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Sebaceous carcinoma of buccal mucosa
Int. J. Oral MaxilloJac. Surg. 1997," 26:293-294 Printed in Denmark. All rights reserved
Capyright 9 Munksgaard ]997 ]ntemationa]Jouma] of
Oral & MaxillofacialSurgery LTSN 0901-5027
Sebaceous carcinoma of buccal mucosa
Chung-ji Liu 1, Kuo-wei Chang 2, Richard C. S. Chang ~ 1Division of Oral and Maxillofacial Surgery, Dental Department, Mackay Memorial Hospital, Taipei; 2Faculty of Dentistry, National Yang-Ming University, Taipei, Taiwan
Report of a case C. Liu, K. Chang, R. C. S. Chang. Sebaceous carcinoma o f buccal mucosa. Report o f a case. Int. J. Oral Maxillofac. Surg. 1997; 26: 293-294. 9 M u n k s g a a r d , 1997 Abstract. A case of sebaceous c a r c i n o m a arising in the buccal m u c o s a is
Accepted for publication 17 January 1997
described.
Fordyce's spots represent ectopic sebaceous glands in the oral cavity. They are c o m m o n l y f o u n d in the buccal mucosa, upper lip, retromolar trigone, anterior t o n s i l l a r pillar, soft palate a n d gingiva. N e a r l y 80% of adults have these spots s,7. A l t h o u g h the incidence of Fordyce's spots in the oral m u c o s a is high, i n t r a o r a l sebaceous neoplasms are very rare. Less t h a n ten cases of neoplasms arising from sebaceous glands, including only one case o f carc i n o m a in the oral cavity, have been described i n , the English-language literature2~'6'sv 1~ A sebaceous carcin o m a presenting in the buccal m u c o s a is described.
Case report A 68-year-old man visited our department chiefly complaining of a rapidly-growing, ulcerative lesion in the right buccal mucosa, which had been present for about three months. The patient had been a heavy smoker for many years. There were no palpable lymph nodes in the neck. Intraoral examination revealed a tender punch-out lesion in the right buccal mucosa adjacent to the mandibular molar (Fig. 1). The lesion was approximately 2.5 cm in diameter. The margin of the mass was reddish but not folded. The remaining oral mucosa was intact and scattered foci of Fordyce's spots were seen in the buccal mucosa. An incisional biopsy revealed a carcinoma, nature
Key words: sebaceous carcinoma; carcinoma; oncology.
to be determined. The patient was admitted, and under general anesthesia the tumor was excised with a 1-cm margin of healthy tissue. The defect was covered with a buccal fat pad. The postoperative course was uneventful and he was seen for regular follow up. During the three-year follow-up period, there was no evidence of recurrence or distant metastasis of the disease.
Histopathology The surgical specimen measured 4•215 cm. A 2.5•215 cm well circumscribed indurated lesion with surface ulceration was noted. Microscopically, the invasive tumor was located mainly in the submucosa and, except in the area of ulceration, the overlying epithelium appeared intact (Fig. 2). Acute and chronic inflammatory cellular infiltration was present in the stroma between the tumor and the overlying epithelium. There was no evidence that the tumor was linked with the overlying intact oral epithelium. The tumor exhibited an infiltrative growth with involvement of the superficial muscular layer of the buccal wall. The neoplasm was composed chiefly of closely packed, basaloid epithelial cell nests with lipidization of variable extent. Accumulation of vacuolated cells, organized into a characteristic sebaceous nodule, was prominent. Those vacuolated cells exhibited positive reaction for Oil-Red-O stain. The basaloid cells exhibited cellular atypia including pleomorphism, increased mitotic activity, vesicular nuclei and giant nucleoli in some cells.
The nuclei of the vacuolated cells were eccentric with little pleomorphism. No dysplastic lesions were noted in the rest of the Fordyce's spots within the specimen. The pathologic diagnosis was sebaceous carcinoma.
