Lymphoplasmacytic Infiltrate of Regional Lymph Nodes in Küttner's Tumor (Chronic Sclerosing Sialadenitis): A Report of 3 Cases

Abstract

Regional lymph nodes of Küttner's tumor from 3 patients showed reactive follicular hyperplasia and prominent interfollicular plasmacytosis. The patients were 71-, 57-, and 73-year-old Japanese men. The polytypic nature of plasma cells was demonstrated by immunohistochemistry. There were numerous IgG-positive plasma cells with scattered IgA-positive or IgM-positive plasma cells. IgG4-positive cells comprised 25% to 40% of IgG-positive plasma cells. Prominent polyclonal hyperimmunoglobulinemia was demonstrated on laboratory test in 2 cases examined. An elevated serum IgG4 level (16%) was also demonstrated in 1 patient. The present 3 cases indicated that regional lymph node of Küttner's tumor may show reactive follicular hyperplasia and prominent interfollicular plasmacytosis and should be differentiated from various benign and malignant lymphoproliferative disorders including systemic rheumatic disease, plasma cell type of Castleman disease, and lymph node involvement of marginal B-cell lymphoma of the mucosa-associated lymphoid tissue type showing prominent plasma cell differentiation.

Systemic IgG4-related lymphadenopathy: a clinical and pathologic comparison to multicentric Castleman's disease

“…In contrast, multicentric Castleman's disease is characterized by increased serum IgG, IgM, and IgA levels, 16 which are caused by the increase in serum interleukin-6. 5 In this series, most patients showed cervical, hilar, mediastinal, and para-aortic lymph node swelling, and the lymph nodes were generally not very large (up to 2 cm). These findings were consistent with previous reports of IgG4-related lymphadenopathy.…”

“…[1][2][3][4][5][6][7][8][9][10] Clinical manifestations are apparent in the pancreas, bile duct, gallbladder, lacrimal gland, salivary gland, retroperitoneum, kidney, lung, and prostate, in which tissue fibrosis is pathologically induced. [1][2][3][4][5][6][7][8][9][10] Recently, many cases have been reported in Western countries, as well as in Japan. 3 However, the precise pathogenesis and pathophysiology of IgG4-related disease remain unclear.…”

“…As patients presenting with prominent fibrosis are frequently suspected of having malignant tumors, IgG4-related disease should be considered in the differential diagnosis to avoid unnecessary surgery. [1][2][3][4][5][6][7][8][9][10] Recent studies have reported that IgG4-related disease sometimes involves the systemic lymph nodes but clinicopathological characteristics have not been well documented yet. 3,4,11 In this study, we sought to clarify the clinicopathologic features of systemic IgG4-related lymphadenopathy.…”

“…Such abnormalities include an elevated serum IgG4 level and numerous IgG4-positive plasma cells infiltrating the affected tissue, and these disorders are classified as IgG4-related diseases. [1][2][3][4][5][6][7][8][9][10][11] Castleman's disease is a rather rare atypical lymphoproliferative disorder 12 classified according to the histopathologic findings of the affected lymph nodes as plasma cell type, hyaline-vascular type or a mixed-type variant of the two. 13,14 Patients with the plasma cell or the mixed-type variant frequently have systemic manifestations (so-called multicentric Castleman's disease), such as low-grade fever, fatigue, loss of appetite, and weight loss.…”

“…17,18 IgG4-related disease sometimes involves regional and/or systemic lymph nodes, 3,4,11 and is often clinically and/or histologically suspected to be multicentric Castleman's disease and/or malignant lymphoma. 5,6,11 However, systemic IgG4-related lymphadenopathy is either not mentioned or only briefly alluded to in previous reports. 3,11 In this study, we examined systemic IgG4-related lymphadenopathy in detail, and its differences from multicentric Castleman's disease, with specific reference to clinical and pathologic findings.…”