Upper Lip Salivary Carcinoma

Figure 10.12b. Computerized tomograms were obtained that identified destruction of bone by the cancer.

Figure 10.12d. The maxillectomy specimen delivered.

Salivary Gland Cancer Symptoms

Figure 10.12f. The large ablative defect. Figure 10.12h. At 1 year postoperatively, the patient showed no signs of recurrent disease.

Figure 10.12f. The large ablative defect. Figure 10.12h. At 1 year postoperatively, the patient showed no signs of recurrent disease.

The surgical treatment of polymorphous low-grade adenocarcinoma of the upper lip or buccal mucosa is similar to that of a mucoepidermoid carcinoma of these regions. The basic approach involves a mucosal-sacrificing wide local excision with attention to submucosal anatomic barriers being included on the specimen so as to ensure tumor-free margins (Figure 10.13).

The use of radiation therapy has been assessed in the management of polymorphous low-grade adenocarcinoma. In a clinicopathologic study of 164 cases of this malignancy, 17 patients underwent incisional, excisional, or wide local excision followed by radiation therapy (Castle, Thompson, and Frommelt et al. 1999). Adjuvant radiation therapy did not affect survival. Their study showed that patients who were treated with radiation therapy were more likely to have evidence of disease at last follow-up when compared with patients who did not have radiation therapy. Furthermore, there was no statistically significant difference in the overall patient outcome based on the type of initial treatment given or for any additional treatment rendered, whether it was additional surgery, radiation therapy, or chemotherapy. Based on this report and others (Crean et al. 1996), the treatment for polymorphous low-grade adeno-carcinoma of minor salivary glands remains surgi-

cal. It has been estimated that approximately 80% of patients survive their disease without evidence of tumor at periods from between several months to 25 years after removal (Wenig and Gnepp 1991). One case has been reported where death occurred as a result of this neoplasm with direct extension to vital structures of the head (Aberle, Abrams, and Bowe et al. 1985). In addition, while rare, metastasis to cervical lymph nodes (Kumar, Stiva-ros, and Barrett et al. 2004) and to distant organs (Hannen, Bulten, and Festen et al. 2000) has been reported from polymorphous low-grade adenocar-cinomas originating in the palate. These reports indicate that cervical lymph node involvement should be suspected in patients with papillary cystic change in the tumor, and that periodic chest X-ray examination should be performed postopera-tively when this variant of tumor is diagnosed.

Figure 10.13b. A very thick tumor was noted on biopsy. The superimposed optical micrometer shows the tumor to be about 9 mm in thickness.

Figure 10.13a. A biopsy proved polymorphous low-grade adenocarcinoma of the buccal mucosa in a 45-year-old woman.

Figure 10.13b. A very thick tumor was noted on biopsy. The superimposed optical micrometer shows the tumor to be about 9 mm in thickness.

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Figures 10.13c and 10.13d. A mucosal-sacrificing wide local excision with 1 cm linear margins and isolation of Stenson's duct was performed.

Blood Clot
Figure 10.13e. The specimen was able to be delivered without tumor spillage.

Figure 10.13f. The defect was reconstructed with a split thickness skin graft and a sialodochoplasty of Stenson's duct.

Figure 10.13g. Acceptable healing is noted without tumor recurrence at 6 months postoperatively. Reprinted with permission from Carlson ER. 1995. Salivary gland pathology—clinical perspectives and differential diagnosis. In: The Comprehensive Management of Salivary Gland Pathology, Oral and Maxillofacial Surgery Clinics of North America 7. Philadelphia: W.B. Saunders, pp. 361-386.

Figure 10.13g. Acceptable healing is noted without tumor recurrence at 6 months postoperatively. Reprinted with permission from Carlson ER. 1995. Salivary gland pathology—clinical perspectives and differential diagnosis. In: The Comprehensive Management of Salivary Gland Pathology, Oral and Maxillofacial Surgery Clinics of North America 7. Philadelphia: W.B. Saunders, pp. 361-386.

