Abstract
This study assessed the clinical and histological features and therapeutic efficacy of 25 cases of sublingual gland tumours from 1998 to 2008. There were 17 female patients and 8 male, the ratio of females to males was 2.1:1. The mean age was 48.6 years. 4 cases were benign tumours (16%). 21 cases were malignant sublingual gland tumours (84%) and of these, 18 were adenoid cystic carcinoma (86%). Adenoid cystic carcinoma was mainly of the histological type, and the other histological classifications included mucoepidermoid carcinoma, pleomorphic adenoma, myoepithelioma, oncocytoma and polymorphous low-grade adenocarcinoma. Sublingual gland tumours are rare and most are malignant. For malignant sublingual gland tumours, early diagnosis and aggressive surgical treatment, especially for tumours with nerve involvement, is the key to improving prognosis. Free radial forearm flap or pectoralis major myocutaneous flap are appropriate methods for mouth floor reconstruction. For benign sublingual gland tumours, the resection of tumour and sublingual gland is the preferred treatment.
70–80% of salivary gland tumours originate in the parotid gland; 7–11% occur in the submandibular gland; less than 1% occur in the sublingual gland. Generally, the smaller the salivary gland involved, the higher the possibility of the tumour being malignant. Malignant tumours comprise 15–32% of parotid tumours, 41–45% of submandibular gland tumours, and 70–90% of sublingual gland tumours . Most tumours of the sublingual gland are malignant, with adenoid cystic carcinoma (ACC) and mucoepidermoid carcinoma (MEC) being the most frequent. Many other malignant tumour types, such as acinic cell carcinoma , malignant mixed tumour , squamous cell carcinoma and clear cell carcinoma , have also been reported. Pleomorphic adenoma is the main histological type of benign tumour.
The aim of this study was to outline the clinicopathologic characteristics of the disease and to evaluate currents treatment modalities.
Materials and methods
During the 10-year period from 1998 to 2008, 1298 patients were treated for salivary gland tumours (not including minor salivary gland tumours of the nasal cavity, paranasal sinuses and pharynx) at the authors’ institution. 25 cases (2%) with the diagnosis of sublingual gland tumours were identified. This group included 17 women and 8 men (ratio: 2.1:1), ranging in age from 32 to 71 years (mean 48.6 years). 4 cases (16%) were benign tumours, 21 cases (84%) were malignant tumours. In the benign tumour group, 2 cases were pleomorphic adenoma, 1 case was myoepithelioma and 1 case was oncocytoma. In the malignant tumour group, 18 cases were ACC, 2 cases were MEC with high grade and 1 case was polymorphous low-grade adenocarcinoma (PLGA). The follow-up period ranged from 8 to 99 months. The mean follow-up was 30.7 months. The clinical and histological features, treatment, staging, and outcome were reviewed ( Tables 1 and 2 ).
| Age | Sex | Presence of mass | Size (cm) | Symptoms | Pathologic diagnosis | Treatment | Follow-up time (months) | |
|---|---|---|---|---|---|---|---|---|
| 1 | 47 | F | 2 mo | 0.6 × 0.6 | Firm, painless, mobile, asymptomatic mass. | Myoepithelioma | Resection of the sublingual gland and mass. | 99 |
| 2 | 41 | F | 6 mo | 2.0 × 2.5 | Firm, progressively enlarging, painless, asymptomatic mass. | Pleomorphic adenoma | Resection of the sublingual gland and mass. | 64 |
| 3 | 65 | F | 3 days | 3.0 × 2.5 | Firm, painless, mobile, asymptomatic mass. | Pleomorphic adenoma | Resection of the sublingual gland and mass. | 23 |
| 4 | 59 | M | 1 mo | 3.7 × 3.1 | Firm, progressively enlarging, painless, asymptomatic mass. | Oncocytoma | Resection of the sublingual gland and mass. | 17 |
| Case no. | Age/sex | TNM stage | Presence of mass | Size (cm) | Presence of pain | Presence of numbness | Pathologic diagnosis | Treatment | Follow-up time |
|---|---|---|---|---|---|---|---|---|---|
| 1 | 46/F | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 2 mo | 3.0 × 1.5 | 2 mo | N | High-grade MEC | Resection of sublingual gland and lingual nerve + neck dissection (level I) + mouth floor reconstruction with FRFF | LFU, 12 mo |
| 2 | 42/F | T 1 cN 0 M 0 /T 1 pN 0 M 0 | 4 mo | 1.0 × 0.5 | 4 mo | 4 mo | ACC | Resection of sublingual gland and lingual nerve + neck dissection (level I–III) | AWD, 84 mo; lung metastasis at 47 mo |
