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FIGURE 3. Images of postoperative flexible laryngoscopy and bronchoscopy demonstrate the improved airway anatomy as a result of mandibular distraction. A, The larynx is now very easy to expose with a regular straight laryngoscopy blade (A). The tongue base is not collapsed against the epiglottis. B, The vocal cords are easy to visualize. C and D, The trachea, carina, and mainstem bronchi were normal in appearance.
distracted to the point of having a severely skeletal class III relationship, the upper airway obstruction has been adequately managed, and as facial growth occurs, the facial aesthetics normalize, obviating the need for any further surgical interventions. We will continue to follow our patient to determine whether an additional operative intervention will be required.
REFERENCES 1. Ferrari D, Bettuzzi C, Donzelli O. Freeman-Sheldon syndrome. A case report and review of the literature. Chir Organi Mov 2008;92:127Y131 2. Robinson PJ. Freeman Sheldon syndrome: severe upper airway obstruction requiring neonatal tracheostomy. Pediatr Pulmonol 1997;23:457Y459 3. Schefels J, Wenzl TG, Merz U, et al. Functional upper airway obstruction in a child with Freeman-Sheldon syndrome. ORL J Otorhinolaryngol Relat Spec 2002;64:53Y56 4. Freeman EA, Sheldon JH. Cranio-carpo-tarsal dystrophy. Arch Dis Child 1938;13:277Y283 5. Stevenson DA, Carey JC, Palumbos J, et al. Clinical characteristics and natural history of Freeman-Sheldon syndrome. Pediatrics 2006;117:754Y762
Minor Salivary Gland Neoplasms Abdullah Dalgic, MD,* Omer Karakoc, MD,* Umit Aydin, MD,* Yusuf Hidir, MD,* Mehmet Gamsizkan, MD,Þ Serdar Karahatay, MD,* Mustafa Gerek, MD* Objective: This study aimed to investigate the clinical presentation, histopathologic and epidemiological aspects, as well as the treatment modalities and outcomes of patients with minor salivary gland tumors (MSGTs).
From the Departments of *Otolaryngology, Head and Neck Surgery, and †Pathology, Gulhane Military Medical School, Ankara, Turkey. Received December 11, 2013. Accepted for publication January 6, 2014. Address correspondence and reprint requests to Abdullah Dalgic, MD, Gulhane Askeri Tip Akademisi, Kbb Ad, 06018, Etlik, Ankara, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000723
Brief Clinical Studies
Subjects and Methods: A series of 23 patients with MSGTs were reviewed retrospectively. Results: This study included 11 (48%) benign and 12 (52%) malignant tumors of minor salivary glands. Minor salivary gland tumors were more common in men (70%) than in women (30%). The mean age was 31.3 years for benign tumors and 46.3 years for malignant tumors. Pleomorphic adenoma was the most common benign tumor, followed by myoepithelioma. Mucoepidermoid carcinoma and adenoid cystic carcinoma were the most common malignant tumors. The most common symptom was a painless mass of the palate. Surgical treatment was performed in all patients. Adjuvant radiotherapy was used in 3 malignant tumors. Twenty-three patients were followed-up for a median of 5 years. Two patients with malignant tumors underwent a second surgery for postoperative local recurrence. They were successfully treated with the second surgery. Conclusions: Minor salivary gland tumors are relatively uncommon neoplasms of the head and neck region. There is limited literature on MSGTs. This study provides a versatile approach for MSGTs from demographic data and clinical presentations to treatment modalities and treatment outcomes. Key Words: Palate, minor salivary gland
S
alivary gland neoplasms are uncommon and account for 3% of all head and neck tumors.1,2 They exhibit a wide range of benign and malignant histologic types. Salivary gland neoplasms originate from the major or minor salivary glands Minor salivary gland tumors (MSGTs) account for 15% to 25% of all salivary gland neoplasms.1,3 Most salivary gland tumors are benign, and by far, the most common site is the parotid gland, followed by the submandibular gland and minor salivary glands.4 The most common location of MSGTs is the oral cavity.5 Minor salivary glands are located throughout the submucosa of the oral cavity. Palate is the most common site for MSGTs in the oral cavity. Pleomorphic adenoma is the most common benign histologic type, whereas mucoepidermoid carcinoma and adenoid cystic carcinoma are the most common malignant histologic types. Unlike major salivary gland neoplasms, most MSGTs are malignant.6 Most studies are concerned with major and MSGTs together, and there are limited articles related to MSGTs separately. The purposes of this study were to investigate the clinical presentation and the histopathologic and epidemiological aspects as well as, concurrently, to report treatment modalities and outcomes in patients with MSGTs.
