UPPER I.IP NECROSIS
302 Discussion
2. Wilkinson IMS: Giant cell arteritis.
Isolated biopsy of a temporal artery may not always confirm the diagnosis of giant-cell arteritis because the disease is often patchy in distribution and the amount of elastic tissue in the vessel wall can vary. Cases of cranial arteritis have been described without an elevated ESR, but the absence of this finding should case extreme doubt on the diagnosis. The diagnosis of giant-cell arteritis in this case was made on the basis of the clinical history, which included the occurrence of sudden blindness and an elevated ESR, and histologic confirmation of the disease at necropsy. It is the first reported case showing tissue necrosis of the upper lip and part of the anterior maxilla.
5.
6.
7. 8. 9. IO. Il.
References
12.
I. Dixon A. St J.. Beardwell C, Kay A. et al: Polymyalgia rheumatica and temporal 25:203. 1966 J Oral Maxdlofac
arteritis.
Ann Rheum Dis
13.
Br J Hosp Med 15: 154. 1976 Wintrobe MM. et al feds): Harrison’s Principles of Internal Medicine, 7th ed. New York. McGraw Hill. 1977. p 394 Missen GAK: Gangrene of the tongue. Br Med J 1.11393. 1961 Henderson AH: Tongue pain with giant cell arteritis. Br Med J 4:337, 1967 Grahame R. Bluestone R, Holt PJ: Recurrent blanching of the tongue due to giant cell arteritis. Ann Intern Med 69:781. 1968 Bearsley BF, MacDonald JG: “Temporal arteritis resulting in infected gangrene of the tongue. Br Med J I : I 151, 1961 Reed C, Inglis MJ: Acute massive gangrene of the tongue. Br Med J 2:575, 1965 Davis AE, Davis TP: Gangrene of the tongue caused by temporal arteritis. Med J Aust 2:459, 1966 Meadows SP: Temporal or giant cell arteritis. Proc R Sot Med 59:329. 1966 Allan P: Giant cell arteritis presenting with necrosis of the tongue. Br J Oral Surg 18:166. 1980 Hicks K, Lee FL: Necrosis of the tongue secondary to cranial arteritis. Br J Oral Surg 18: 166, 1980 Browne WG: Oral necrosis accompanying giant cell arteritis. J Oral Surg 40:450, 1982
Surg
43302-304.1985
Sialadenoma
Papilliferum
KENNETH D. BASS, DDS, AND B. J. COSENTINO,
Sialadenoma papilliferum is a rare benign tumor of salivary gland origin first reported in the literature by Abrams and Fink in 1969.’ These authors described two lesions in the parotid and palatal salivary glands as being histomorphologically similar to the syringadenoma papilliferum of the sweat gland.2 A recent review of the literature revealed a series of 11 previously reported cases of sialadenoma papilliferum. Nasu et al.‘, in their review of nine cases, reported that the majority of the tumors had developed in men older than 40 years of age, with a palatal location being most common. They described the lesions grossly to be exophytic, papillary, or verrucous proliferations that were less than 1 cm in diameter. Complete surgical excision was believed to be curative, although follow-up periods are not yet adequate to reach a final conclusion in this area. In 1978, Solomon et a1.4 reported a recurrent squamous papiIlary tumor of the palate with regional lymph node metastases that was perhaps a maligReceived from the Department of Oral and Maxillofacial Surgery at Baylor College of Dentistry. Dallas, Texas. Address correspondence and reprint requests to Dr. Bass: Department of Oral and Maxillofacial Surgery, Baylor College of Dentistry. 3302 Gaston Avenue, Dallas, TX 75246.
nant variety summarizes and clinical ported cases
DDS
of sialadenoma papilliforum. Table 1 the available pertinent demographic information on the 11 previously reand the present case. Report of a Case
A 76-year-old, obese, white female was seen in the oral surgery clinic desiring to have her remaining teeth extracted. She did not report any other oral disease. The medical history indicated that she was under treatment for congestive heart failure, arteriosclerotic heart disease, hypertension, and diabetes mellitus. Chronic medical problems included hepatic cirrhosis and mild anemia. Medications included tolbutamide, digoxin, and furosemide. She was a chronic cigarette smoker and had consumed three to five alcoholic drinks per day for the past 3.5 years. Clinical examination demonstrated gross dental neglect, with multiple nonrestorable caries, Class II mobility in many of the remaining teeth, marginal gingivitis, and a pedunculated, nontender, exophytic. red lesion approximately I x 1 cm on the left fauciai pillar [Fig. I). Periapical radiographs revealed extensive horizontal bone loss with no additional significant pathosis noted on panoramic films. The patient’s temperature was 98.6”F; blood pressure, 160/70 mm Hg; pulse, 88 beatsimin and regular: and respirations, 22/min. The patient was 65 inches tall and weighed 180 pounds. An excisional biopsy was performed using local anes-
BASS AND COSENTINO
FIGURE 1 (left, top). Clinical view of sialadenoma papilliferum of the left faucial pillar. FIGURE 2. (right. fop). Low-power microscopic view of lesion showing papillary projections and narrow stalk-like connective tissue base. Hematoxylin and eosin. x 2.5. FIGURE 3. (lefr, bottom). High-power microscopic view of lesion showing thickened pseudostratified columnar epithelium surrounding a glandular-type tissue stroma. Hematoxylin and eosin, x400.
