Küttner's tumor: Unusual presentation with bilateral involvement of the lacrimal and submandibular glands

Abstract

Chronic sclerosing sialadenitis, known as Küttner's tumor (KT), is a chronic inflammatory disease of the salivary gland characterized by progressive periductal fibrosis, dilated ducts with a dense lymphocyte infiltration and lymphoid follicle formation, and acinar atropy. We report a unique case of KT in a 30-year-old female who presented with time-serial involvement of the bilateral lacrimal and submandibular glands with reactive follicular hyperplasia of bilateral upper cervical lymph nodes. All tissue samples of the submandibular and lacrimal glands were typical of KT on pathologic examination. There was no evidence of lymphoepithelial lesions or intraepithelial lymphocytes. This is the first case of KT involving both lacrimal and submandibular glands, suggesting that KT is closely related to an active local immune process.