PATIENTS AND METHODS The records of patients who underwent surgical intervention for MSGTs at the Department of Otorhinolaryngology, Head and Neck Surgery, Gulhane Military Medical School (Ankara, Turkey), between January 1994 and November 2010, were reviewed. Inclusion criteria were a histopathologic diagnosis of MSGTs as well as preoperative and postoperative adequate information. Exclusion criteria included subjects who have salivary gland tumors except in the minor salivary gland and subjects who do not have adequate followup data. The Gulhane Military Medical School ethics committee approved the study. The medical history, age, sex, duration of symptoms, clinical presentation, preoperative investigations, histopathologic diagnosis, as well as the treatment modality and treatment outcome of the patients were reviewed retrospectively.
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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TABLE 1. The Demographic Data, Histopathologic Diagnosis, and Origins of the Masses of Patients Who Have MSGTs
Histopathology of Tumor Benign tumors Pleomorphic adenoma
Mean Number age, y
Sex
Duration of Symptoms, mo
Tumor Location
11 8 3
31.3
M:8 F:3
17
Soft palate:3 Hard palate:8
12 6 6
46.3
M:8 F:4
9
Soft palate:3 Hard palate:7 Retromolar:1 Gingivobuccal:1
23
38.3
M:16 F:7
13
Soft palate:6 Hard palate:15
Myoepithelioma Malignant tumors Mucoepidermoid CA Adenoid cystic CA All tumors
Retromolar:1 Gingivobuccal:1
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All tumors were treated surgically through local excision with wide margins. For malignant tumors, surgery was the primary treatment modality, mostly alone. Wide local excision combined with maxillectomy was performed on 8 of 12 patients with malignant tumors. Four patients with malignant tumors had complementary surgery immediately after the first surgery because of positive surgical margin in the frozen section. Neck dissection was not performed because there was no presence of lymphadenopathy clinically or radiologically. Adjuvant radiotherapy was performed on 3 patients: 1 patient with mucoepidermoid carcinoma in the retromolar trigon, 1 patient with adenoid cystic carcinoma in the palate, and 1 patient with low-grade mucoepidermoid carcinoma. Radiotherapy was applied on these patients because of a positive surgery margin, and no recurrence was seen. The postoperative local recurrence was seen in 2 patients in the malignant tumor group: 1 patient with adenoid cystic carcinoma and 1 patient with mucoepidermoid carcinoma that was located in the palate. An extended surgery was performed on these patients. The patients were followed up, with a median of 5 years (range, 1Y13 y).
CA, carcinoma; F, female; M, male.
Histopathologic diagnosis was made according to the World Health Organization’s histologic typing of salivary gland tumors. Independent samples t-test was used for comparing the mean ages in malignant and benign tumor groups. P value less than 0.05 was considered statistically significant.
RESULTS A total of 23 MSGTs were identified. Of these, 11 were benign (48%) and 12 (52%) were malignant tumors. There were 7 women (30%) and 16 men (70%). The group of benign tumors included 3 women and 6 men; the group of malignant tumors included 3 women and 7 men. The mean age was 31.3 years (range, 16Y75 y) for the benign tumor group and 46.3 years (range, 21Y76 y) for the malignant tumor group. The mean age of the malignant tumor group was 15 years older than the mean age of the benign tumor group. However, the difference in the mean age between the malignant and benign tumor groups was not statistically significant (P = 0.162, P > 0.05). Pleomorphic adenoma was the most common benign tumor, accounting for 73% (8 patients) of benign tumors and 30% of all patients. Myoepithelioma was the second most common benign tumor (27%, 3 patients). Mucoepidermoid carcinoma and adenoid cystic carcinoma were the most common malignant MSGT. Both of them had an equal frequency of 50% (6 patients). The common symptom of the patients was a painless mass in the mouth located on the palate (17 patients, 73%). Three patients (13%) complained of pain. The mean duration time of symptoms was 2 to 36 months and the mean time was 13 months. The mean time was 9 months for the malignant tumor group and 17 months for the benign tumor group. Locations of the tumors were the soft palate in 7 patients, the hard palate in 13 patients, the retromolar area in 2 patients, and the gingivobuccal mucosa in 1 patient. Table 1 shows the histopathologic diagnoses, demographic data, and tumor location of the subjects. The preoperative imaging was performed on all patients. Computed tomography (CT) was the most common preoperative diagnostic procedure that was performed on all patients. Fine-needle aspiration biopsy (FNAB) was performed on 6 patients, and FFNAB was accurate in all of them: 3 pleomorphic adenoma, 2 adenoid cystic carcinomas, and 1 myoepithelioma.