Acinic Cell Adenocarcinoma

Acinic cell adenocarcinoma is a very rare malignancy of the minor salivary glands. It has been estimated to represent approximately 2.5-3% of salivary gland tumors in general (Guimaraes et al. 1989; Spiro 1986) and about 4% of minor salivary gland tumors (Castellanos and Lally 1982). Indeed, acinic cell adenocarcinoma is not represented in many studies of minor salivary gland tumors (Chau and Radden 1986; Isacsson and Shear 1983; Jaber 2006), and other studies show only a very limited number of these cases (Lopes et al. 1999; Toida, Shimokawa, and Makita et al. 2005). The acinic cell adenocarcinoma behaves most similarly to the low-grade mucoepidermoid carcinoma (Ord 1994). In fact, like the low-grade mucoepidermoid carcinoma, the acinic cell adenocarcinoma was originally purported to be a benign neoplasm (Ellis and Auclair 1991a). For the first half of the twentieth century, these tumors were thought to be benign. In 1953, Buxton and his group were the first to ascribe a malignant character to many of these tumors (Buxton, Maxwell, and French 1953). These were identified as serous cell adenocarcinomas, after which time Foote and Frazell classified these tumors as acinic cell adenocarcinomas (Foote and Frazell 1953).

The AFIP registry shows 886 acinic cell adenocarcinomas, of which 753 were located in the major salivary glands (85%), and 133 (15%) in the minor salivary glands. The most common site of minor salivary gland involvement was the buccal mucosa, accounting for 43 cases (32%), followed by the lip (38 cases, 29%). Tumors in the upper lip were three times more common than tumors in the lower lip. The palate was the only other significant anatomic site to be affected by this tumor, and accounted for 22 cases (17%). A female preponderance was noted, with a mean age of 44 years.

Surgery for acinic cell adenocarcinoma is performed in a similar fashion as that of low-grade mucoepidermoid carcinoma. Tumors of the buccal mucosa and upper lip are treated with mucosal-sacrificing wide local excisions, including 1 cm linear margins, with attention to the necessary sacrifice of surrounding anatomic barriers (Figure 10.14). Tumors of the palate can be treated with bone-sparing, periosteally sacrificing wide local excisions with split thickness sacrifice of the soft palate. Computerized tomograms may be obtained preoperatively to confirm the lack of bone erosion. Recurrences and regional and distant metastases are rare when these malignancies are treated according to these recommendations. Five and 10-year survival rates are generally quite favorable and reported as 82% and 68%, respectively (Hickman, Cawson, and Duffy 1984).

Epithelial-Myoepithelial Carcinoma

The epithelial-myoepithelial carcinoma of minor salivary gland origin has been categorized as an intermediate-grade malignancy according to the AFIP classification (Ellis and Auclair 1991b). Only 57 cases were identified in their series, with 50 cases diagnosed in the major salivary glands (88%), and 7 cases (12%) in the minor salivary glands (Corio 1991). Of the 7 cases in the minor salivary glands, 4 were located in the palate, 1 in the tongue, and 2 cases were not specified as to anatomic location. A mean age of 59 years was noted in these 57 cases. This tumor is known to be highly differentiated, yet it is malignant due to infiltrative and destructive growth patterns, the presence of necrosis, perineural involvement, and metastases (Corio et al. 1982). Corio et al. pre-

Upper Lip Tumor

Figures 10.14a and 10.14b. An acinic cell adenocarcinoma of the right buccal mucosa in a 52-year-old patient. The histopathology of the biopsy shows a typical blue dot tumor (b).

Figure 10.14d. Excision of the specimen occurred without tumor spillage.

Figure 10.14e. The defect was reconstructed with mucosal flaps so as to not distort the appearance of the upper lip.