Discussion Sebaceous c a r c i n o m a is an u n c o m m o n m a l i g n a n c y which mainly occurs on the eyelid 9. E x t r a o c u l a r sebaceous carc i n o m a can occur o n any b o d y surface t h a t c o n t a i n s sebaceous glands. N e a r l y 80% o f adults have sebaceous glands in the oral cavity, b u t only one case of i n t r a o r a l sebaceous c a r c i n o m a has been d o c u m e n t e d 2. The incidence of i n t r a o r a l sebaceous c a r c i n o m a is extremely low, but it can occur in any region t h a t c o n t a i n s n o r m a l or ectopic sebaceous glands. T h e differential diagnosis of sebaceous m a l i g n a n c y should be m a d e with caution. It is i m p o r t a n t to differentiate sebaceous c a r c i n o m a f r o m neoplasms, including basal cell c a r c i n o m a with sebaceous differentiation, s q u a m ous cell c a r c i n o m a with hydrophilic swelling a n d m u c o e p i d e r m o i d carcin o m a . In the case described, there was a b a s a l o i d cytological appearance, however, there was n o peripheral palisa d i n g of the cell nests a n d n o separating artifact lying between the mucin-
294
Liu et al.
Fig. 1. Punch-out lesion in right buccal mucosa measuring approx. 2.5 cm in diameter.
ous stroma and the t u m o r nests that could denote a diagnosis o f basal cell carcinoma. A diagnosis o f intraoral basal cell carcinoma (with sebaceous differentiati6n) was, therefore, excluded. Due to the lack o f interconnection between the t u m o r nests and the overlying epithelium, and the absence o f intracellular mucin in the vacuolated cytoplasm, a carcinoma arising from oral epithelium or salivary epithelium was eliminated. A review o f 91 cases of extraocular sebaceous carcinomas indicated that sebaceous carcinoma is a locally aggressive malignancy, which often invades surrounding tissue with occasional metastasis L. A wide excision o f the sebaceous carcinoma is, therefore, the treatment o f choice.
Fig. 2. Invasive carcinoma composed chiefly of basaloid cells and clusters of vacuolated cells in the submucosa (HE • insert shows that basaloid cells are characterized by hyperchromatism, pleomorphism, and high mitotic activity (HE •
References 1. BMLET JW, ZIMMERMAN MC, ARNSTEIN DR WOLLMAN JS, MICK~L RA. Sebaceous carcinoma of the head and neck. Case report and literature review. Arch Otolaryngol Head Neck Surg 1992: 118: 1245-9. 2. DAMMDID, O'CONNOR WN, WHITE DK, DRUMMOND JR MORROW LW, KENADY DE. Intraoral sebaceous carcinoma, Oral Surg Oral Med Oral Pathol 199l: 72: 709--I 1. 3. EPKER BN, HENNV FA. Intra-oral sebaceous gland adenoma. Cancer 1971:27: 987 9. 4. FERGUSON JW, GEARY CP, MACALISTER AD. Sebaceous cell adenoma: rare intraoral occurrence of a tumor which is a frequent marker of Torre's syndrome. Pathology 1987: 19:204 8. 5. GuIDUCCl AA. Ectopic sebaceous glands. Dermatol 1962: 125:44 63. 6. LIPANIC, WOYTASttJJ, GREENEGW. Seb-
aceous adenoma of the oral cavity. J Oral Maxillofac Surg 1983: 41: 56-60. 7. MIL~S AEW. Sebaceous glands in the lip and cheek mucosa of man. Br Dent J 1958: 105: 235-48. 8. MILLER AS, McCREA MW. Sebaceous gland adenoma of the buccal mucosa: report of case. J Oral Surg 1968: 26:593 5. 9. NELSON BR, HAMLET KR, GILLARD M, RAILAN D, JOHNSONTM, ARBOR A. Sebaceous carcinoma. J Am Acad Dermatol 1995: 33: 1-t5. 10. ORLIANAI, SALMANL, REDDIT, YAMANE GM, CHAUDrIRY AR Sebaceous adenoma of the oral mucosa. J Oral Med 1987: 42:38 9. Address: Richard C. S. Chang, DDS, M S Faculty of Dentistry National Yang-Ming University Taipei Taiwan 112
Capyright 9 Munksgaard ]997 ]ntemationa]Jouma] of
Oral & MaxillofacialSurgery LTSN 0901-5027
Sebaceous carcinoma of buccal mucosa
Chung-ji Liu 1, Kuo-wei Chang 2, Richard C. S. Chang ~ 1Division of Oral and Maxillofacial Surgery, Dental Department, Mackay Memorial Hospital, Taipei; 2Faculty of Dentistry, National Yang-Ming University, Taipei, Taiwan
Report of a case C. Liu, K. Chang, R. C. S. Chang. Sebaceous carcinoma o f buccal mucosa. Report o f a case. Int. J. Oral Maxillofac. Surg. 1997; 26: 293-294. 9 M u n k s g a a r d , 1997 Abstract. A case of sebaceous c a r c i n o m a arising in the buccal m u c o s a is
Accepted for publication 17 January 1997
described.