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DISCUSSION Salivary gland neoplasms constitute less than 1% of all tumors and 3% to 5% of all head and neck neoplasms. Minor salivary gland tumors are uncommon and comprise 15% to 20% of all salivary gland neoplasms. The most common tumor of MSGTs is pleomorphic adenoma.1,7Y11 The most common malignant neoplasms are mucoepidermoid carcinoma and adenoid cystic carcinoma.12 In our study, pleomorphic adenoma was the most common tumor type (30%) and the most common benign tumor. The most common malignant tumors were mucoepidermoid carcinoma (26%) and adenoid cystic carcinoma (26%). These data are closely related to that of other studies. The frequency of benign and malignant MSGTs is a controversial issue according to the literature. Most studies reveal that benign tumors are more common than malignant tumors.1,3,7,11 However, several studies report that malignant tumors were seen more frequently than benign tumors.9,10,13 In this study, benign tumors (52%) were slightly higher than malignant tumors (48%). Minor salivary gland tumors may occur in the oral cavity, paranasal sinuses, nasopharynx, and larynx. The most common location of MSGTs is the oral cavity and the palate is the most common place for MSGTs. It was reported in different studies that 40% to 80% of MSGTs originated from the palate.1,3,7,10 Similarly, in the current study, the palate was the most common site for all MSGTs with the rate of 91% ( 21 subjects in 23). In the benign tumor group, the hard palate was the site in 8 subjects and the soft palate was the site in 3 subjects. In the malignant tumor group, the hard palate was the site in 7 subjects and the soft palate was the site in 3 subjects. Most studies have shown that MSGTs are more common in females than in males.7,8 However, it was found in the current study that MSGTs were more common in men (70%) than in women (30%). This difference can be a result of racial factors. Minor salivary gland tumors can occur at any age, but the maximum incidence is in the fourth decade of life for benign tumors and in the fifth decade of life for malignant tumors. However, some authors have reported that the peak incidence occurs in the period between the fifth and seventh decades of life.14 In this study, the peak incidence of MSGTs was in the fourth decade, ranging from 16 to 75 years. For malignant tumors, the peak incidence was in the sixth decade, ranging from 21 to 76 years. It has been reported that the difference in the mean appearing age of MSGTs among the patients with benign and malignant tumors ranged from 3.4 to 13.3 years.9 Although several of these studies * 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
& Volume 25, Number 3, May 2014
revealed a statistically significant difference in the mean age, many studies revealed no statistically significant difference.7,9Y11 In this study, there was no statistically significant difference between the mean age of benign and malignant MSGTs (P = 0.073 and P < 0.05). Signs and symptoms depend on the location and size of tumor.15 Patients commonly complain of a painless mass in the palate. Pain, paresthesia, dysphagia, speech impairment, and referred otalgia may also occur. In the current study, we found that a painless mass in the palate was the most common presentation (17 patients, 73%). The mean time of symptoms before the treatment was averagely 13 months, ranging from 2 to 36 months. It is very difficult to distinguish benign MSGTs from malignant MSGTs only through a physical examination. Preoperative imaging, incisional biopsy, or FNAB must be done preoperatively in planning the surgery. In the literature, it is stated that histopathologic correlation was increased when preoperative investigations such as FNAB and CT were used.16,17 In this study, preoperatively, CT and FNAB were used for the investigation. Treatment of oral cavity MSGTs includes complete surgical excision. Benign neoplasms can be treated solely through surgical excision. Pleomorphic