Bilateral Carcinoma in Situ of Wharton's Duct after Chronic Obstructive Sialadenitis: Inflammation as the Cause of Malignancy?

Abstract

A case of bilateral carcinoma in situ of Wharton's duct after chronic sialadenitis is reported. The patient, a 54-year-old man, complained of recurrent pain and swelling in the left lower submandibular region. Computed tomography showed large stones in the hilar area of both submandibular glands. The patient underwent bilateral submandibular excision. Histologic and immunohistochemical examination revealed squamous metaplasia with areas of carcinoma in situ in both right and left ducts adjacent to the calculus. To the best of our knowledge, this is the first case report in the literature describing an association between obstructive sialadenitis and carcinoma in situ of Wharton's duct. We discuss etiologic factors and chronic inflammation as a possible cause of malignancy.

Keywords

obstructive sialadenitis squamous cell carcinoma submandibular gland Wharton's duct

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References

Goforth

. Carcinoma developing in the parotid (Stensen's) duct. Am J Med Sci 1927;173:624–9.

Lyall

Golomb

. Carcinoma of Stenson's duct: A case report. Ann Surg 1954;139:364–7.

Peracchio

. Primary squamouscell carcinoma of Stensen's duct. Oral Surg Oral Med Oral Pathol 1958;11:123–7.

Vigorita

Huvos

Gerold

. Squamous-cell carcinoma of Stensen's duct. Head Neck Surg 1980;2:513–7.

Owens

Fligiel

Ward

. Primary carcinoma of Stensen's duct. Otolaryngol Head Neck Surg 1982;90:671–3.

Steiner

Gould

Miller

Johnson

. Malignant tumors of Stensen's duct. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;87:73–7.

Maisel

Pearce

Connolly

Pearce

. Carbon-black carcinoma of Stensen's duct; report of a case. AMA Arch Surg 1959;78:331–9.

Tominaga

Uchino

Ishimaru

Inokuchi

Sato

Kudo

. Squamous cell carcinoma arising from Stensen's duct. Radiat Med 2006;24:639–42.

Wakoh

Imoto

Otonari-Yamamoto

. Image interpretation for squamous cell carcinoma of Stensen duct. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;106:e27–e32.

10.

Kim

Klein

Glastonbury

Eisele

. Primary squamous cell carcinoma of Stensen's duct in a patient with HIV: The role of magnetic resonance imaging and fine-needle aspiration. Head Neck 2009;31:278–82.

11.

Ellis

Auclair

. Tumors of the salivary glands. In: Ellis

Auclair

, eds. Atlas of tumor pathology (3rd series, Fasc. 17). Washington, DC: Armed Forces Institute of Pathology, 1996:251–7.

12.

Preuss

Klussmann

Wittekindt

Drebber

Beutner

Guntinas-Lichius

. Submandibular gland excision: 15 years of experience. J Oral Maxillofac Surg 2007;65:953–7.

13.

Triolo

. Nineteenth century foundations of cancer research advances in tumor pathology, nomenclature, and theories of oncogenesis. Cancer Res 1965;25:75–106.

14.

Kawanishi

Hiraku

Pinlaor

. Oxidative and nitrative DNA damage in animals and patients with inflammatory diseases in relation to inflammation-related carcinogenesis. Biol Chem 2006;387:365–72.

15.

O'Byrne

Dalgleish

. Chronic immune activation and inflammation as the cause of malignancy. Br J Cancer 2001;85:473–83.

16.

Angiero

Berenzi

Benetti

. Expression of p16, p53 and Ki-67 proteins in the progression of epithelial dysplasia of the oral cavity. Anticancer Res 2008;28:2535–9.

17.

Takeda

Sugihara

Hirayama

Hirano

Tanuma

Semba

. Immunohistological evaluation of Ki-67, p63, CK19 and p53 expression in oral epithelial dysplasias. J Oral Pathol Med 2006;35:369–75.