Fordyce's spots represent ectopic sebaceous glands in the oral cavity. They are c o m m o n l y f o u n d in the buccal mucosa, upper lip, retromolar trigone, anterior t o n s i l l a r pillar, soft palate a n d gingiva. N e a r l y 80% of adults have these spots s,7. A l t h o u g h the incidence of Fordyce's spots in the oral m u c o s a is high, i n t r a o r a l sebaceous neoplasms are very rare. Less t h a n ten cases of neoplasms arising from sebaceous glands, including only one case o f carc i n o m a in the oral cavity, have been described i n , the English-language literature2~'6'sv 1~ A sebaceous carcin o m a presenting in the buccal m u c o s a is described.
Case report A 68-year-old man visited our department chiefly complaining of a rapidly-growing, ulcerative lesion in the right buccal mucosa, which had been present for about three months. The patient had been a heavy smoker for many years. There were no palpable lymph nodes in the neck. Intraoral examination revealed a tender punch-out lesion in the right buccal mucosa adjacent to the mandibular molar (Fig. 1). The lesion was approximately 2.5 cm in diameter. The margin of the mass was reddish but not folded. The remaining oral mucosa was intact and scattered foci of Fordyce's spots were seen in the buccal mucosa. An incisional biopsy revealed a carcinoma, nature
Key words: sebaceous carcinoma; carcinoma; oncology.
to be determined. The patient was admitted, and under general anesthesia the tumor was excised with a 1-cm margin of healthy tissue. The defect was covered with a buccal fat pad. The postoperative course was uneventful and he was seen for regular follow up. During the three-year follow-up period, there was no evidence of recurrence or distant metastasis of the disease.
Histopathology The surgical specimen measured 4•215 cm. A 2.5•215 cm well circumscribed indurated lesion with surface ulceration was noted. Microscopically, the invasive tumor was located mainly in the submucosa and, except in the area of ulceration, the overlying epithelium appeared intact (Fig. 2). Acute and chronic inflammatory cellular infiltration was present in the stroma between the tumor and the overlying epithelium. There was no evidence that the tumor was linked with the overlying intact oral epithelium. The tumor exhibited an infiltrative growth with involvement of the superficial muscular layer of the buccal wall. The neoplasm was composed chiefly of closely packed, basaloid epithelial cell nests with lipidization of variable extent. Accumulation of vacuolated cells, organized into a characteristic sebaceous nodule, was prominent. Those vacuolated cells exhibited positive reaction for Oil-Red-O stain. The basaloid cells exhibited cellular atypia including pleomorphism, increased mitotic activity, vesicular nuclei and giant nucleoli in some cells.
The nuclei of the vacuolated cells were eccentric with little pleomorphism. No dysplastic lesions were noted in the rest of the Fordyce's spots within the specimen. The pathologic diagnosis was sebaceous carcinoma.
