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IgG4-Related Sclerosing Sialadenitis: Report of Three Cases.
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HOME > J Pathol Transl Med > Volume 45; 2011 > Article
Case Report IgG4-Related Sclerosing Sialadenitis: Report of Three Cases.
Ji Seon Bae, Joo Young Kim, Sang Hak Han, Seung Ho Choi, Kyung Ja Cho
Journal of Pathology and Translational Medicine 2011;45(0):S36-S40
DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S36
1Department of Otorhinolaryngology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea. shchoi@amc.seoul.kr
2Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea. kjc@amc.seoul.kr
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Chronic sclerosing sialadenitis, Mikulicz disease or Kuttner tumor has been recently recognized as a spectrum of IgG4-related sclerosing disease. IgG4-related disease is characterized by a high serum IgG4 level and tissue infiltration of IgG4-positive plasmacytes. We report three cases of chronic sclerosing sialadenitis with variably associated systemic involvement. All patients presented with a submandibular mass or swelling, and all the resected submandibular glands showed diffuse lymphocytic infiltration, lymphoid follicles, and septal fibrosis. Two of the specimens revealed numerous IgG-positive plasma cells, most of which were IgG4-positive on immunohistochemical staining. One of them was associated with dacryoadenitis and hypophysitis. The other patient had ureterorenal lesions. Immunohistochemical study was unavailable in remaining one case, but the histologic features along with elevated IgG level and associated pancreatitis supported the diagnosis. All patients received steroid therapy postoperatively and are doing well. Salivary gland involvement in IgG4-related fibrosclerosis should be recognized in systemic medical pathology.

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