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Minor Salivary Gland Basal Cell Adenocarcinoma A Systematic Review and Report of a New Case

David W. Cuthbertson, MD1; Nikhila Raol, MD1; John Hicks, MD, DDS, PhD2; Linda Green, MD2,3; Robert Parke, MD1,3
[+] Author Affiliations
1Bobby R. Alford Department of Otolaryngology–Head and Neck Surgery, Baylor College of Medicine, Houston, Texas
2Department of Pathology and Immunology, Baylor College of Medicine, Houston, Texas
3Michael E. Debakey Department of Veteran’s Affairs Medical Center, Houston, Texas
JAMA Otolaryngol Head Neck Surg. 2015;141(3):276-283. doi:10.1001/jamaoto.2014.3344.
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Importance  Basal cell adenocarcinoma (BCAC) of the minor salivary gland is an extremely rare disease: the most recent substantive literature review reports only 25 cases. Owing to the rarity of this disease, it has not yet been well characterized in the literature.

Objective  We sought to expand the knowledge of minor salivary gland BCAC by performing an exhaustive literature review and adding to it a new case that is rare owing to the tumor’s size, aggressive nature, and mixed histologic pattern. The review emphasizes epidemiologic patterns, diagnostic characteristics, treatment patterns, and expected prognosis for minor salivary gland BCAC.

Evidence Acquisition  In June 2012, PubMed was queried using the term “salivary gland basal cell adenocarcinoma,” and the resultant articles were reviewed. Those specifically mentioning a minor salivary gland BCAC were included in this study. Those that did not differentiate minor salivary gland BCAC from major salivary gland BCAC were excluded. The search was not limited by language and included articles from North America, Europe, Africa, and Asia from 1978 to June 2012.

Results  The PubMed search resulted in 195 articles, of which 33 articles reported at least 1 case of minor salivary gland BCAC. We report herein 72 cases of minor salivary gland BCAC (71 cases from the literature review and 1 new case reported herein). The mean patient age at the time of presentation was 56 years (range, 24-90 years), and the disease showed no sex predilection. The most common location was the palate, and the average lesions size was 2.4 cm (range, 0.7-4.2 cm). The treatment modality of choice was wide local excision (n = 57; 79%). There was a high local recurrence rate (n = 30; 41%) but a low rate of distant metastasis (n = 8; 11%).

Conclusions and Relevance  We present a comprehensive review of minor salivary gland BCAC, describing nearly 3 times as many cases as has been previously reported. This review characterizes a rare disease and increases awareness of the disease among otolaryngologists. Minor salivary gland BCAC is similar to major salivary gland BCAC and minor salivary gland tumors in general with regard to patient age, sex, tumor site, treatment modality, recurrence, metastasis, and mortality.

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Figure 1.
Preoperative Maxillofacial Computed Tomographic Images With Contrast From the Present Case Report

A, Sagittal view shows the mass eroding through the hard palate (red arrowhead) and extending into pterygopalatine fossa (asterisk). B, Axial view shows the mass extending into the nasal cavity (plus sign) and pterygopalatine fossa (asterisk). In both panels, the tumor edges are marked with yellow arrowheads.

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Figure 2.
Hematoxylin-Eosin–Stained Specimens of Basal Cell Adenocarcinoma Involving the Palate From the Present Case Report

A and B, Tumor with tubular pattern composed of centrally placed large polygonal cells with a moderate amount of cytoplasm and peripherally placed smaller cells with modest amount of cytoplasm and dense nuclei (original magnification ×200 [A] and ×400 [B]). C, Prominent vascular invasion by tumor cells (arrowheads) (original magnification ×100). D, Perineural invasion by tumor cells (arrowheads) (original magnification ×200). E, Perineural invasion by tumor cells at higher magnification (original magnification ×400). F, Tumor invading between reactive bone trabeculae (original magnification ×200). G, Tumor invading skeletal muscle (original magnification ×200). H, Tumor infiltrating adjacent nonneoplastic minor salivary gland lobule; note the absence of a tumor capsule (original magnification ×100). In all panels where they appear, N indicates nerve fibers; S, skeletal muscle.

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