Chronic sclerosing sialadenitis (Kuttner's tumor): Unusual presentation with bilateral involvement of major and minor salivary glands

Blanco, Mario; Mesko, Thomas W.; Cura, Marco; Cabello-Inchausti, Beria

doi:10.1053/adpa.2003.50004

Cited by 41 publications

(27 citation statements)

References 5 publications

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“…Salivary flow rate is usually normal or slightly decreased [8] . CSS typically affects the submandibular gland; however, involvement of the parotid, the sublingual gland and the minor salivary glands has also been reported [3,11,12] . To the best of our knowledge, CSS affecting the sublingual gland is exceedingly rare [12] .…”

Section: Discussionmentioning

confidence: 99%

Chronic sclerosing sialadenitis of the sublingual gland: case report and literature review

Thomopoulos¹,

Nikitakis²,

Daskalopoulos³

et al. 2018

SDS

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Chronic sclerosing sialadenitis (CSS) is a chronic inflammatory condition of uncertain etiology, primarily affecting the submandibular glands. It has recently been proposed that CSS belongs to the immunoglobulin G4 (IgG4)-related disease spectrum, although the exact relationship between these two entities requires further elucidation. Our aim is to present a case of bilateral CSS of the sublingual glands and discuss the possible underlying pathogenetic mechanisms. A 66-year-old woman presented with a 7-month history of an asymptomatic firm swelling of the floor of the mouth. Clinically, a diffuse, multinodular swelling of elastic to firm consistency was observed across the sublingual folds bilaterally. An incisional biopsy revealed a lymphoplasmacytic infiltrate with lymphoid follicle formation, along with acinar degeneration, fibrosclerosis and ductal dilation. Immunohistochemical investigation ruled out lymphoproliferative diseases revealing a polyclonal lymphoplasmacytic population with only a few scattered IgG4-positive plasma cells. IgG4 serum levels were also normal. No treatment was administered with progressive spontaneous resolution of the swelling and no evidence of recurrence at 2 years follow-up. CSS is a benign salivary gland disorder, which, at least in a subset of cases, may represent an IgG4-associated disorder. Therefore, immunohistochemical evaluation of the IgG4/IgG plasma cell ratio and the IgG4 serum levels is a recommended investigation in the diagnostic work-up of CSS cases.

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Section: Discussionmentioning

confidence: 99%

Chronic sclerosing sialadenitis of the sublingual gland: case report and literature review

Thomopoulos¹,

Nikitakis²,

Daskalopoulos³

et al. 2018

SDS

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“…The effects of microliths (sialoliths), infectious agents, secretory dysfunctions, duct abnormalities, and immune processes have been considered (5,6,7,10).…”

Section: Discussionmentioning

confidence: 99%

“…Unusual cases affecting both parotid glands, minor salivary glands, lacrimal glands as well as multiglandular involvement were also reported (1,3,4,6,7,13,14).…”

Section: Diagnosismentioning

confidence: 99%

“…Squamous and goblet cell metaplasia may be seen in the ductal system. Stage 4 (end stage): Shows cirrhosis-like stroma with marked parenchymal loss and sclerosis (7,13).…”

Section: Diagnosismentioning

confidence: 99%

“…Possible differential diagnosis of this entity includes other benign infl ammatory lesions of salivary glands, benign lymphoepitelial lesions, extranodal marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue, sclerosing lymphoma, neoplasms of the salivary glands and radiation effects (4,6,7,8,10).…”

Section: Diagnosismentioning

confidence: 99%

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Kuttner tumor

Uhliarova¹,

Svec²

2012

BLL

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Abstract:Objective: Kuttner tumor or chronic sclerosing sialadenitis is a benign infl ammatory condition of the salivary gland. Clinically, it produces a fi rm swelling of the gland and may be diffi cult to distinguish from neoplasia. This is an under-recognized entity in the surgical pathology and cytology literature. We describe our experience with Kuttner tumor. Methods: Retrospectively, we analyzed a group of 7 patients with chronic sclerosing sialadenitis treated from January 1999 to March 2010 at the Department of Otorhinolaryngology, FD Roosevelt Faculty Hospital in Banska Bystrica, Slovakia. The authors evaluated age and sex distribution, patient's history, diagnostic procedures, extent and success of surgical treatment and postoperative complications. Results: Four patients with chronic sclerosing sialadenitis were male and 3 were female. The mean age of patients was 55 years (ranging from 43 to 70 years). Submandibular gland was affected in 6 cases (85.7 %), involvement of both parotid glands was found in one patient (14.3 %). All patients in our study group were treated surgically. In 6 cases submandibular gland extirpation was performed via standard transcervical approach. Total parotidectomy with facial nerve preservation was realized in one patient with bilateral involvement of parotid glands. Conclusion: Chronic sclerosing sialadenitis is a condition that is perhaps more common than thought but usually mis-recognised as it is only after excision of the gland that the correct diagnosis is made (Tab. 1, Ref. 17 Kuttner tumor, also known as chronic sclerosing sialadenitis or cirrhosis of the salivary gland is a fi broinfl ammatory disease of the salivary glands, characteristically of the submandibular gland. Chronic sclerosing sialadenitis belongs to the spectrum of IgG4-related diseases (1,11,16).Clinically, it produces a fi rm swelling of the glands and may be diffi cult to distinguish from neoplasia (10). It is characterized histologically by periductal fi brosis, dense lymphocytic infi ltration with lymphoid follicle formation, loss of the acini, and eventually, marked sclerosis of the salivary gland (2). Excision of the mass, usually carried out diagnostically, is anadequate treatment (10).We report our experience of Kuttner tumor. Etiopathogenesis, clinical symptoms, diagnostic procedures and treatment are discussed. MethodsThis retrospective study analyzed a group of 152 patients with salivary gland disorder treated surgically at the Department of Otorhinolaryngology, Faculty Hospital of FD Roosevelt in Banska Bystrica, Slovakia, within the period from January 1999 to March 2010. Among these, 7 patients (4.6 %) were confi rmed to be Kuttner tumor, based on the clinicopathological testing, and represented our study group. The authors evaluated age and sex distribution, patient's history, diagnostic procedures, extent and success of surgical treatment and postoperative complications. ResultsSeven cases of chronic sclerosing sialadenitis were identifi ed. There were 4 males (57.1 %) and 3 females...

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Radiology Quiz Case 1: Diagnosis

2010

Arch Otolaryngol Head Neck Surg

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Chronic sclerosing sialadenitis (Kuttner's tumor): Unusual presentation with bilateral involvement of major and minor salivary glands

Cited by 41 publications

References 5 publications

Chronic sclerosing sialadenitis of the sublingual gland: case report and literature review

Chronic sclerosing sialadenitis of the sublingual gland: case report and literature review

Kuttner tumor

Radiology Quiz Case 1: Diagnosis

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