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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 1  |  Page : 89-91

Kuttner tumor involving minor salivary glands: A very rare case report


1 Departments of Oral Medicine and Maxillofacial Radiology, Mamata Dental College, Khammam, Telangana, India
2 Departments of Oral and Maxillofacial Pathology, Mamata Dental College, Khammam, Telangana, India

Date of Submission28-May-2014
Date of Acceptance04-Jul-2014
Date of Web Publication26-Sep-2014

Correspondence Address:
Moni Thakur
Department of Oral and Maxillofacial Pathology, Mamata Dental College, Giriprasad Nagar, Khammam - 507 002, Telangana
India
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Source of Support: Department of Oral and Maxillofacial Radiology and Department of Oral and Maxillofacial Pathology, Conflict of Interest: None


DOI: 10.4103/0972-1363.141869

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   Abstract 

Kuttner tumor, also known as chronic sclerosing sialadenitis, was first described by Dr. H. Kuttner in 1896. It is an uncommon, chronic, benign tumor-like lesion predominantly involving the submandibular glands of middle-aged individuals, and presents as a firm, painful swelling. Histologically, it is characterized by progressive periductal sclerosis, acinar atrophy, and gland infiltration by lymphocytes. A case of Kuttner tumor involving the minor salivary glands is reported.

Keywords: Chronic sclerosing sialadenitis, Kuttner tumor, minor salivary glands


How to cite this article:
Reddy Kundoor VK, Thakur M, Guttikonda VR, Thummala R. Kuttner tumor involving minor salivary glands: A very rare case report . J Indian Acad Oral Med Radiol 2014;26:89-91

How to cite this URL:
Reddy Kundoor VK, Thakur M, Guttikonda VR, Thummala R. Kuttner tumor involving minor salivary glands: A very rare case report . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2020 May 26];26:89-91. Available from: http://www.jiaomr.in/text.asp?2014/26/1/89/141869


   Introduction Top


Kuttner tumor (KT) was first described in 1896 by Dr. H. Kuttner. [1],[2],[3] It is also known as chronic sclerosing sialadenitis, which is an uncommon, chronic benign tumor-like lesion predominantly involving the submandibular glands [3],[4],[5] of middle-aged adults as a firm, painful swelling. [1] The lesion has been recognized as a distinct clinicopathologic entity in the latest edition of World Health Organization classification of tumor-like lesions of the salivary glands. [3],[4],[6] Histologically, KT is characterized by progressive periductal sclerosis, acinar atrophy, and gland infiltration by lymphocytes. [1],[2],[4],[7] Chronic sclerosing sialadenitis is a condition that is perhaps more common than thought, but usually misrecognized as it is only after excision of the gland that the correct diagnosis is made. [8] Recent research has shown that in KT, T-lymphocyte immune reaction predominates. [1],[9] We present a case of a female patient with KT of minor salivary glands arising in the upper lip, in order to raise awareness of this interesting and not well-known entity.


   Case Report Top


A 20-year-old female patient presented with a swelling on her upper lip since 6 months. The swelling was small in size initially and gradually increased in size to attain the present size. Her medical history was non-contributory, with no history of trauma to the area and no dryness of the mouth. Extraoral examination revealed a solitary swelling present on the upper lip on the right side, extending medio-laterally from the midline of the upper lip to the corner of the mouth and superio-inferiorly from below the nasolabial fold to the vermilion border of the upper lip, and measured 2 × 2.5 cm. It was firm, well defined, oval, mobile, and tender [Figure 1]. Intraorally the swelling involved the labial mucosa of the upper lip on the right side, extending medio-laterally from the frenum to the corner of the mouth and superio-inferiorly from the labial vestibule to the border of the upper lip, and measured 1.5 × 2.5 cm. Pulp vitality was checked in relation to 11, 12, and 13, which were found to be vital. Aspiration of the swelling yielded a straw-colored cystic fluid suggestive of a soft tissue cyst. Ultrasonography revealed a cystic lesion [Figure 2] and a provisional clinical diagnosis of nasolabial cyst was considered.
Figure 1: Extraoral photograph showing a swelling on the right side of upper lip

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Surgical excision of the lesion was done under general anesthesia including adjacent normal mucosa and the specimen was submitted for histopathologic examination. Histopathology revealed acinar atrophy, fibrosis, and dense inflammatory cell infiltrate suggesting chronic sclerosing sialadenitis (KT) [Figure 3]. Further, an immunohistochemistry study with CD20 was performed to confirm the lymphocytic infiltration that tested positive [Figure 4].
Figure 2: Ultrasonography showing cystic lesion

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Figure 3: Histopathology showing acinar atrophy, fibrosis, and dense inflammatory cell infiltrate

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Figure 4: Immunohistochemistry with CD20 showing lymphocytic infiltration

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The patient was kept under periodic observation and no recurrence was noted after 1 month follow-up [Figure 5].
Figure 5: Postoperative photograph after 1 month of follow-up