adenomas must be completely excised with an adequate margin. Incomplete excision causes recurrence of tumor and difficulty in future operations.18 Complete surgical excision was performed on all benign tumors, and no recurrence was seen in the postoperative period in our patients. Treatment of malignant tumors may require palatectomy for palatal lesions, removal of the buccinator muscle for cheek lesions, and removal of the maxilla or mandible if they are involved.18 When an extended resection is performed, reconstructive surgery may be needed to be performed. In our series of patients, reconstructive surgery was required in 8 patients with malignant tumors and in 1 patient with benign tumor. Reconstruction by local flap was performed on 3 of 9 patients and by obturator prosthesis in 6 of 9 patients. Frozen section biopsy was used during the surgery, and a subsequent resection of tumor was necessary to 4 of 12 patients (33%) with malignant tumors because of a positive surgical margin. Neck dissections are recommended for clinically positive necks and for patients with high-grade mucoepidermoid carcinoma. Neck dissection was not performed because our patients had N0 neck and had no high-grade mucoepidermoid carcinoma. Malignant tumors can be treated through radiotherapy and chemotherapy in addition to surgery. The patients were followed up periodically in terms of local recurrence. In our series, the mean postoperative follow-up time was 5 years. There is a limited number of studies examining only MSGTs. Furthermore, there is a great histopathologic variability in salivary gland tumors according to classifications of the World Health Organization.19 Management of other types of salivary neoplasms that were not mentioned in our study is more challenging because of their relative infrequency, inconsistent classification, and variable biologic behavior.20,21 A better understanding of the histogenesis of neoplasms of the salivary glands by further studies about ultrastructural and molecular analysis may provide significant information about their behavior and therapeutic response.22
CONCLUSIONS Minor salivary gland tumors are uncommon and a relatively small group in salivary gland tumors. Hence, studies focusing on minor salivary gland tumors are limited. Our study is important to light the way for clinical presentation, treatment, and the outcomes in patients with MSGTs. Malignant and benign MSGTs were found almost equally in our study. Surgical excision with safety margin is suffi-
Brief Clinical Studies
cient in most of the patients especially in benign tumors. There is a necessity for studies in larger patient series related with MSGTs.
REFERENCES 1. Auclair PL, Ellis GL, Gnepp DR, et al. Salivary gland neoplasms: general considerations. In: Ellis GL, Auclair PL, Gnepp DR, eds. eds. Surgical Pathology of the Salivary Glands. Philadelphia: WB Saunders Co, 1991:135Y164 2. Leegaard T, Lindeman H. Salivary gland tumours: clinical picture and treatment. Acta Otolaryngol 1970;263:155Y159 3. Eveson JW, Cawson RA. Tumours of the minor (oropharyngeal) salivary glands: a demographic study of 336 cases. J Oral Pathol 1985;14:500Y509 4. Spiro RH. Salivary neoplasms: overview of a 35-year experience with 2.807 patients. Head Neck Surg 1986;8:177Y184 5. Strick MJ, Kelly C, Soames JV, et al. Malignant tumours of the minor salivary glandsVa 20 year review. Br J Plast Surg 2004;57: 624Y631 6. Ma DQ, Yu GY. Tumours of the minor salivary glands. A clinicopathologic study of 243 cases. Acta Otolaryngol 1987;103:325Y331 7. Rivera-Bastidas H, Ocanto RA, Acevedo AM. Intraoral minor salivary gland tumours: a retrospective study of 62 cases in a Venezuelan population. J Oral Pathol Med 1996;25:1Y4 8. Van der Wal JE, Snow GB, Van der Wal I. Histological reclassification of 101 intraoral salivary gland tumours (new WHO classification). J Clin Pathol 1992;45:834Y835 9. Van Heerden WFP, Raubenheumer EJ. Intraoral salivary gland neoplasms: a retrospective study of seventy cases in an African