Kuttner's Tumour: Chronic Sclerosing Sialadenitis- a Mimicker of Malignancy
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International Surgery Journal Sahoo PK et al. Int Surg J. 2020 Feb;7(2):514-519 http://www.ijsurgery.com pISSN 2349-3305 | eISSN 2349-2902 DOI: http://dx.doi.org/10.18203/2349-2902.isj20200307 Original Research Article Kuttner’s tumour: chronic sclerosing sialadenitis- a mimicker of malignancy Prakash Kumar Sahoo, Suman Saurav Rout*, A. Apalla Naidu, K. Ujwal Reddy, Vishnu Teja Muddu Department of Surgery, Institute of Medical sciences and SUM Hospital, Bhubaneswar, Odisha, India Received: 06 August 2019 Revised: 30 December 2019 Accepted: 02 January 2020 *Correspondence: Dr. Suman Saurav Rout, E-mail: [email protected] Copyright: © the author(s), publisher and licensee Medip Academy. This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License, which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. ABSTRACT Background: Kuttner’s tumour is a condition of the submandibular gland which is underreported as a distinct entity. Also called as the chronic sclerosing sialadenitis it resembles a plasmocytic and lymphocytic inflammatory process and presents as a hard and painful mass which clinically mimics a malignancy and raises significant concerns. The objective of the present study was to evaluate cases of Kuttner’s tumor and discuss its clinical and pathological aspects to distinguish it from a definite malignancy and create a grown acceptance of the presence of such an entity in our setting. Methods: We collected 170 cases of submandibular swellings and evaluated in detail the clinical and pathological aspects of eight cases out of them which were later diagnosed to as Kuttner’s tumour. Results: The age of the patients varied between 23 to 61 years (mean age 42.5 years) with 3 males and 5 females. 6 patients reported with a firm to hard painless submandibular mass (5 left sided; 3 right sided) while 2 patients experienced intermittent discomfort. The mean duration of presentation of symptoms was 5.3 months. Fine needle aspiration cytology was done in 6 cases preoperatively. Following submandibular sialadenectomy histopathology showed salivary gland tissue with preserved lobular architecture, but with marked fibrosis, acinar atrophy, and a dense lymphoplasmacytic infiltrates. Conclusions: Kuttner’s tumor may be pre operatively distinguished from a malignancy with improved imaging and a good image guided FNA Cytology saving the clinician and the patient from a lot of dilemma. However, histopathology and immunohistochemistry would be the key in establishing the diagnosis. Keywords: Kuttner's, Chronic sclerosing sialadenitis, Submandibular tumour INTRODUCTION inflammatory lesions of the salivary glands more than anything else. This lesion has been recognised as a Kuttner first defined chronic sclerosing sialadenitis (CSS) distinct clinicopathological entity in the latest edition of of submandibular gland when he described 4 of such World Health Organisation classification of salivary cases of chronic inflammatory salivary gland disease in gland tumors.2 CSS is clinically characterised by a firm, 1896.1 occasionally painful mass of the submandibular glands. Fine needle aspiration cytology (FNAC) being a simple Difficult to distinguish these lesions from malignancies and effective modality to work up salivary gland masses, of salivary glands clinically and dreaded, such masses its efficacy in diagnosing this particular entity is still 3-5 have been now been described and proven to be as questionable. Plasmocytic and lymphocytic periductal International Surgery Journal | February 2020 | Vol 7 | Issue 2 Page 514 Sahoo PK et al. Int Surg J. 2020 Feb;7(2):514-519 infiltrate which eventually lead to the encasement of METHODS ducts with thick fibrous tissue of the salivary glands are the characteristic features of Kuttner’s tumor. This clinicopathological study entails 8 cases of Kuttner’s tumor, which were treated at Institute of Medical Although described more than a century ago, this clinical Sciences and SUM Hospital, Bhubaneswar, Odisha, entity is still under-reported and preoperatively it remains India, a tertiary care hospital, during the period from July unrecognised by many surgeons. The submandibular 2014 to January 2019. We evaluated and followed up 170 masses are often removed following a preliminary cases which presented with submandibular masses with diagnosis of carcinoma, thus inciting substantial worry to or without pain. All the cases were clinically assessed, both patients and clinicians. This state of affairs is preliminary cytology done and subjected to surgery attributed to the under-reporting of Kuttner’s tumor in which was then followed by a histopathology. Out of literature. We report here a series of cases of CSS and these, 8 patients were diagnosed as CSS (Kuttner’s discuss its clinical and pathological aspects to create a tumor) histopathologically and thus were included in the clearer picture and a grown acceptance of the presence of present study (Table 1). Due consent was taken from each such an entity in our setting. of the above patient before any investigations, surgery and inclusion in the above study. The statistical analysis Objectives was done using SPSS. The objectives of the present study were to evaluate cases RESULTS of Kuttner’s tumor and discuss its clinical and pathological aspects to distinguish it from a definite The incidence of Kuttner’s tumor in our setting was malignancy and a grown acceptance of the presence of <0.05% of all submandibular masses encountered during such an entity in our setting. the above time period (i.e., exactly 0.04%; 8 out of 170) (Figure 1 and 2). Figure 1: Epidemiology of firm painless, submandibular masses. 