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Titre du document / Document title

Systemic lgG4-related lymphadenopathy : a clinical and pathologic comparison to multicentric Castleman's disease

Auteur(s) / Author(s)

SATO Yasuharu ; KOJIMA Masaru ; TAKATA Katsuyoshi ; MORITO Toshiaki ; ASAOKU Hideki ; TAKEUCHI Tamotsu ; MIZOBUCHI Kohichi ; FUJIHARA Megumu ; KURAOKA Kazuya ; NAKAI Tokiko ; ICHIMURA Kouichi ; TANAKA Takehiro ; TAMURA Maiko ; NISHIKAWA Yuriko ; YOSHINO Tadashi ;

Résumé / Abstract

ΙgG4-related disease sometimes involves regional and/or systemic lymph nodes, and often clinically and/or histologically mimics multicentric Castleman's disease or malignant lymphoma. In this study, we examined clinical and pathologic findings of nine patients with systemic lgG4-related lymphadenopathy. None of these cases were associated with human herpes virus-8 or human immunodeficiency virus infection, and there was no T-cell receptor or immunoglobulin gene rearrangement. Histologically, systemic lgG4-related lymphadenopathy was classified into two types by the infiltration pattern of lgG4-positive cells: interfollicular plasmacytosis type and intra-germinal center plasmacytosis type. The interfollicular plasmacytosis type showed either Castleman's disease-like features or atypical lymphoplasmacytic and immunoblastic proliferation-like features. By contrast, the intra-germinal center plasmacytosis type showed marked follicular hyperplasia, and infiltration of lgG4-positive cells mainly into the germinal centers, and some cases exhibited features of progressively transformed germinal centers. Interestingly, eight of our nine (89%) cases showed eosinophil infiltration in the affected lymph nodes, and examined patients showed high elevation of serum IgE. Laboratory examinations revealed elevation of serum lgG4 and soluble interleukin-2 receptors. However, the levels of interleukin-6, C-reactive protein, and lactate dehydrogenase were within normal limits or only slightly elevated in almost all patients. One patient showed a high interleukin-6 level whereas C-reactive protein was within the normal limit. Autoantibodies were examined in five patients and detected in four. Compared with the previously reported cases of multicentric Castleman's disease, our patients with systemic lgG4-related lymphadenopathy were significantly older and had significantly lower C-reactive protein and interleukin-6 levels. In conclusion, in our systemic lgG4-related lymphadenopathy showed pathologic features only partially overlapping those of multicentric Castleman's disease, and serum data (especially C-reactive protein and interleukin-6) are useful for differentiating the two. Our findings of eosinophil infiltration in the affected tissue and elevation of serum IgE may suggest an allergic mechanism in the pathogenesis of systemic lgG4-related lymphadenopathy.

Revue / Journal Title

Modern pathology    ISSN  0893-3952 

Source / Source

2009, vol. 22, no4, pp. 589-599 [11 page(s) (article)]

Langue / Language


Editeur / Publisher

Nature Publishing Group, Basingstoke, ROYAUME-UNI  (1988) (Revue)

Mots-clés d'auteur / Author Keywords

systemic lgG4-related lymphadenopathy


C-reactive protein




immunoglobulin E


multicentric castleman's disease


Localisation / Location

INIST-CNRS, Cote INIST : 21992, 35400018746017.0120

Nº notice refdoc (ud4) : 21311770

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