population. Oral Surg Oral Med Oral Pathol 1991;71:579Y582 10. Lopes MA, Kowalski LP, Santos GC, et al. Clinicopathologic study of 19 intraoral minor salivary gland tumours. J Oral Pathol Med 1999;28:264Y267 11. Loyola AM, De Araujo VC, De Sousa SOM, et al. Minor salivary gland tumours. A retrospective study of 16 cases in a Brazilian population. Eur J Cancer B Oral Oncol 1995;31B:197Y201 12. Bradley PJ. Submandibular gland and minor salivary gland neoplasms. Curr Opin Otolaryngol Head Neck Surg 1999;7:72Y78 13. Jansisyanont P, Blanchaert RH Jr, Ord RA. Intraoral minor salivary gland neoplasm: a single institution experience of 80 cases. Int J Oral Maxillofac Surg 2002;31:257Y261 14. Vicente OP, Marque´s NA, Ayte´s LB, et al. Minor salivary gland tumours: a clinicopathological study of 18 cases. Med Oral Patol Oral Cir Bucal 2008;13:E582YE588 15. Guzzo M, Locati LD, Prott FJ, et al. Major and minor salivary gland tumours. Crit Rev Oncol Hematol 2010;74:134Y148 16. Bandyopadhyay A, Das TK, Raha K, et al. A study of fine needle aspiration cytology of salivary gland lesions with histopathological corroboration. J Indian Med Assoc 2005;103:312Y314 17. Chan MK, McGuire LJ, King W, et al. Cytodiagnosis of 112 salivary gland lesions. Correlation with histologic and frozen section diagnosis. Acta Cytol 1992;36:353Y363 18. Gates GA. Malignant neoplasms of the minor salivary glands. N Engl J Med 1982;306:718Y722 19. Seifert G, Sobin LH. The World Health Organization’s Histological Classification of Salivary Gland Tumours. A commentary on the second edition. Cancer 1992;70:379Y385 20. Bell RB, Dierks EJ, Homer L, et al. Management and outcome of patients with malignant salivary gland tumours. J Oral Maxillofac Surg 2005;63:917 21. Ampil FL, Misra RP. Factors influencing survival of patients with adenoid cystic carcinoma of the salivary glands. J Oral Maxillofac Surg 1994;45:1005 22. Suen JY, Hanna EY, Lee Chun YS. Benign neoplasms of the salivary glands. In: Cummings CW, et al., eds. Otolaryngology Head and Neck Surgery 4th ed. 2005:1348
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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FIGURE 3. Images of postoperative flexible laryngoscopy and bronchoscopy demonstrate the improved airway anatomy as a result of mandibular distraction. A, The larynx is now very easy to expose with a regular straight laryngoscopy blade (A). The tongue base is not collapsed against the epiglottis. B, The vocal cords are easy to visualize. C and D, The trachea, carina, and mainstem bronchi were normal in appearance.
distracted to the point of having a severely skeletal class III relationship, the upper airway obstruction has been adequately managed, and as facial growth occurs, the facial aesthetics normalize, obviating the need for any further surgical interventions. We will continue to follow our patient to determine whether an additional operative intervention will be required.
REFERENCES 1. Ferrari D, Bettuzzi C, Donzelli O. Freeman-Sheldon syndrome. A case report and review of the literature. Chir Organi Mov 2008;92:127Y131 2. Robinson PJ. Freeman Sheldon syndrome: severe upper airway obstruction requiring neonatal tracheostomy. Pediatr Pulmonol 1997;23:457Y459 3. Schefels J, Wenzl TG, Merz U, et al. Functional upper airway obstruction in a child with Freeman-Sheldon syndrome. ORL J Otorhinolaryngol Relat Spec 2002;64:53Y56 4. Freeman EA, Sheldon JH. Cranio-carpo-tarsal dystrophy. Arch Dis Child 1938;13:277Y283 5. Stevenson DA, Carey JC, Palumbos J, et al. Clinical characteristics and natural history of Freeman-Sheldon syndrome. Pediatrics 2006;117:754Y762
Minor Salivary Gland Neoplasms Abdullah Dalgic, MD,* Omer Karakoc, MD,* Umit Aydin, MD,* Yusuf Hidir, MD,* Mehmet Gamsizkan, MD,Þ Serdar Karahatay, MD,* Mustafa Gerek, MD* Objective: This study aimed to investigate the clinical presentation, histopathologic and epidemiological aspects, as well as the treatment modalities and outcomes of patients with minor salivary gland tumors (MSGTs).
