An unusual presentation of chronic sclerosing sialadenitis of submandibular gland (Kuttner's tumour).

Kuttner's tumour, also known as chronic sclerosing sialadenitis, is a localised form of IgG4 disease which presents as asymptomatic submandibular gland swelling. The diagnosis is usually based on histopathology and immunohistochemistry. A 33-year-old woman presented with acute-onset pain and swelling in the right submandibular region. Clinical examination showed an enlarged submandibular gland, and CT showed a calculus in the Wharton's duct. After treating the acute phase with antibiotics, the patient underwent submandibular gland excision and calculus removal. Histopathology of the specimen showed areas of periductal sclerosis, acinar atrophy and intense lymphoplasmacytic infiltrates with occasional eosinophils. The IgG4 to IgG plasma cell ratio was >40%, suggestive of an IgG4-related disease. The authors have chosen to report this case because of the unusual presentation of IgG4 disease as acute sialadenitis.