A clinical analysis of nine new pediatric and adolescent cases of benign minor salivary gland neoplasms and a review of the literature

Journal of Medical Case Reports

Robert B Brannon¹  Priyanshi Ritwik² 

[1] Division of Oral and Maxillofacial Pathology, LSU School of Dentistry, 1100 Florida Avenue, New Orleans, LA, 70119, USA;Department of Pediatric Dentistry, LSU School of Dentistry, 1100 Florida Avenue, New Orleans, LA, 70119, USA

关键词: Pediatric;    Benign tumors;    Minor salivary gland;    Benign tumors;   

Others  :  1195454
DOI  :  10.1186/1752-1947-6-287

 received in 2011-10-24, accepted in 2012-03-02,  发布年份 2012

【 摘 要 】

Introduction

Minor salivary gland neoplasms of epithelial origin are rare in children and adolescents and most are not well documented, except for a few small series and case reports. This study represents a retrospective clinical analysis of nine cases of benign epithelial salivary gland neoplasms accessioned over a 35-year period at the Louisiana State University School of Dentistry and combines the data with well-documented cases from the English-language literature.

Methods

A retrospective clinical analysis of nine cases of benign epithelial salivary gland neoplasms was performed over a 35-year period at the Louisiana State University School of Dentistry and combined with data of well-documented cases from the English-language literature.

Results

The nine benign salivary gland neoplasms in patients aged 19 months to 18 years accounted for 2.3% of the Louisiana State University School of Dentistry accessioned salivary gland tumors. These nine cases comprised eight pleomorphic adenomas and one cystadenoma. There were 40 cases in the literature, of which 34 were pleomorphic adenomas. Combining the data for the 42 pleomorphic adenomas resulted in a mean age of 12 years with a 2.8:1 female predilection. The hard palate and/or soft palate were the most common site (69.1%). The average duration and size was 2.1 years and 2.4cm, respectively. Bone involvement occurred in seven cases. Wide local excision was the treatment most often employed. Cases followed for two years or more had a recurrence rate of 13.0%. The remaining seven neoplasms in the combined data comprised myoepithelioma, cystadenoma and sialadenoma papilliferum.

Conclusions

A relatively long duration (2 years) of a submucosal mass in a minor salivary gland-bearing area with or without bone involvement occurring in a child or adolescent should raise the question of a possible salivary gland neoplasm. A pleomorphic adenoma is the most common benign salivary gland neoplasm in the first and second decade of life. Complete surgical excision affords the best chance of preventing recurrence for pleomorphic adenomas. The recurrence rate of pleomorphic adenomas with two or more years follow-up is 13.0%. Other types of minor salivary gland neoplasms are exceedingly rare and therefore data is sparse, precluding any valid conclusions.

【 授权许可】

   
2012 Ritwik and Brannon; licensee BioMed Central Ltd.

【 图 表 】

Figure 1.

