Chronic sclerosing dacryoadenitis - Report of 2 cases

1068 1092

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Authors: Ji Eun Kwon ; Sang Kyum Kim ; Sang-Ryul Lee ; Woo-Ick Yang ; Haeryoung Kim

MeSH: Adult ; Aged ; Autoimmune Diseases/immunology ; Autoimmune Diseases/pathology* ; Cell Count ; Cholangitis, Sclerosing/immunology ; Cholangitis, Sclerosing/pathology ; Chronic Disease ; Dacryocystitis/immunology ; Dacryocystitis/pathology ; Female ; Humans ; Immunoglobulin G/blood* ; Lymph Nodes/immunology ; Lymph Nodes/pathology* ; Lymphatic Diseases/immunology ; Lymphatic Diseases/pathology* ; Male ; Middle Aged ; Pancreatitis/immunology ; Pancreatitis/pathology ; Plasma Cells/immunology ; Plasma Cells/pathology ; Sclerosis/immunology ; Sclerosis/pathology ; Sialadenitis/immunology ; Sialadenitis/pathology

Keywords: IgG4-related sclerosing disease ; IgG4-related autoimmune disease ; autoimmune pancreatitis ; lymph node ; lymphadenopathy ; lymphoma ; immunohistochemistry

Abstract: Chronic sclerosing dacryoadenitis is a rare and under-recognized chronic inflammatory disease of the lacrimal gland. We describe 2 patients with a localized type of chronic sclerosing dacryoadenitis. Both patients presented with a slowly growing painless mass of the eyelid mimicking a tumorous lesion. The morphologic findings of the masses excised under the clinical diagnosis of lymphoma closely recapitulate those of chronic sclerosing sialadenitis (Kuttner tumor). Immunohistochemical staining demonstrated an increased population of IgG4-positive plasma cells confirming that this disease also belongs to the spectrum of a recently described IgG4-related sclerosing disease