Figure 10.14c. A mucosal-sacrificing wide local excision observing 1 cm linear margins is performed.

Figure 10.14f. Acceptable healing without tumor recurrence is noted at 1 year postoperatively.

a b sented 16 cases of this neoplasm and found 12 cases to involve the parotid gland, 3 cases in the submandibular gland, and 1 case in the buccal mucosa (Corio et al. 1982).

Standardized recommendations for surgery for the epithelial-myoepithelial carcinoma are difficult to make due to the rare nature of this malignancy. Nonetheless, evaluation of involved anatomic barriers with physical examination and

CT scans generally permits an effective approach to eradication of these malignancies in various minor salivary gland sites (Figure 10.15). In such circumstances, the surgeon respects well-established principles of linear and anatomic barrier margins when operating on salivary gland tumors, while also relying on past experience with other low- and intermediate-grade minor salivary gland malignancies. In so doing, tumor-free margins can

Upper Lip Salivary Carcinoma

Figure 10.15a. A mass of the right maxillary gingiva in a 12-year-old girl.

Figure 10.15b. Panoramic radiograph demonstrates tumor involvement of the bone between the first premolar and canine teeth with divergence of the roots of these teeth.

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Teeth With Divergent Roots

Figures 10.15c and 10.15d. Computerized tomograms demonstrate a soft tissue mass with involvement of the maxillary bone.

Teeth With Divergent Roots

Figure 10.15e. Incisional biopsy showed epithelial-myo-epithelial carcinoma.

Teeth With Divergent Roots

Figure 10.15e. Incisional biopsy showed epithelial-myo-epithelial carcinoma.

Figure 10.15f. A partial maxillectomy observing 1 cm linear margins in bone and soft tissue was performed.

Partial Maxillectomy Specimen

Figures 10.15g and 10.15h. The specimen was able to be removed without tumor spillage.

Figures 10.15g and 10.15h. The specimen was able to be removed without tumor spillage.

Figure 10.15i. Final histopathology showed destruction of bone by the cancer.

Figure 10.15j. The specimen radiograph demonstrated acceptable bone margins.

Figures 10.15k and 10.151. The resultant ablative defect of the maxilla (k) was reconstructed with an immediate obturator device (l).

Figure 10.15m. The use of the obturator permitted contracture of the defect as noted at 1 month postoperatively.

Figure 10.15n. By 3 months postoperatively, the defect had demarcated significantly.

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Figures 10.15p. This interim obturator allowed for seal of the defect and function with teath.

Figures 10.15o. An interim obturator was fabricated.

Figure 10.15q. The patient functioned well with the definitive obturator, which permitted additional contracture of the maxillary defect.

Figures 10.15r, 10.15s, and 10.15t. Soft tissue reconstruction was accomplished with a buccal fat flap and advancement of the buccal mucosa.

Figure 10.15u. The appearance of the healed flap is noted at 1 year postoperatively.

be obtained while performing surgery similar to that for a diagnosis of low- or intermediate-grade mucoepidermoid carcinoma. Recurrences have been reported (Corio 1991), but appropriate surgical management of epithelial-myoepithelial carcinomas of the minor salivary glands should be performed with curative intent. Quantitative survival statistics are not published in the literature.

SURGICAL MANAGEMENT OF THE NECK FOR MINOR SALIVARY GLAND MALIGNANCIES

Surgical management of the neck is a controversial and intriguing concept for surgeons managing oral/ head and neck malignant disease. At the core of this discipline is an assessment of occult disease in patients with clinically negative necks. To this end, there seems to be a consensus in the literature that occult neck disease is relatively uncommon related to minor salivary gland malignancies compared to squamous cell carcinoma of the oral/head and neck region. Moreover, it is also uncommon for patients with minor salivary gland malignancies to present with clinically palpable neck disease related to these tumors. Spiro found 53 patients presenting with cervical metastases among 378 patients (14%) with minor salivary gland malignancies (Spiro, Thaler, and Hicks et al. 1991). Another 26 patients (7%) developed subsequent cervical metastases for an overall rate of nodal involvement of 21%. Inter-

