Elsevier

Oral Oncology

Volume 40, Issue 3, March 2004, Pages 348-352
Oral Oncology

CASE REPORT
Hyalinizing clear cell carcinoma of salivary gland: an aggressive variant

https://doi.org/10.1016/j.oraloncology.2003.08.023Get rights and content

Abstract

Although hyalinizing clear cell carcinoma (HCCC) had been previously illustrated by several authors, it was not until 1994 that this tumour was characterized by Milchgrub et al. [Am J Surg Pathol (1994),18,74] and separated from the heterogeneous group of clear cell carcinomas described in the literature. HCCC is a distinctive infiltrative low-grade, monomorphic, glycogen-rich clear cell carcinoma with prominent stromal hyalinization occurring most often in the minor salivary glands of adult women. A case of hyalinizing clear cell carcinoma arising in the tongue of an adult female is described with special reference to the presence of minor foci of mitotic activity, necrosis and anaplasia in this otherwise typical low-grade carcinoma. Widespread metastases and death within a year of initial presentation in this case suggests that there may be a subset of this indolent tumour in which these features are associated with a poor prognosis.

Section snippets

Case report

In December 2000, a 57 year-old female presented to the Ear Nose and Throat Department in St. James's Hospital, Dublin, Ireland, with a 4-month history of severe pain in her right ear and throat along with profound anaesthesia of the right side of her tongue. Coinciding with this history of pain, the patient had a rash affecting her ear and neck on that side, diagnosed as shingles. On examination, there was a large ulcerated mass at the base of her tongue with a palpable cervical lymph node on

Pathology

The excision contained a large tumour mass (3.5 cm in maximum diameter) occupying the body and posterior tongue. While an ulcerated elevated nodule was present, sectioning revealed that the bulk of tumour was submucosal. The cut surface of the tumour was firm and white with a whorled appearance. It extended to the deep margins of the resection and there was extensive nodal involvement by tumour with a total of seven positive nodes retrieved from levels I, II, III and IV of the right neck

Discussion

With increasing diagnostic experience, the class of “adenocarcinomas, not otherwise specified” in salivary glands is diminishing.2 In 1980, Batsakis3 was the first to use the term clear cell carcinoma of the salivary gland to describe what had previously been reported as a variant of ductal carcinoma or epithelial–myoepithelial carcinoma.4, 5 In 1994, the group was defined even further when a unique tumour with distinctive morphological features was identified by Milchgrub et al. and called a

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