Abstract

Low-grade cribriform cystadenocarcinoma (LGCCC) is a malignant salivary gland tumor. LGCCC occurs rarely, with the parotid gland being the most commonly afflicted site. Few cases arise in other sites. The present study reports a case of LGCCC that occurred at the palatal gland of the hard palate. A 56-year-old female was referred to Saitama Medical University Hospital (Moroyama, Saitama, Japan) due to an intraoral mass. Since cytological examination and biopsy led to a diagnosis favoring a neoplasm, but with uncertain malignant potential, the tumor was resected with a safe surgical margin. The specimen was thoroughly examined. Microscopically, there was a well-demarcated, unilocular cyst with the lumen lined by tumor cells. The tumor cells were arranged in tubular, cribriform and solid structures in the area of the intracystic mass lesions. Nuclear atypia was inconspicuous, although mitotic figures were observed throughout the tumor. Neither local nor perineural invasion was present. On immunohistochemistry, the tumor cells were diffusely positive for S-100 protein. Myoepithelial markers, calponin and p63, highlighted the cells rimming the cystic mass. The final histopathological diagnosis was of LGCCC. The tumor was completely resected. At 1 year post-resection, the patient exhibited no recurrence or distant metastasis. LGCCC is regarded as clinically indolent. However, there is little literature available to aid with prognosis prediction due to the rarity of LGCCC cases. Thus, greater experience and longer follow-up periods are necessary to find the optimal/curative treatment for patients with LGCCC and to clarify the pathophysiology.

Highlights

  • Salivary gland tumors produce a variety of histological patterns making tumor classification difficult [1].Low‐grade cribriform cystadenocarcinoma (LGCCC) is a malignant salivary gland originally reported as a variant of salivary duct carcinoma [2]

  • The 2005 World Health Organization classification considers this neoplasm to be a variant of cystadenocarcinoma, mainly due to its cystic morphology, since no definite association has been found between salivary duct carcinoma and LGCCC [1]

  • The present study reports a case of LGCCC that occurred at the palatal gland of the hard palate

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Summary

Introduction

Salivary gland tumors produce a variety of histological patterns making tumor classification difficult [1]. The 2005 World Health Organization classification considers this neoplasm to be a variant of cystadenocarcinoma, mainly due to its cystic morphology, since no definite association has been found between salivary duct carcinoma and LGCCC [1]. In this classification system, LGCCC is defined by its histological similarity to breast atypical ductal hyperplasia or low‐grade ductal carcinoma in situ [1]. Magnetic resonance imaging showed a high‐intensity cystic mass with fluid internally on T1‐weighted imaging of the right palatal region (Fig. 1B). The tumor was completely resected, and 1 year later, the patient exhibited no recurrence or distant metastasis

Discussion
Brandwein‐Gensler MS and Gnepp DR
12. Weinreb I
Findings
19. Regezi JA and Batsakis JG
Full Text
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