Otolaryngology–Head and Neck Surgery (2007) 136, 672-673 CASE REPORT Sebaceous adenoma of the parotid gland in a 2-year-old male Kevin C. Welch, MD, John C. Papadimitriou, MD, Robert Morales, MD, and Jeffrey S. Wolf, MD, Baltimore, MD S ebaceous adenomas of the major salivary glands are uncommon tumors. Although sebaceous differentiation within the major salivary glands is a frequent histopathologic finding (approximately 20% to 42% of parotid glands1-3), sebaceous neoplasms of salivary gland origin are not. A number of publications review the clinical and histologic features of sebaceous neoplasms, however, these reports focus on adult patients. The finding of these tumors in a pediatric patient is exceedingly rare.4 We present a case of a sebaceous adenoma of the parotid in a 2-year-old male. Our patient is a 2-year-old full-term healthy male who was referred to the University of Maryland Department of Otorhinolaryngology with a 1-year history of a progressively enlarging painless left-sided neck mass. An MRI (Fig 1) revealed a mildly to moderately enhancing heterogenous mass that measured 4.7 ⫻ 3.0 ⫻ 5.2 cm in the vicinity of the left parotid gland. There was no associated lymphadenopathy. Before the referral, he had undergone an open biopsy that was interpreted as a dermoid tumor. Our review of the biopsy specimen favored the diagnosis of a sebaceous neoplasm. Physical examination revealed an otherwise healthy appearing Asian-American male. He had a 5-cm nontender mass in the location of the left parotid gland. The overlying skin was normal in appearance with the exception of the previous biopsy scar. There was no facial nerve weakness on examination. A clinical diagnosis of a sebaceous neoplasm was made, and the patient underwent a left total parotidectomy with facial nerve preservation. An encapsulated mass that occupied the superficial and deep lobes of the parotid gland was identified and removed with preservation of the facial nerve. From the Departments of Otorhinolaryngology (Drs Welch and Wolf); Pathology (Dr Papadimitriou); and Radiology (Dr Morales); University of Maryland School of Medicine Baltimore. Figure 1 Axial T2-weighted MRI demonstrates a well-circumscribed tumor within the region of the left parotid gland. The tumor has heterogenous signal intensity representative of the sebaceous components of this type of tumor. The final pathology report was consistent with a sebaceous adenoma, which is a benign adnexal tumor that is characterized by abundant islands of sebaceous cells admixed in a fibrous stroma surrounded by cystic and ductlike structures5 (Fig 2). These sebaceous cells are noninvasive and fail to demonstrate any significant pleomorphism Reprint requests: Kevin C. Welch, MD, Department of Otorhinolaryngology, 16 S. Eutaw St, Suite 500, Baltimore, MD 21201. E-mail address: [email protected]. 0194-5998/$32.00 © 2007 American Academy of Otolaryngology–Head and Neck Surgery Foundation. All rights reserved. doi:10.1016/j.otohns.2006.08.029 Welch et al Sebaceous adenoma of the parotid gland in a . . . Figure 2 Sebaceous adenoma under magnification; cystic elements of the lesion are lined by both squamous and sebaceous cells. The surrounding stroma is fibrotic (original magnification, ⫻200). or atypia. In a gross examination, sebaceous adenomas appear well-circumscribed, are pale yellow in color, and are rubbery to palpation. The patient had an uneventful postoperative course and was discharged to home on postoperative day four. 673 sule could be identified in the five specimens. The specimens were otherwise characterized by foreign body reaction, histiocytes, fibrosis, and varying degrees of cyst formation. Cawson et al5 reviewed the histology of sebaceous adenomas and noted their preponderance of solid nests of sebaceous glands that were admixed in a fibrous stroma. They also noted squamous metaplasia in their tissue samples. Although their report referred to a sebaceous adenoma that occurred in a minor salivary gland, Izutsu et al6 also describe their findings as well-circumscribed and possessing well-differentiated sebaceous units with ductal formation. The management of sebaceous adenoma consists of excision en toto. With complete excision, recurrence rates would be very low. There is no information with respect to the accuracy of fine needle aspiration of these tumors. CONCLUSION Sebaceous adenomas are benign tumors that are infrequently encountered in adult patients. We presented an extremely rare case of a sebaceous adenoma of the parotid in a pediatric patient. Although other neoplasms are much more frequently encountered in the pediatric population, one should maintain sebaceous adenomas in the differential diagnosis of parotid masses. DISCUSSION Sebaceous adenomas are rare in adult patients and have been reported only once in the pediatric population.4 These adenomas occur most commonly in the parotid gland; however, they have been reported2,6 in the submandibular, sublingual, and minor salivary glands. The largest compilation of information on sebaceous neoplasms is provided by Gnepp and Brannon.2 In their review of the AFIP database, 21 cases of salivary gland sebaceous neoplasms were reported. Five were sebaceous adenomas, nine were sebaceous lymphadenomas, five were sebaceous carcinomas, and two were sebaceous lymphadenocarcinomas. In their series, sebaceous adenomas were found in the parotid gland in three of five cases, the submandibular gland in one case, and the buccal mucosa in another. All tumors were well demarcated or circumscribed; however, no consistent cap- REFERENCES 1. Auclair PL, Ennis GL, Gnepp DR. Other benign neoplasms. In: Auclair PL, Ennis GL, Gnepp DR, editors. Surgical pathology of the salivary glands. Philadelphia: WB Saunders Co.; 1991. p. 252–268. 2. Gnepp DR, Brannon R. Sebaceous neoplasms of salivary gland origin: report of 21 cases. Cancer 1984;53:2155–70. 3. Meza-Chavez L. Sebaceous glands in normal and neoplastic parotid glands possible significance of sebaceous glands in respect to the origin of tumors of the salivary glands. Am J Pathol 1949;25:627. 4. Roth A. Congenital giant sebaceous adenoma of the parotid gland. Arch Anat Cytol Pathol 1985;33(1):55–7. 5. Cawson RA, Gleeson MJ, Eveson JW. Adenomas of salivary glands. In: Pathology and surgery of the salivary glands. Oxford, UK: Isis Medical Media, Ltd.; 1997. p. 108 –9. 6. Izutsu T, Kumamoto H, Kimizuka S, et al. Sebaceous adenoma in the retromolar region: report of a case with a review of the English literature. Int J Oral Maxillofac Surg 2003;32(4):423– 6.
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