Chronic sclerosing sialadenitis (Küttner's tumor): an uncommon cause of submandibular swelling

Sir,

Chronic sclerosing sialadenitis (CSS) or Küttner’s tumour, a rare benign condition affecting submandibular gland, is now believed to be a localised type of IgG4-related disease. It presents as an asymptomatic swelling. Histopathology is the gold standard for diagnosis.¹ Here we discuss the same in a 55-year-old male presenting as a painless submandibular swelling.

A 55-year-old male presented with painless right submandibular swelling since 4 to 5 months. On examination there was a firm right submandibular mass measuring 5 x 4 cm. Clinically the features were suggestive of right chronic submandibular sialadenitis. Whole right submandibular gland was excised and sent for histopathological examination.

Grossly a submandibular tissue measuring 4×3×2 cm was received. Outer surface was encapsulated and nodular. Cut surface was grey white firm and lobulated with intervening areas of fibrosis. Section from submandibular glands shows preserved lobular architecture with periductal fibrosis and dense mixed inflammatory cell infiltration composed of lymphocytes plasma cells and eosinophils (Figure 1). Focal acinar destruction by mononuclear cells was seen. An impression of chronic sclerosing sialadenitis or Kuttner tumor was made. There were no lymphoid follicles that were well formed or squamous metaplasia. Also the periductal sclerosis and parenchymal loss was absent. So a Seifert's stage 2 was considered at this stage.

Sialadenitis is the inflammation of the salivary glands and CSS is a long standing inflammatory process of poorly understood etiology affecting unilateral or bilateral glands.² It frequently involves the submandibular gland and manifests as a painful swelling. This may be due to sialoliths or inspissated secretions but the exact mechanism is not fully understood.³ Owing to the presence of the lymphocytic inflammatory response an autoimmune basis has also been proposed. FNAC, though not done in the above case, provides great help by differentiation from malignant mimics.²

Submandibular sialadenectomy is believed to be the treatment of choice as it ensures complete removal of mass and provides histopathological confirmation as well as for Seifert staging of the disease based on the intensity of inflammation.⁴ In early stages there is maintained lobular architecture, dense periductal fibrosis, lymphoplasmacytic infiltration with or without follicle formation and presence of eosinophils, while in the later stages there is sclerosis with parenchymal atrophy.²

To sum up this case highlights how a long standing submandibular gland swelling histopathologically showing maintained lobular architecture, periductal fibrosis and lymphoplasmatic infiltrate with or without eosinophils help in diagnosis of CSS or Küttner’s tumor. These can simple pointers help in differentiating the same from a malignancy and eliminate the undue stress off the same.

REFERENCES

1. 1. Poghosyan A, Misakyan M, Sargsyan A, Khachatryan P, Hakobyan G. Chronic sclerosing sialadenitis (Küttner’s tumor) of the submandibular salivary gland: Our experience of one case report. Clin Case Rep. 2019;7:1600–4.
2. 2. Sahoo PK, Rout SS, Naidu AA, Reddy KU, Muddu VT. Kuttner’s tumour: chronic sclerosing sialadenitis- a mimicker of malignancy. Int Surg J. 2020;7:514–9.
3. 3. Lakshmanan S, Manimaran V, Valliappan V, Arumugam V. An unusual presentation of chronic sclerosing sialadenitis of submandibular gland (Kuttner’s tumour). Bio Med J Case Rep. 2019;12:e231189.
4. 4. Samara E, Mattine S. Kuttner’s tumor of the parotid gland: An unusual presentation. Adv Oral Maxillofac Surg. 2021;1:100007.