J Oral Maxillofac Surg 65:541-549, 2007 Pleomorphic Adenoma of the Palate in Children and Adolescents: A Report of 2 Cases and Review of the Literature John Spencer M. Daniels, FDSRCS (England), FFDRCS (Ireland),* Iqbal Ali, MDS,† Ibrahim Mohammed Al Bakri, BDS,‡ and Balakrishnan Sumangala, MD, DCP§ Benign salivary gland neoplasms are rare in children compared with adults. 1-6 The most common type is pleomorphic adenoma, representing 66.6% to 90% of these tumors in pediatric patients. 1,2,4,5,7 Most benign salivary gland tumors occur in the major salivary glands, 6 with about 70% to 85% of pleomorphic ade- nomas occurring in the parotid gland. 5,7 Pleomorphic adenoma is the most common in- traoral minor salivary gland tumor. The palate is the most commonly affected site; other intraoral sites include the upper and lower lips, buccal mucosa, gingival, and tongue. 8 Pleomorphic adenoma arising in the palate of children and adolescents is unusual. Only about 5% to 10% of pleomorphic adenomas of the minor salivary glands occur in patients age 20 years or younger, with most of these cases occurring in the second decade of life. 9-11 Byars et al, 7 in a 1957 review of 470 cases of salivary gland tumors in patients age 18 years and younger over a 30-year period, reported the first 2 cases of palatal pleomorphic adenoma in children aged 7 years and 9 years. Lack and Upton 3 reviewed 80 patients age 18 years and younger over a 58-year period and found only 1 case of palatal pleomorphic adenoma involving a 10-year-old boy. In a literature review, Noghreyan et al 6 found only 5 reported cases in the same age group and added 1 new case of their own. Jorge et al 12 reported 5 cases of juvenile in- traoral pleomorphic adenoma in patients under age 18 years, including 2 cases of palatal pleomorphic adenoma in an 11-year-old boy and a 17-year-old fe- male. A review of the current English-language literature reveals a total of 14 reported cases of pleomorphic adenoma of the palate in children and adolescents age 18 years and younger ( Table 1). 1,3,4,6,7,9-14 Of these 14 cases, 11 involve patients under age 14 years 1,3,4,6,7,9,12,14 and 3 cases involve patients age 14 to 18 years. 4,11,12 Most studies to date have reported a greater prevalence of minor salivary gland tumors in females than in males, with ratios ranging from 1.2:1 to 1.9:1. 15,16 Here we report 2 more cases of pleomorphic ade- noma of the palate in 2 male patients age 5 years and 16 years, and also review the literature on the inci- dence of this adenoma in children and adolescents. In addition, we discuss strategies for diagnosing and treating pleomorphic adenoma of the palate. Report of Cases CASE 1 A 5-year-old boy was referred to our Department of Oral and Maxillofacial Surgery with a 20-day (or possibly longer) history of painless swelling in the left hard palate. His medical history was unremarkable. Examination revealed a nontender, slightly fluctuant swelling in the left hard palate, measuring 2 1.5 cm, situated adjacent to the upper left deciduous molars, which were not carious. He had no permanent teeth. Plain radiography showed that the permanent teeth were still developing. No bony defect was seen in the palate on the upper occlusal x-ray. The provisional diagnosis was salivary gland neoplasm. An appointment was made for preoperative investigations, including fine-needle biopsy, but the patient failed to attend. Two years later, this patient, now age 7 years, was re- ferred back to the Dental Center with the same complaint of palatal swelling that was now increasing. Examination re- Received from King Khalid Hospital, Najran, Saudi Arabia. *Consultant Oral and Maxillofacial Surgeon and Head, Depart- ment of Oral and Maxillofacial Surgery, Regional Dental Center. †Specialist Oral and Maxillofacial Surgeon, Department of Oral and Maxillofacial Surgery, Regional Dental Center. ‡Director, Regional Dental Center, Department of Endodontics. §Chief, Histopathology and Laboratory Services, Department of Pathology. Address correspondence and reprint requests to Dr Daniels: King Khalid Hospital, PO Box 1120, Najran, Saudi Arabia; e-mail: spencer_daniels@yahoo.co.uk © 2007 American Association of Oral and Maxillofacial Surgeons 0278-2391/07/6503-0027$32.00/0 doi:10.1016/j.joms.2006.08.005 DANIELS ET AL 541