r s estingly, 9 patients underwent an elective neck dissection, all of whom showed histologically confirmed metastatic disease. The authors do not, however, discuss the incidence of occult and clinically apparent metastases as a function of anatomic site of the primary minor salivary gland malignancy. Sadeghi et al. identified 9 patients presenting with cervical metastases related to minor salivary gland malignancies, 5 of which were present in the tongue base (Sadeghi, Tran, and Mark et al. 1993). Beckhardt et al. found N+ necks in only 3% of their patients with malignant minor salivary gland tumors of the palate, while Chung identified only 2 of 20 patients with malignant salivary gland tumors of the palate presenting with cervical metastases (Beckhardt, Weber, and Zane et al. 1995; Chung, Rahman, and Constable 1978). The latter three studies only discussed clinical staging of the neck without comments regarding their histology such that limited information is available regarding the true rate of metastasis to the cervical lymph nodes. In the final analysis, it seems that the incidence of occult neck disease related to a minor salivary gland malignancy of the oral cavity is sufficiently low to negate the need for elective neck dissection. Indications for neck dissection in these patients, therefore, are limited to patients who present with cervical metastases, those patients whose preoperative imaging studies document changes in the cervical lymph nodes consistent with metastatic disease, and those patients with high-grade malignancies, regardless of the clinical and radiographic imaging results.

THE ROLE OF RADIATION THERAPY IN THE MANAGEMENT OF MINOR SALIVARY GLAND MALIGNANCIES

It was once thought that salivary gland malignancies were radioresistant (Dragovic 1995). This previously stated misconception can no longer be considered valid. As such, radiation therapy is indicated in the postoperative management of all high-grade malignant minor salivary gland tumors, as well as in patients with positive surgical margins, positive regional lymph nodes, and recurrent tumor (Dragovic 1995). This being the case, it is important to remember that surgery is the primary therapy for minor salivary gland malignancies. Shingaki et al.'s review of the role of radiation therapy in 44 patients with salivary gland cancers,

34 of whom were treated for minor salivary gland cancers, examined the results of surgery vs. surgery and postoperative radiation therapy in these patients (Shingaki et al. 1992). Interestingly, no patients experienced recurrent disease when negative surgical margins were found in the specimen, regardless of whether surgery or surgery and postoperative radiation therapy was performed. All patients with positive surgical margins developed recurrent disease when surgery was the modality of treatment, and 8 of 15 patients (53%) with positive surgical margins developed recurrent disease when their salivary gland cancer was treated with a combination of surgery and postoperative radiation therapy. While not broken down to major vs. minor salivary gland primary sites, these results do point to the significant benefit of obtaining negative margins in the resected specimen.

THE ROLE OF CHEMOTHERAPY IN THE MANAGEMENT OF MINOR SALIVARY GLAND MALIGNANCIES

There are few reports on the benefit of systemic chemotherapy in the management of salivary gland cancers. Chemotherapy is generally reserved for the palliative management of advanced, non-resectable disease where radiation therapy has already been administered. Most patients for whom chemotherapy is considered will have diagnoses of mucoepidermoid carcinoma, adenoid cystic carcinoma, or high-grade adenocarcinoma (Laurie and Licitra 2006). The expression of c-kit in adenoid cystic carcinoma, overexpression of her-2 in muco-epidermoid carcinoma, overexpression of epithelial growth factor receptor in adenocarcinoma, and androgen receptor positivity in salivary duct carcinoma makes the use of imatinib, trastuzumab, cetuximab, and antiandrogen therapy at least theoretically beneficial (Laurie and Licitra 2006). While these agents may be of value in treating difficult cases of minor salivary gland malignancies, there is a need to conduct high-quality clinical trials in patients with these cancers.

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