Case Report Chronic Sclerosing Sialadenitis (Küttner’s tumour) of the Parotid Gland Güçlü Kaan BERIAT1, Sefik HalitA KMANSU1, Sinan KOCATÜRK1, Ömür ATAOGLU2 Submitted: 4 Dec 2009 1 Department of Otolaryngology, Faculty of Medicine, Ufuk University, No:86, Accepted: 18 Mar 2010 Konya Avenue, 06520 Balgat, Ankara, Turkey 2 Department of Pathology, Faculty of Medicine, Gazi University, No:43, Konya Avenue, 06100 Bahçelievler, Ankara, Turkey Abstract Chronic sclerosing sialadenitis is a chronic inflammatory salivary gland disease. Küttner reported 4 cases of submandibular gland lesions for the first time in 1896. Chronic sclerosing sialadenitis is a very rare inflammatory lesion of the parotid gland and cannot be easily distinguished from salivary malignant masses. We reported a 28-year-old male with a painful parotid tumour, which grew slowly for 4 years. Keywords: chronic illness, inflammation, oral surgery, parotid gland, sclerosis, sialadenitis Introduction Case Report A series of patients with unilateral, hard, A 28-year-old male was investigated at tumour-like masses of the submandibular the Otolaryngology Outpatient Department. gland were diagnosed with chronic sclerosing He first noted the mass 4 years earlier. Physical sialadenitis by Küttner in 1896 (1). This disease is examination revealed a tender, hard and fixed, clinically similar to salivary gland neoplasms and 3 x 2 cm mass at the angle of the left maxillary is classified as a tumour-like lesion of the salivary arch, and it seemed to be attached to underlying glands by the World Health Organization (2). structures. No other masses or adenopathy were Chronic sclerosing sialadenitis is clinically noted in the head or neck. No related events characterised by a firm, relatively painful swelling were present in the patient’s medical history. He of one of the submandibular glands. This disorder reported no other symptoms or complaints. His is characterised by plasmocytic and lymphocytic facial nerve function was intact. A sonographically periductal infiltrate eventually leading to guided fine-needle biopsy was performed. encasement of ducts with thick fibrous tissue (3). Cytology revealed only lymphocytes and other Histologically, chronic sclerosing blood elements; however, no glandular epithelial sialadenitis is characterised by periductal cells were present. Magnetic resonance imaging sclerosis, acinar atrophy, and infiltration of the detected a 3 x 2.5 x 2 cm, smooth-surfaced, gland by lymphocytes, which some studies have multilobular mass in the left parotid gland. This recognised as predominantly activated B cells mass had a heterogenic opaque appearance after with a subpopulation of helper-inducer T cells. intravenous contrast injection. No pathologic The distribution pattern of these lymphocytes nodes were identified (Figures 1 and 2). Left total suggested that the response was immunological. parotidectomy was conducted with mass excision However, sialoliths and mucous plugs were found and preservation of the facial nerve. in 29% to 83% of the lesions. This association The specimen collected for pathological was meaningful for some authors, such that it examination measured 2.5 cm at its longest was considered in favour of a cause and result diameter. It was a yellow-white in colour, and relationship. Other possible aetiologies of chronic its cut surface demonstrated a firm consistency. sclerosing sialadenitis are ascending bacterial The whole tissue sample was submitted for infections of the oral cavity and duct obstruction pathological examination. Pathology revealed by foreign bodies. chronic sclerosing sialadenitis. The microscopic examination revealed a collagenised fibrous tissue in all sections. Residual salivary gland tissue seemed to be embedded in the fibrous tissue in Malaysian J Med Sci. Oct-Dec 2010; 17(4 ): 57-61 www.mjms.usm.my © Penerbit Universiti Sains Malaysia, 2010 For permission, please email:[email protected] 57 Malaysian J Med Sci. Oct-Dec 2010; 17(4): 57-61 Figure 1: Magnetic resonance image (coronal view) Figure 2: Magnetic resonance image (transverse of the head showed chronic sclerosing view) of the head showed chronic sclerosing sialadenitis located on the zygomatic arch sialadenitis located within the left cheek (white arrows). (white arrows). some areas (Figure 3). In the salivary gland tissue, metaplasia with proliferation of ductal mononuclear cells, mostly lymphocytes, were also epithelium. Periductal lymphoid observed (Figure 4). Mononuclear cells were also follicles are well developed. There is observed in the fibrous stroma. No evidence of fibrosis in the centres of the lobules malignancy was observed, and the final diagnosis accompanied by atrophy of acini. was chronic sclerosing sialadenitis. Post- Stage 3.Even more