Cribriform Adenocarcinoma of Minor Salivary Gland Origin Principally Affecting the Tongue

Abstract

We present a series of 23 cases of a distinctive, hitherto poorly recognized low-grade adenocarcinoma, with several histologic features reminiscent of papillary carcinoma of the thyroid, and which mostly but not exclusively occurs in the tongue. All the tumors were unencapsulated and were divided into lobules that were composed mainly of cribriform and solid growth patterns. Therefore, we propose the name "cribriform adenocarcinoma of minor salivary gland origin (CAMSG)." All the patients were adults with a mean age at diagnosis of 55.8 years (range, 25 to 85 y). Fourteen of the 23 tumors were localized in the tongue, 3 in the soft palate, 2 in the retromolar buccal mucosa, 3 in the lingual tonsils, and 1 in the upper lip. Fifteen patients of 23 had synchronous metastases in the cervical lymph nodes at the time of diagnosis, bilateral in 3 cases. In 3 patients, the nodal metastasis was the first evidence of disease, later investigation revealing primary neoplasms in the base of tongue and tonsil, respectively. In addition, 1 patient developed a cervical lymph node metastasis 8 years after excision of a primary tumor of the tongue. Data on treatment and follow-up were available in 14 cases. The patients were treated by radical excision with clear margins (12 cases) or by simple excision (2 cases). Neck dissection was performed in 10 patients; 9 received radiotherapy, but none were treated by chemotherapy. Clinical follow-up ranged from 2 months to 13 years (mean, 6 y and 5 mo). Twelve patients are alive with no evidence of recurrent or metastatic disease after treatment, 1 patient died 2 years after surgery without evidence of tumor, and 1 patient is alive with recurrent tumor of the palate.

Update from the 4th Edition of the World Health Organization Classification of Head and Neck Tumours: Oropharynx

“…Cribriform adenocarcinoma of minor salivary glands (CAMSG) is a salivary gland carcinoma that often arises in the tongue and especially the tongue base [33,34]. CAMSG is similar and yet subtly different form PLGA.…”

Salivary Gland Tumor “Wishes” to Add to the Next WHO Tumor Classification: Sclerosing Polycystic Adenosis, Mammary Analogue Secretory Carcinoma, Cribriform Adenocarcinoma of the Tongue and Other Sites, and Mucinous Variant of Myoepithelioma

“…It was first described in 1999 as a unique tumor [15], although there are two earlier case reports of ''tubular adenocarcinoma'' of the tongue [16] and of ''papillary adenocarcinoma'' of the tongue [17] that are likely this entity. Since this initial report there has been one additional case report [18] and the largest series of these lesions to date (23 cases), was recently published [19]. The most common site of presentation is the tongue (74 %) with 9 % each involving the palate and tonsil area; 8 % arise in other intraoral sites.…”

Epithelial Salivary Gland Tumors in Two Distant Geographical Locations, Finland (Helsinki and Oulu) and Israel (Tel Aviv): A 10-Year Retrospective Comparative Study of 2,218 Cases

“…The histopathologic diversity of these tumors is not proportionate to their relatively low frequency. This is reflected by the continuously changing classifications that result from periodic re-definition of the nature of the tumors (e.g., acinic cell carcinoma and mucoepidermoid carcinoma [3]), the divergence of new histopathologic subtypes (e.g., oncocytic, oncocytic-sebaceous, apocrine, double clear subtypes of epithelial-myoepithelial carcinoma [4] and others), and the recognition of potentially new entities (e.g., mammary apocrine secreting carcinoma [5], cribriform carcinoma of the tongue [6]). It can be expected that the current classification will be further modified to adjust to updated clinicopathologic correlations as more clinical data accumulate and our molecular techniques improve.…”