From the Departments of *Otolaryngology, Head and Neck Surgery, and †Pathology, Gulhane Military Medical School, Ankara, Turkey. Received December 11, 2013. Accepted for publication January 6, 2014. Address correspondence and reprint requests to Abdullah Dalgic, MD, Gulhane Askeri Tip Akademisi, Kbb Ad, 06018, Etlik, Ankara, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000723
Brief Clinical Studies
Subjects and Methods: A series of 23 patients with MSGTs were reviewed retrospectively. Results: This study included 11 (48%) benign and 12 (52%) malignant tumors of minor salivary glands. Minor salivary gland tumors were more common in men (70%) than in women (30%). The mean age was 31.3 years for benign tumors and 46.3 years for malignant tumors. Pleomorphic adenoma was the most common benign tumor, followed by myoepithelioma. Mucoepidermoid carcinoma and adenoid cystic carcinoma were the most common malignant tumors. The most common symptom was a painless mass of the palate. Surgical treatment was performed in all patients. Adjuvant radiotherapy was used in 3 malignant tumors. Twenty-three patients were followed-up for a median of 5 years. Two patients with malignant tumors underwent a second surgery for postoperative local recurrence. They were successfully treated with the second surgery. Conclusions: Minor salivary gland tumors are relatively uncommon neoplasms of the head and neck region. There is limited literature on MSGTs. This study provides a versatile approach for MSGTs from demographic data and clinical presentations to treatment modalities and treatment outcomes. Key Words: Palate, minor salivary gland
S
alivary gland neoplasms are uncommon and account for 3% of all head and neck tumors.1,2 They exhibit a wide range of benign and malignant histologic types. Salivary gland neoplasms originate from the major or minor salivary glands Minor salivary gland tumors (MSGTs) account for 15% to 25% of all salivary gland neoplasms.1,3 Most salivary gland tumors are benign, and by far, the most common site is the parotid gland, followed by the submandibular gland and minor salivary glands.4 The most common location of MSGTs is the oral cavity.5 Minor salivary glands are located throughout the submucosa of the oral cavity. Palate is the most common site for MSGTs in the oral cavity. Pleomorphic adenoma is the most common benign histologic type, whereas mucoepidermoid carcinoma and adenoid cystic carcinoma are the most common malignant histologic types. Unlike major salivary gland neoplasms, most MSGTs are malignant.6 Most studies are concerned with major and MSGTs together, and there are limited articles related to MSGTs separately. The purposes of this study were to investigate the clinical presentation and the histopathologic and epidemiological aspects as well as, concurrently, to report treatment modalities and outcomes in patients with MSGTs.
PATIENTS AND METHODS The records of patients who underwent surgical intervention for MSGTs at the Department of Otorhinolaryngology, Head and Neck Surgery, Gulhane Military Medical School (Ankara, Turkey), between January 1994 and November 2010, were reviewed. Inclusion criteria were a histopathologic diagnosis of MSGTs as well as preoperative and postoperative adequate information. Exclusion criteria included subjects who have salivary gland tumors except in the minor salivary gland and subjects who do not have adequate followup data. The Gulhane Military Medical School ethics committee approved the study. The medical history, age, sex, duration of symptoms, clinical presentation, preoperative investigations, histopathologic diagnosis, as well as the treatment modality and treatment outcome of the patients were reviewed retrospectively.
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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TABLE 1. The Demographic Data, Histopathologic Diagnosis, and Origins of the Masses of Patients Who Have MSGTs
Histopathology of Tumor Benign tumors Pleomorphic adenoma
Mean Number age, y
Sex
Duration of Symptoms, mo
Tumor Location
11 8 3
31.3
M:8 F:3
17
Soft palate:3 Hard palate:8
12 6 6
46.3
M:8 F:4
9
Soft palate:3 Hard palate:7 Retromolar:1 Gingivobuccal:1
23
38.3
M:16 F:7
13
Soft palate:6 Hard palate:15
Myoepithelioma Malignant tumors Mucoepidermoid CA Adenoid cystic CA All tumors
Retromolar:1 Gingivobuccal:1
& Volume 25, Number 3, May 2014
All tumors were treated surgically through local excision with wide margins. For malignant tumors, surgery was the primary treatment modality, mostly alone. Wide local excision combined with maxillectomy was performed on 8 of 12 patients with malignant tumors. Four patients with malignant tumors had complementary surgery immediately after the first surgery because of positive surgical margin in the frozen section. Neck dissection was not performed because there was no presence of lymphadenopathy clinically or radiologically. Adjuvant radiotherapy was performed on 3 patients: 1 patient with mucoepidermoid carcinoma in the retromolar trigon, 1 patient with adenoid cystic carcinoma in the palate, and 1 patient with low-grade mucoepidermoid carcinoma. Radiotherapy was applied on these patients because of a positive surgery margin, and no recurrence was seen. The postoperative local recurrence was seen in 2 patients in the malignant tumor group: 1 patient with adenoid cystic carcinoma and 1 patient with mucoepidermoid carcinoma that was located in the palate. An extended surgery was performed on these patients. The patients were followed up, with a median of 5 years (range, 1Y13 y).