| 3 | 67/F | T 2 cN 0 M PUL /T 2 pN 0 M PUL | 4 yr | 4.0 × 3.0 | 1 mo | 1 mo | ACC | Chemotherapy. No surgery for lung metastasis | DOD, 21 mo; lung and brain metastasis |
| 4 | 45/M | T 1 cN 0 M 0 /T 1 pN 0 M 0 | 4 mo | 2.0 × 1.5 | N | N | ACC | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I–III) | LFU, 8 mo |
| 5 | 39/F | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 20 days | 2.5 × 2.0 | N | N | ACC | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I–III) | AWD, 61 mo; local recurrence/lung and chest wall metastasis at 27 mo |
| 6 | 41/F | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 6 mo | 2.5 × 2.5 | N | N | ACC | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I–III) | NED, 50 mo |
| 7 | 36/F | T 1 cN 0 M 0 /T 1 pN 0 M 0 | 3 yr | 1.5 × 1.0 | N | N | ACC | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I–III) + mouth floor reconstruction with FRFF | NED, 50 mo |
| 8 | 71/F | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 8 mo | 2.3 × 1.5 | N | N | ACC | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I–III) | LFU, 15 mo |
| 9 | 59/F | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 6 mo | 3.1 × 1.9 | 6 mo | 2 mo | ACC | Resection of sublingual gland and lingual nerve + segment mandibulectomy + neck dissection (level I–III) + mandible reconstruction with titanium plate + mouth floor reconstruction with FRFF | NED, 36 mo |
| 10 | 47/F | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 7 days | 3.0 × 1.0 | N | N | ACC | Resection of sublingual gland and lingual nerve + neck dissection (level I–III) + RT + Chemotherapy | NED, 33 mo |
| 11 | 56/F | T 1 cN 0 M 0 /T 1 pN 0 M 0 | 3 mo | 2.0 × 1.5 | N | 7 days | High-grade MEC | Resection of sublingual gland | NED, 28 mo |
| 12 | 59/M | T 1 cN 0 M 0 /T 1 pN 1 M 0 | 2 mo | 1.5 × 1.5 | 10 days | 10 days | ACC | Resection of sublingual gland and lingual nerve + neck dissection (level I-III) + RT | NED, 27 mo |
| 13 | 32/F | T 3 cN 0 M 0 /T 3 pN 0 M 0 | 7 yr | 6.8 × 3.3 | 7 yr | 1 yr | ACC | Resection of sublingual gland, lingual nerve and hypoglossal nerve + segment mandibulectomy + neck dissection (level I-III) + mandible reconstruction with titanium plate + mouth floor reconstruction with PMMF | AWD, 23 mo; vertebrae metastasis at 18 mo |
| 14 | 35/M | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 6 mo | 3.5 × 2.5 | 10 days | 10 days | PLGA | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I-III) + mouth floor reconstruction with PMMF | LFU, 12 mo |
| 15 | 57/M | T 2 cN 1 M 0 /T 2 pN 0 M 0 | 3 mo | 2.5 × 2.0 | N | N | ACC | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I–V) + mouth floor reconstruction with FRFF + RT | NED, 14 mo |
| 16 | 52/M | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 10 days | 4.0 × 2.0 | N | N | ACC | Resection of sublingual gland, lingual nerve and hypoglossal nerve + segment mandibulectomy + neck dissection (level I-III) + mouth floor reconstruction with FRFF + Chemotherapy | NED, 13 mo |
| 17 | 39/F | T 3 cN 0 M 0 /T 3 pN 2b M 0 | 14 days | 4.8 × 3.0 | N | N | ACC | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I–III) + mouth floor reconstruction with PMMF +RT | AWD, 36 mo; Local recurrence/lung metastasis at 30 mo |
| 18 | 46/F | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 2 mo | 2.5 × 2.0 | 2 mo | N | ACC | Resection of sublingual gland, lingual nerve and hypoglossal nerve + segment mandibulectom + neck (level I–III) + mouth floor reconstruction with FRFF | NED, 14 mo |
| 19 | 39/F | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 20 days | 4.0 × 2.0 | N | N | ACC | Resection of sublingual gland and lingual nerve + marginal mandibulectomy + neck dissection (level I–III) + mouth floor reconstruction with FRFF + RT | NED, 9 mo |
| 20 | 39/M | T 2 cN 0 M 0 /T 2 pN 0 M 0 | 6 mo | 2.5 × 1.5 | 6 mo | 15 days | ACC | Resection of sublingual gland, lingual nerve and hypoglossal nerve + neck dissection (level I–III) + mouth floor reconstruction with FRFF | NED, 8 mo |
| 21 | 57/M | T 3 cN 0 M 0 /T 3 pN 0 M 0 | 2 yr | 4.4 × 2.9 | 2 yr | N | ACC | Resection of sublingual gland, lingual nerve and hypoglossal nerve + segment mandibulectomy + neck dissection (level I–III) + mouth floor reconstruction with PMMF | NED, 11 mo |
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