【 参考文献 】

• [1]Luna MA, Batsakis JG, el-Naggar AK: Salivary gland tumors in children. Ann Otol Rhinol Laryngol 1991, 100:869-871.
• [2]Flaitz CM: Mucoepidermoid carcinoma of the palate in a child. Pediatr Dent 2000, 22:292-293.
• [3]Byars LT, Ackerman LV, Peacock E: Tumors of salivary gland origin in children: a clinical pathologic appraisal of 24 cases. Ann Surg 1957, 146:40-51.
• [4]Crawford WH Jr, Guernsey LH: Pleomorphic adenoma of the palate: report of a case. Oral Surg Oral Med Oral Pathol 1967, 23:116-126.
• [5]Galich R: Salivary gland neoplasms in childhood. Arch Otolaryngol 1969, 89:878-882.
• [6]Buehrle R, Friedberg J: Mixed salivary gland tumor of the palate in a child. Arch Otolaryngol 1972, 96:163-164.
• [7]Budnick SD: Minor-salivary-gland tumors in children. ASDC J Dent Child 1982, 49:44-47.
• [8]Yamamoto H, Fukumoto M, Yamaguchi F, Sakata K, Oikawa T: Pleomorphic adenoma of the buccal gland in a child. Int J Oral Maxillofac Surg 1986, 15:474-477.
• [9]McIlveen LP, Sharp HK, Schuman NJ: Pleomorphic adenoma of a minor salivary gland: report of a case. Quintessence Int 1987, 18:211-213.
• [10]Lack EE, Upton MP: Histopathologic review of salivary gland tumors in childhood. Arch Otolaryngol Head Neck Surg 1988, 114:898-906.
• [11]Rogers TR, Johnson JV, Newland JR: Pleomorphic adenoma of the anterior tongue in a 12-year-old girl. J Oral Maxillofac Surg 1989, 47:89-90.
• [12]Fonseca I, Martins AG, Soares J: Epithelial salivary gland tumors of children and adolescents in southern Portugal: a clinicopathologic study of twenty-four cases. Oral Surg Oral Med Oral Pathol 1991, 72:696-701.
• [13]Austin JR, Crockett DM: Pleomorphic adenoma of the palate in a child. Head Neck 1992, 14:58-61.
• [14]Noghreyan A, Gatot A, Maor E, Fliss DM: Palatal pleomorphic adenoma in a child. J Laryngol Otol 1995, 109:343-345.
• [15]López-Cedrún JL, Gonzalez-Landa G, Birichinaga B: Pleomorphic adenoma of the palate in children: report of a case. Int J Oral Maxillofac Surg 1996, 25:206-207.
• [16]de Courten A, Lombardi T, Samson J: Pleomorphic adenoma of the palate in a child: 9-year follow-up. Int J Oral Maxillofac Surg 1996, 25:293-295.
• [17]Chen YK, Lin LM, Lin CC, Yan YH: Palatal pleomorphic adenoma in a child with osteoid formation: report of case. ASDC J Dent Child 1998, 65:209-211.
• [18]Bayles SW, Todd NW, Muller S, Rabkin D: Pleomorphic adenoma of the pediatric tongue. Otolaryngol Head Neck Surg 1999, 120:934-936.
• [19]Shaaban H, Bruce J, Davenport PJ: Recurrent pleomorphic adenoma of the palate in a child. Br J Plast Surg 2001, 54:245-247.
• [20]Jorge J, Pires FR, Alves FA, Perez DE, Kowalski LP, Lopes MA, Almeila OP: Juvenile intraoral pleomorphic adenoma: report of five cases and review of the literature. Int J Oral Maxillofac Surg 2002, 31:273-275.
• [21]Daniels JS, Ali I, Al Bakri IM, Sumangala B: Pleomorphic adenoma of the palate on children and adolescents: a report of 2 cases and review of the literature. J Oral Maxillofac Surg 2007, 65:541-549.
• [22]Lotufo MA, Junior CA, Mattos JP, Franca CM: Pleomorphic adenoma of the upper lip in a child. J Oral Sci 2008, 50:225-228.
• [23]Dhanuthai K, Sappayatosok K, Kongin K: Pleomorphic adenoma of the palate in a child: a case report. Med Oral Patol Oral Cir Bucal 2009, 14:E73-E75.
• [24]Mahajan D, Khurana N, Setia N: Sialadenoma papilliferum in a young patient: a case report of review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007, 103:e51-e54.
• [25]Kahn LB, Schoub L: Myoepithelioma of the palate. Histochemical and ultrastructural observations. Arch Pathol 1973, 95:209-212.
• [26]Nesland JM, Olafsson J, Sobrinho-Simoes M: Plasmacytoid myoepithelioma of the palate. A case report with ultrastructural findings and review of the literature. J Oral Pathol 1981, 10:14-21.
• [27]Lins JE, Gnepp DR: Myoepithelioma of the palate in a child. Int J Pediatr Otorhinolaryngol 1986, 11:5-13.
• [28]Perez DE, Lopes MA, de Almeida OP, Jorge J, Kowalski LP: Plasmacytoid myoepithelioma of the palate in a child. Int J Pediatr Dent 2007, 17:223-227.
• [29]Eveson JW, Cawson RA: Tumours of the minor (oropharyngeal) salivary glands: a demographic study of 336 cases. J Oral Pathol 1985, 14:500-509.
• [30]Waldron CA, el-Mofty SK, Gnepp DR: Tumors of the intraoral minor salivary glands: a demographic and histologic study of 426 cases. Oral Surg Oral Med Oral Pathol 1988, 66:323-333.
• [31]Kusama K, Iwanari S, Aisaki K, Wada M, Ohtani J, Itoi K, Hanai K, Shimizu K, Komiyama K, Kudo I, Moro I: Intraoral minor salivary gland tumors: a retrospective study of 129 cases. J Nihon Univ Sch Dent 1997, 39:128-132.
• [32]Seifert G, Okabe H, Caselitz J: Epithelial salivary gland tumors in children and adolescents. Analysis of 80 cases (Salivary Gland Register 1965–1984). ORL J Otorhinolaryngol Relat Spec 1986, 48:137-149.
• [33]Mehta D, Willging JP: Pediatric salivary gland lesions. Semin Pediatr Surg 2006, 15:76-84.
• [34]Neville BW, Damm DD, Allen CM, Bouquot JE: Oral and Maxillofacial Pathology. 3rd edition. St. Louis: Saunders Elsevier; 2009:480.
• [35]Chau MNY, Radden BG: A clinical-pathological study of 53 intra-oral pleomorphic adenomas. Int J Oral Maxillofac Surg 1989, 18:158-162.
• [36]Ellis GL, Auclair PL: Benign epithelial neoplasms. In Tumors of the salivary glands. Washington, DC: Armed Forces Institute of Pathology; 1996:57-68. Rosai J (Series Editor) Atlas of tumor pathology: series 3, fascicle 17