Discussion Sebaceous c a r c i n o m a is an u n c o m m o n m a l i g n a n c y which mainly occurs on the eyelid 9. E x t r a o c u l a r sebaceous carc i n o m a can occur o n any b o d y surface t h a t c o n t a i n s sebaceous glands. N e a r l y 80% o f adults have sebaceous glands in the oral cavity, b u t only one case of i n t r a o r a l sebaceous c a r c i n o m a has been d o c u m e n t e d 2. The incidence of i n t r a o r a l sebaceous c a r c i n o m a is extremely low, but it can occur in any region t h a t c o n t a i n s n o r m a l or ectopic sebaceous glands. T h e differential diagnosis of sebaceous m a l i g n a n c y should be m a d e with caution. It is i m p o r t a n t to differentiate sebaceous c a r c i n o m a f r o m neoplasms, including basal cell c a r c i n o m a with sebaceous differentiation, s q u a m ous cell c a r c i n o m a with hydrophilic swelling a n d m u c o e p i d e r m o i d carcin o m a . In the case described, there was a b a s a l o i d cytological appearance, however, there was n o peripheral palisa d i n g of the cell nests a n d n o separating artifact lying between the mucin-
294
Liu et al.
Fig. 1. Punch-out lesion in right buccal mucosa measuring approx. 2.5 cm in diameter.
ous stroma and the t u m o r nests that could denote a diagnosis o f basal cell carcinoma. A diagnosis o f intraoral basal cell carcinoma (with sebaceous differentiati6n) was, therefore, excluded. Due to the lack o f interconnection between the t u m o r nests and the overlying epithelium, and the absence o f intracellular mucin in the vacuolated cytoplasm, a carcinoma arising from oral epithelium or salivary epithelium was eliminated. A review o f 91 cases of extraocular sebaceous carcinomas indicated that sebaceous carcinoma is a locally aggressive malignancy, which often invades surrounding tissue with occasional metastasis L. A wide excision o f the sebaceous carcinoma is, therefore, the treatment o f choice.
Fig. 2. Invasive carcinoma composed chiefly of basaloid cells and clusters of vacuolated cells in the submucosa (HE • insert shows that basaloid cells are characterized by hyperchromatism, pleomorphism, and high mitotic activity (HE •
References 1. BMLET JW, ZIMMERMAN MC, ARNSTEIN DR WOLLMAN JS, MICK~L RA. Sebaceous carcinoma of the head and neck. Case report and literature review. Arch Otolaryngol Head Neck Surg 1992: 118: 1245-9. 2. DAMMDID, O'CONNOR WN, WHITE DK, DRUMMOND JR MORROW LW, KENADY DE. Intraoral sebaceous carcinoma, Oral Surg Oral Med Oral Pathol 199l: 72: 709--I 1. 3. EPKER BN, HENNV FA. Intra-oral sebaceous gland adenoma. Cancer 1971:27: 987 9. 4. FERGUSON JW, GEARY CP, MACALISTER AD. Sebaceous cell adenoma: rare intraoral occurrence of a tumor which is a frequent marker of Torre's syndrome. Pathology 1987: 19:204 8. 5. GuIDUCCl AA. Ectopic sebaceous glands. Dermatol 1962: 125:44 63. 6. LIPANIC, WOYTASttJJ, GREENEGW. Seb-
aceous adenoma of the oral cavity. J Oral Maxillofac Surg 1983: 41: 56-60. 7. MIL~S AEW. Sebaceous glands in the lip and cheek mucosa of man. Br Dent J 1958: 105: 235-48. 8. MILLER AS, McCREA MW. Sebaceous gland adenoma of the buccal mucosa: report of case. J Oral Surg 1968: 26:593 5. 9. NELSON BR, HAMLET KR, GILLARD M, RAILAN D, JOHNSONTM, ARBOR A. Sebaceous carcinoma. J Am Acad Dermatol 1995: 33: 1-t5. 10. ORLIANAI, SALMANL, REDDIT, YAMANE GM, CHAUDrIRY AR Sebaceous adenoma of the oral mucosa. J Oral Med 1987: 42:38 9. Address: Richard C. S. Chang, DDS, M S Faculty of Dentistry National Yang-Ming University Taipei Taiwan 112