70 60 50 40 30 number of pts number 20 10 0 male female benign 46 62 malignant 34 20 kuttner's tumor 3 5 Figure 2: Incidence of various submandibular pathologies. International Surgery Journal | February 2020 | Vol 7 | Issue 2 Page 515 Sahoo PK et al. Int Surg J. 2020 Feb;7(2):514-519 The age of the patients varied between 23 to 61 years Six patients reported with a firm to hard painless (mean age 41.5 years) with 3 males and 5 females. The submandibular mass (5 left sided; 3 right sided) while 2 age distribution is shown in Figure 3 with a 50% patients experienced intermittent discomfort. The mean incidence in 5th decade of life. duration of presentation of symptoms was 5.3 months. Table 1: Clinical details of patients with Kuttner’s tumor (CSS). Case Age Side Clinical manifestations FNAC findings Calculi no. /sex affected Scanty acini and normal-looking Painless, firm, non-tender, Left sided ductal cells, with a variable 1 45/M Left Absent submandibular mass for 6 months degree of chronic inflammatory cell infiltration Recurrent occurring firm, slightly tender Epithelial proliferation; chronic 2 23/F with intermittent discomfort left sided Left inflammation Present submandibular mass for 8 months Painless, firm nontender right sided Variable degree of chronic 3 36/F Right Absent submandibular mass for 2 months inflammatory cell infiltration Painless, firm to hard, nontender left 4 50/F submandibular mass; intermittent Left Not done Present discomfort for 6 months Recurrent occurring soft to firm, non- Epithelial proliferation with 5 43/M tender left submandibular mass for 4 Left Absent chronic inflammation months Painless, firm to hard non tender right Lymphoproliferative lesion 6 44/M Right Absent submandibular mass for 1 year scanty acini and normal-looking Painless, firm non tender left ductal cells, with a variable 7 30/F Left Absent submandibular mass for 3 months degree of chronic inflammatory cell infiltration Painless, firm nontender right sided Epithelial proliferation; chronic 8 61/F Right Absent submandibular mass for 2 months inflammation FNAC was done in all 6 cases pre operatively (Figure 4 4 and 5). Only 1 patient on FNAC had features suggestive of a lympho-proliferative disease though malignancy was not 2 ruled out. 1 1 0 21-30 31-40 41-50 51-60 61-70 kuttner's tumor Figure 3: Age distribution of patients with Kuttner’s tumor. All the patients were subjected to ultrasonography of the submandibular region of both the sides. Calculi were detected in 2 cases along with dilated duct which were confirmed by submandibular sialography and computed Figure 4: FNAC the aspirate is moderately cellular tomography imaging (Figure 5). and shows ducts scattered in a background of inflammatory cells. International Surgery Journal | February 2020 | Vol 7 | Issue 2 Page 516 Sahoo PK et al. Int Surg J. 2020 Feb;7(2):514-519 Table 2: Fine needle aspiration findings. duct was duely identified and secured. The gross samples were firm, indurated, lobulate yellowish white. Variable degree of chronic inflammation with scanty Histopathology showed salivary gland tissue with acini (Figure 4). preserved lobular architecture including the lobular septa. Normal looking ductal cells (Figure 4). Marked periductal fibrosis was noted in all cases though Tubular ductal elements surrounding collagen fibres the extent varied in each case. Dense lymphoplasmocytic around Plasmolymphocytic cells (Figure 5). infiltrate (Figure 7) was noted both within and in between the lobules. There was acinar atrophy with residual ducts being encased by thick collagen bundles. Figure 5: FNAC numerous lymphoid cells admixed with plasma cells and histiocytes. Figure 7: Histopathology of the gland showing chronic plasmolymphocytic infiltrate with acinar atrophy and ductal sclerosis. DISCUSSION CSS is an inflammatory process that primarily affects the submandibular gland of middle aged adults and presents clinically as a painful swelling .The inflammatory lesions in Küttner's tumor may occur on one side (unilateral) or both sides (bilateral), predominantly involving the submandibular gland, but is also known to occur in other major and minor salivary glands including the parotid gland.6-9 Salivary tumors especially those of submandibular glands show a great deal of morphological diversity, as well as variations in the nature of the lesion (malignant vs benign) 35-40% of submandibular tumors.