title: Abundant IgG4-positive plasma cell infiltration characterizes chronic sclerosing sialadenitis (Küttner's tumor).
abstract: Chronic sclerosing sialadenitis (CSS) is a cryptogenic tumor-like condition of the salivary gland(s). While immune-mediated processes are suspected in its pathogenesis, and CSS is occasionally reported to be associated with sclerosing pancreatitis, an IgG4-related disease, the exact immunopathologic processes of CSS remain speculative. In this study, we examined the clinicopathologic findings of CSS (12 cases) in comparison with sialolithiasis (8 cases) and Sjogren's syndrome (13 cases), and tried to clarify whether CSS is an IgG4-related disease or not. Submandibular gland(s) were affected in all cases of CSS. CSS cases could be divided into two types: 5 cases were associated with sclerosing lesions in extrasalivary glandular tissue (systemic type), while only salivary gland(s) were affected in the remaining 7 cases (localized type). In the former type, which showed male predominance, bilateral salivary glands were frequently affected, and eosinophilia and elevations of gamma-globulin and IgG in serum were frequently found. Histologically, all cases of CSS showed marked lymphoplasmacytic infiltration admixed with fibrosis and the destruction of glandular lobules. Obliterative phlebitis was found in the affected salivary glands in all cases of CSS. Immunohistochemically, the proportion of IgG4/IgG-positive plasma cells was more than 45% in CSS, while it was less than 5% in controls. The resemblance of the clinicopathologic features of CSS with those of sclerosing pancreatitis suggests the participation of a similar immunopathologic process with IgG4 disturbance in CSS. The abundance of IgG4-positive plasma cells in the lesions would be useful for distinguishing CSS from other forms of sialadenitis.
authors.affiliation: Department of Human Pathology, Kanazawa University Graduate School of Medicine, 13-1 Takaramachi, Kanazawa 920-8640, Japan.
authors.forename: Satoshi
authors.initials: S
authors.lastname: Kitagawa
authors.forename: Yoh
authors.initials: Y
authors.lastname: Zen
authors.forename: Kenichi
authors.initials: K
authors.lastname: Harada
authors.forename: Motoko
authors.initials: M
authors.lastname: Sasaki
authors.forename: Yasunori
authors.initials: Y
authors.lastname: Sato
authors.forename: Hiroshi
authors.initials: H
authors.lastname: Minato
authors.forename: Kishichiro
authors.initials: K
authors.lastname: Watanabe
authors.forename: Hiroshi
authors.initials: H
authors.lastname: Kurumaya
authors.forename: Kazuyoshi
authors.initials: K
authors.lastname: Katayanagi
authors.forename: Shinji
authors.initials: S
authors.lastname: Masuda
authors.forename: Hideki
authors.initials: H
authors.lastname: Niwa
authors.forename: Koichi
authors.initials: K
authors.lastname: Tsuneyama
authors.forename: Katsuhiko
authors.initials: K
authors.lastname: Saito
authors.forename: Joji
authors.initials: J
authors.lastname: Haratake
authors.forename: Kiyoshi
authors.initials: K
authors.lastname: Takagawa
authors.forename: Yasuni
authors.initials: Y
authors.lastname: Nakanuma
mesh_terms: D000368:Aged; D015209:Cholangitis, Sclerosing; D002908:Chronic Disease; D005260:Female; D006801:Humans; D007074:Immunoglobulin G / Q000276:immunology*; D008297:Male; D008875:Middle Aged; D010195:Pancreatitis / Q000150:complications; D010950:Plasma Cells / Q000276:immunology*; D015494:Salivary Gland Calculi / Q000150:complications; D012598:Sclerosis / Q000473:pathology; D012793:Sialadenitis / Q000150:complications / Q000276:immunology* / Q000473:pathology; D012859:Sjogren's Syndrome / Q000150:complications
chemical_list: D007074:Immunoglobulin G