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   Discussion Top


KT, first described by Kuttner more than a century ago, [4] is an under-recognized entity and still a rare lesion with only a few publications in the literature. [3] The etiology of KT is still unknown. One of the reasons could be the presence of sialoliths causing ductal obstruction accounting for the changes. [1],[2] The effects of infectious agents, secretory dysfunctions, ductal abnormalities, and immune processes have also been described, and seem to be plausible explanations for the development of tumor in these glands. [2] The clinical presentation of KT varies from an asymptomatic swelling to recurrent pain, and some authors state that KT only affects the submandibular gland. Nevertheless, unusual cases affecting the parotid gland, simultaneously affecting parotid and submandibular glands, and simultaneously affecting both parotids, both submandibular, as well as minor salivary glands were recently reported. A case that bilaterally affected the submandibular and lacrimal glands was also described. [1] This may be the fourth case of KT related to minor salivary glands. [1],[2],[9] The reported ages of patients range from 13 to 81 years, with a mean age of 44 years at surgery. [5] The patient's age in the case reported was 20 years. According to Seifert, KT may evolve through four different histological stages as follows:

Stage 1: Focal chronic inflammation with nests of lymphocytes around the salivary ducts, which are moderately dilated and contain inspissated secretion.

Stage 2: More marked diffuse lymphocytic infiltration and more severe periductal fibrosis. The ductal system shows inspissated secretion and focal metaplasia with proliferation of ductal epithelium. Periductal lymphoid follicles are well developed. There is fibrosis in the center of the lobules, accompanied by atrophy of acini.

Stage 3: More prominent lymphocytic infiltration, with lymphoid follicle formation, parenchymal atrophy, periductal hyalinization, and sclerosis with squamous and goblet cell metaplasia in the ductal system.

Stage 4 (end-stage): Cirrhosis-like, with marked parenchymal loss and sclerosis (the "burnt-out" phase). [7]

Our case corresponded to stage 2 KT of Seifert's [7] histological staging. Differential diagnoses of KT include simple chronic sialadenitis, granulomatous sialadenitis, necrotizing sialometaplasia, sialolithiasis, inflammatory pseudotumor, benign lymphoepithelial lesions, and radiation effects. [1],[3] Other differential diagnoses for swelling of the lip include mucocele, lipoma, salivary gland tumors, minor salivary gland calculi, and nasolabial cyst. [10] Immunohistochemistry studies for CD20, CD45RO, and CD8 were positive for lymphocytes. [1] Other studies showed abundance of CD3, CD4, and CD8 positive T cells. [7] In the present case, immunohistochemistry study with CD20 was performed to confirm the lymphocytic infiltration and it tested positive for lymphocytes. Management can be conservative by adopting a "watch and wait" approach in the case where the mass is otherwise asymptomatic. In symptomatic cases, the mass is surgically excised. Prognosis is very good as these are benign lesions that do not tend to recur. [8]


   Conclusion Top


KT is a rare disease which mimics malignant neoplasm. [1] Hence, it should be considered in the differential diagnosis of swellings in the submandibular region and lip. To the best of our knowledge, this appears to be the fourth case report of KT in minor salivary glands and the first in Indian literature.

 
   References Top

1.Jham BC, Freire AR, Silveira-Júnior JB, Aguiar MC, Mesquita RA. Kuttner tumor involving minor salivary glands in a patient undergoing radiotherapy in the head and neck. Oral Oncol Extra 2006;42:181-3.  Back to cited text no. 1
    
2.Paul R, Shekar K, Singh M. Kuttner tumour: An unusual cause of enlargement of a minor salivary gland in the lip. Br J Oral Maxillofac Surg 2010;48:152-3.  Back to cited text no. 2
    
3.Agale SV, Momin YA, Agale VG. Kuttner tumour: A report of an underdiagnosed entity. J Assoc Physicians India 2010;58:694-5.  Back to cited text no. 3
    
4.Cheuk W, Chan JK. Kuttner tumor of the submandibular gland: Fine-needle aspiration cytologic findings of seven cases. Am J Clin Pathol 2002;117:103-8.  Back to cited text no. 4
    
5.Larbcharoensub N, Pongtippan A. Bilateral Kuttner tumor of submandibular glands; a case report and review of the literature. J Med Assoc Thai 2005;88:430-5.  Back to cited text no. 5
    
6.Seifert G. Tumour-like lesions of the salivary glands. The new WHO classification. Pathol Res Pract 1992;188:836-46.  Back to cited text no. 6
[PUBMED]    
7.Mochizuki Y, Omura K, Kayamori K, Sakamoto K, Shimamoto H, Yamaguchi A. Küttner's tumor of the sub-mandibular gland associated with fibrosclerosis and follicular hyperplasia of regional lymph nodes: A case report. J Med Case Rep 2011;5:121.  Back to cited text no. 7
    
8.Kiverniti E, Singh A, Clarke P. Küttner's tumour: An unusual cause of salivary gland enlargement. Hippokratia 2008;12:56-8.  Back to cited text no. 8
    
9.Blanco M, Mesko T, Cura M, Cabello-Inchausti B. Chronic sclerosing sialadenitis (Kuttner's tumour): Unusual presentation with bilateral involvement of major and minor salivary glands. Ann Diagn Pathol 2003;7:25-30.  Back to cited text no. 9
    
10.Wood NK, Goaz PW. Differential Diagnosis of Oral and Maxillofacial Lesions. 5 th ed. St. Louis: CV Mosby; 2007. p.572-5  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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