did not correlate with the severity of sialadenitis or levels of autoantibodies. Instead, it was associated with increased frequency of plasmacytoid DCs in the gland. Conclusion Our data suggest that generalized inflammatory stimuli can accelerate the development of SS‐like disease in (NZB × NZW)F 1 mice, and that gland dysfunction in SS can develop prior to the generation of a robust adaptive autoimmune response.
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