Abundant IgG4-positive plasma cell infiltration characterizes chronic sclerosing sialadenitis (Kuttner's tumor)
Abundant IgG4-positive plasma cell infiltration characterizes chronic sclerosing sialadenitis (Kuttner's tumor)
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DOI：
10.1097/01.pas.0000164031.59940.fc
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发表时间：
2005-06-01
影响因子：
5.6
通讯作者：
Nakanuma, Y
中科院分区：
文献类型：
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作者：
Kitagawa, S;Zen, Y;Nakanuma, Y
慢性硬化性唾液腺炎 (CSS) 是一种隐源性唾液腺肿瘤样疾病。虽然怀疑其发病机制涉及免疫介导过程，并且偶尔有报道称 CSS 与硬化性胰腺炎（一种 IgG4 相关疾病）有关，但 CSS 的确切免疫病理过程仍然是推测性的。在本研究中，我们检查了 CSS（12 例）的临床病理学结果，与唾液酸石症（8 例）和干燥综合征（13 例）进行比较，并试图澄清 CSS 是否是 IgG4 相关疾病。所有 CSS 病例的颌下腺均受到影响。 CSS病例可分为两种类型：5例伴有唾液外腺组织硬化性病变（全身型），其余7例仅累及唾液腺（局部型）。前一型以男性为主，双侧唾液腺常受累，血清中常出现嗜酸粒细胞增多、丙种球蛋白和IgG升高。组织学上，所有 CSS 病例均显示明显的淋巴浆细胞浸润，伴有纤维化和腺小叶破坏。在所有 CSS 病例中，受影响的唾液腺均发现闭塞性静脉炎。免疫组化显示，CSS中IgG4/IgG阳性浆细胞比例超过45%，而对照组低于5%。 CSS 的临床病理逻辑特征与硬化性胰腺炎的相似性表明 CSS 中参与了类似的免疫病理过程和 IgG4 紊乱。病变中大量 IgG4 阳性浆细胞有助于区分 CSS 和其他形式的唾液腺炎。
Chronic sclerosing sialadenitis (CSS) is a cryptogenic tumor-like condition of the salivary gland(s). While immunemediated 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 logic 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.