CA, carcinoma; F, female; M, male.
Histopathologic diagnosis was made according to the World Health Organization’s histologic typing of salivary gland tumors. Independent samples t-test was used for comparing the mean ages in malignant and benign tumor groups. P value less than 0.05 was considered statistically significant.
RESULTS A total of 23 MSGTs were identified. Of these, 11 were benign (48%) and 12 (52%) were malignant tumors. There were 7 women (30%) and 16 men (70%). The group of benign tumors included 3 women and 6 men; the group of malignant tumors included 3 women and 7 men. The mean age was 31.3 years (range, 16Y75 y) for the benign tumor group and 46.3 years (range, 21Y76 y) for the malignant tumor group. The mean age of the malignant tumor group was 15 years older than the mean age of the benign tumor group. However, the difference in the mean age between the malignant and benign tumor groups was not statistically significant (P = 0.162, P > 0.05). Pleomorphic adenoma was the most common benign tumor, accounting for 73% (8 patients) of benign tumors and 30% of all patients. Myoepithelioma was the second most common benign tumor (27%, 3 patients). Mucoepidermoid carcinoma and adenoid cystic carcinoma were the most common malignant MSGT. Both of them had an equal frequency of 50% (6 patients). The common symptom of the patients was a painless mass in the mouth located on the palate (17 patients, 73%). Three patients (13%) complained of pain. The mean duration time of symptoms was 2 to 36 months and the mean time was 13 months. The mean time was 9 months for the malignant tumor group and 17 months for the benign tumor group. Locations of the tumors were the soft palate in 7 patients, the hard palate in 13 patients, the retromolar area in 2 patients, and the gingivobuccal mucosa in 1 patient. Table 1 shows the histopathologic diagnoses, demographic data, and tumor location of the subjects. The preoperative imaging was performed on all patients. Computed tomography (CT) was the most common preoperative diagnostic procedure that was performed on all patients. Fine-needle aspiration biopsy (FNAB) was performed on 6 patients, and FFNAB was accurate in all of them: 3 pleomorphic adenoma, 2 adenoid cystic carcinomas, and 1 myoepithelioma.
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DISCUSSION Salivary gland neoplasms constitute less than 1% of all tumors and 3% to 5% of all head and neck neoplasms. Minor salivary gland tumors are uncommon and comprise 15% to 20% of all salivary gland neoplasms. The most common tumor of MSGTs is pleomorphic adenoma.1,7Y11 The most common malignant neoplasms are mucoepidermoid carcinoma and adenoid cystic carcinoma.12 In our study, pleomorphic adenoma was the most common tumor type (30%) and the most common benign tumor. The most common malignant tumors were mucoepidermoid carcinoma (26%) and adenoid cystic carcinoma (26%). These data are closely related to that of other studies. The frequency of benign and malignant MSGTs is a controversial issue according to the literature. Most studies reveal that benign tumors are more common than malignant tumors.1,3,7,11 However, several studies report that malignant tumors were seen more frequently than benign tumors.9,10,13 In this study, benign tumors (52%) were slightly higher than malignant tumors (48%). Minor salivary gland tumors may occur in the oral cavity, paranasal sinuses, nasopharynx, and larynx. The most common location of MSGTs is the oral cavity and the palate is the most common place for MSGTs. It was reported in different studies that 40% to 80% of MSGTs originated from the palate.1,3,7,10 Similarly, in the current study, the palate was the most common site for all MSGTs with the rate of 91% ( 21 subjects in 23). In the benign tumor group, the hard palate was the site in 8 subjects and the soft palate was the site in 3 subjects. In the malignant tumor group, the hard palate was the site in 7 subjects and the soft palate was the site in 3 subjects. Most studies have shown that MSGTs are more common in females than in males.7,8 However, it was found in the current study that MSGTs were more common in men (70%) than in women (30%). This difference can be a result of racial factors. Minor salivary gland tumors can occur at any age, but the maximum incidence is in the fourth decade of life for benign tumors and in the fifth decade of life for malignant tumors. However, some authors have reported that the peak incidence occurs in the period between the fifth and seventh decades of life.14 In this study, the peak incidence of MSGTs was in the fourth decade, ranging from 16 to 75 years. For malignant tumors, the peak incidence was in the sixth decade, ranging from 21 to 76 years. It has been reported that the difference in the mean appearing age of MSGTs among the patients with benign and malignant tumors ranged from 3.4 to 13.3 years.9 Although several of these studies * 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