prominent lymphocytic operative complications and recurrence were not infiltration with lymphoid follicle encountered in the 2-year follow-up. formation, parenchymal atrophy, periductal hyalinisation, and sclerosis as well as quamous and goblet cell Discussion metaplasia in the ductal system. Stage 4.Cirrhosis-like with marked parenchymal Chronic sclerosing sialadenitis is an loss and sclerosis (the “burnt-out” inflammatory process that primarily affects the phase). submandibular gland and presents clinically as a painful swelling. Histologically, chronic sclerosing The origin of chronic sclerosing sialadenitis sialadenitis demonstrates a loss in acinar tissue, has been attributed to many etiologic agents. dense fibrosis, which is mainly periductal, and Sialoliths and mucous plugs are found in 29% lymphocytic infiltration with lymphoid follicle to 83% of cases, but it is not clear whether formation in some cases (4). the sialoliths are the causes or results of the According to Seifert (5), chronic sclerosing inflammatory process (2,6,7). In this case, no sialadenitis may progress through 4 different sialoliths were found. Anything that causes an histological stages: obstruction of salivary flow or stasis of secretions Stage 1.Focal chronic inflammation with can lead to acinar cell swelling, necrosis, ductal aggregates of lymphocytes around dilation, and retention of salivary secretions moderately dilated salivary ducts accompanying oedema and inflammatory cell containing infiltration (8). Salivary gland stones may cause inspissated secretion is present. obstruction of salivary flow or stasis of secretions. Stage 2.Diffuse lymphocytic infiltration and Seifert and Donath (5) proposed a theory of severe periductal fibrosis are more obstructive electrolyte sialadenitis in which a marked. The ductal system shows secretion disorder produces inspissated secretion inspissated secretion and focal that obstructs the small ducts, which leads to 58 www.mjms.usm.my Case Report | Chronic sclerosing sialadenitis Figure 3: Histology of the salivary gland tissue Figure 4: Histology showed the salivary gland residue showed chronic sclerosing sialadenitis embedded in collagenised fibrous tissue. On (Küttner’s tumor) with only a few residual the upper right, a focus of mononuclear cells ducts persisting, marked fibrosis and foci of is also shown. (Haematoxylin and eosin, lymphocytic infiltrate often with germinal 100x magnification) centers. (Hematoxylin and eosin, 200x magnification) inflammation, fibrosis, parenchymal atrophy, arthritis, which is consistent with immune and an immune reaction of the duct system. pathogenesis (11). Immunohistochemical studies of the Usually, these cases do not demonstrate lymphoid population in the chronic sclerosing symptoms related to sialadenitis. However, sialadenitis revealed abundant cytotoxic T cells sialolithiasis is the most common cause especially near ducts and acini. The B cell reaction of sialadenitis in symptomatic patients. was less pronounced and largely restricted Histologically, various degrees of atrophy, to lymph follicles. There was an intimate fibrosis, and chronic inflammation are seen, relationship between the T-cell-dominated but lymphoplasmocytic periductal infiltrate and inflammatory infiltrate and acinar and duct cells. fibrous encasement of ducts, which are typical of The monoclonal and oligoclonal populations chronic sclerosing sialadenitis, are not present. of cytotoxic T cells and their histopathological The causes of granulomatous sialadenitis range behaviours suggested that chronic sclerosing from infections to duct obstruction caused by sialadenitis may be the result of an immune calculi or malignancies. Granulomas of different process triggered by intraductal epithelial agents. types can be present, and a xanthogranulomatous The lymphocytes that infiltrate the epithelial variant has been described (12). component of chronic sclerosing sialadenitis are Necrotising sialometaplasia occurs mainly mainly B cells and are often characterised by a in the minor salivary glands of the palate and lack of Bcl-2 expression (9,10). may be confused with carcinoma. In necrotising The duration of symptoms before the sialometaplasia, an ischemic aetiology is patient seeks treatment is variable, from 1 month thought to produce ulcerating lesions with to about 3 decades, and the induration and partial necrosis of glands associated with enlargement often lead clinicians to diagnose regeneration and squamous metaplasia of chronic sclerosing sialadenitis as a salivary gland the adjacent ducts. Its localisation and lack of neoplasm (7). Possible differential diagnoses of lymphoplasmocytic infiltration differentiates this entity include other benign inflammatory necrotising sialometaplasia