& Volume 25, Number 3, May 2014
revealed a statistically significant difference in the mean age, many studies revealed no statistically significant difference.7,9Y11 In this study, there was no statistically significant difference between the mean age of benign and malignant MSGTs (P = 0.073 and P < 0.05). Signs and symptoms depend on the location and size of tumor.15 Patients commonly complain of a painless mass in the palate. Pain, paresthesia, dysphagia, speech impairment, and referred otalgia may also occur. In the current study, we found that a painless mass in the palate was the most common presentation (17 patients, 73%). The mean time of symptoms before the treatment was averagely 13 months, ranging from 2 to 36 months. It is very difficult to distinguish benign MSGTs from malignant MSGTs only through a physical examination. Preoperative imaging, incisional biopsy, or FNAB must be done preoperatively in planning the surgery. In the literature, it is stated that histopathologic correlation was increased when preoperative investigations such as FNAB and CT were used.16,17 In this study, preoperatively, CT and FNAB were used for the investigation. Treatment of oral cavity MSGTs includes complete surgical excision. Benign neoplasms can be treated solely through surgical excision. Pleomorphic adenomas must be completely excised with an adequate margin. Incomplete excision causes recurrence of tumor and difficulty in future operations.18 Complete surgical excision was performed on all benign tumors, and no recurrence was seen in the postoperative period in our patients. Treatment of malignant tumors may require palatectomy for palatal lesions, removal of the buccinator muscle for cheek lesions, and removal of the maxilla or mandible if they are involved.18 When an extended resection is performed, reconstructive surgery may be needed to be performed. In our series of patients, reconstructive surgery was required in 8 patients with malignant tumors and in 1 patient with benign tumor. Reconstruction by local flap was performed on 3 of 9 patients and by obturator prosthesis in 6 of 9 patients. Frozen section biopsy was used during the surgery, and a subsequent resection of tumor was necessary to 4 of 12 patients (33%) with malignant tumors because of a positive surgical margin. Neck dissections are recommended for clinically positive necks and for patients with high-grade mucoepidermoid carcinoma. Neck dissection was not performed because our patients had N0 neck and had no high-grade mucoepidermoid carcinoma. Malignant tumors can be treated through radiotherapy and chemotherapy in addition to surgery. The patients were followed up periodically in terms of local recurrence. In our series, the mean postoperative follow-up time was 5 years. There is a limited number of studies examining only MSGTs. Furthermore, there is a great histopathologic variability in salivary gland tumors according to classifications of the World Health Organization.19 Management of other types of salivary neoplasms that were not mentioned in our study is more challenging because of their relative infrequency, inconsistent classification, and variable biologic behavior.20,21 A better understanding of the histogenesis of neoplasms of the salivary glands by further studies about ultrastructural and molecular analysis may provide significant information about their behavior and therapeutic response.22
CONCLUSIONS Minor salivary gland tumors are uncommon and a relatively small group in salivary gland tumors. Hence, studies focusing on minor salivary gland tumors are limited. Our study is important to light the way for clinical presentation, treatment, and the outcomes in patients with MSGTs. Malignant and benign MSGTs were found almost equally in our study. Surgical excision with safety margin is suffi-
Brief Clinical Studies
cient in most of the patients especially in benign tumors. There is a necessity for studies in larger patient series related with MSGTs.
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* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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