thesia, and the specimen evaluation.
Histologic
was submitted
for histologic
Findings
Gross Description. The specimen was 1.0 x 1.0 x 0.6 cm of reddish tissue with papillary projections. Histologic sections showed Microscopic Description.
a proliferative epithelial neoplasm with fragmentation. The lesion was exophytic but did not appear to arise from the surface stratified squamous epithelium. The lesional epithelium was a thick, pseudostratified, columnar type with a brush border and a few clear cells arranged in a papilliferous pattern around a thin stalk of connective tissue. Although several areas of subepithelial connective
tissue showed an atypical cytology, no actual invasion or disruption of the basement membrane was seen (Figs. 2, 31. The diagnosis was sialadenoma papilliferum. Follow up during the immediate two-month postoperative period revealed a well-healed surgical site without evidence of recurrence. Subsequent information was unavailable as the patient died from other causes. Discussion
Sialadenoma papilliferum is an exophytic, papillary lesion of salivary-gland origin. It is apparently slow growing and asymptomatic. The microscopic
304
SIALADENOMA
Table 1. Reported Cases of Sialadenoma Papilliferum Year
Age, Sex, Race
1969
71, M, B
1969
57, M. B
Cracker et al5
1972
71. M, W
Jensen and Reingold Drummond et al.’
1973 1978
48, M, W 71. M, W
Freedman and Lummermans
1978
68, M, -
Solomon et al4
1978 1978
68, M, 62, M, B
Castigliano and Gold9
1954
58, M, W
McCoy and Eckert’O
1980
77, F, W
Nasu et al3 Bass and Cosentino
1981 1985
61, M, 76, F, W
Abrams and Finck’
Location Right parotid gland Right hard palate Left buccal mucosa Hard palate Left retromolar area Hard palate Hard palate Right soft palate Right hard palate Right buccal mucosa Hard palate Left faucial pillar
anatomy of the lesion bears out the clinical appearance. Although this case presented with some atypical connective-tissue features, the lesion was benign in behavior. However, a malignant counterpart to sialadenoma papilliferum has been suggested by Solomon et aL4 Nasu et a1.3 have indicated a controversy over histogenesis, i.e., whether the lesion is hyperplastic or neoplastic. The tumor’s small size, encapsulation, and association with inflammatory infiltrations indicate a primarily hyperplastic origin, while the double-layered ductal epithelium, inward ductal epithelial proliferation, and cytologic differences between tumor cells and simple hyperplastic duct cells favor a neoplastic origin. Abrams and Finck’ considered the cell of or-
PAPILLIFERUM
igin to be pleuripotential myoepithelial cells, and Nasu et a1.3 believed that a ductal cell origin was more probable considering the microscopic appearance of their specimens. Regardless of theories of histogenesis and cellular origin, the sialadenoma papilliferum may simulate a malignant process and require careful histologic differentiation. The differential diagnosis should include squamous papilloma, verrucous carcinoma, papillary cystadenoma, and verruca dyskeratoma. l’*lZ Summary
The twelfth case of intraoral sialadenoma papilliferum is reported. The tumor appeared on the left faucial pillar of a 76-year-old white woman and is the second lesion reported to have occurred in a woman. References 1. Abrams AM, Finck FM: Sialadenoma papilliferum. Cancer 24:1057, 1969 2. Helwig EB, Hackney VC: Syringadenoma papilliferum. Arch Dermatol 71:361, 1955 3. Nasu M, Takagi M. Ishikawa G: Sialadenoma papilliferum: report of case. J Oral Surg 39:367, 1981 4. Solomon MP, Rosen Y, Alfonso A: Intraoral papillary squamous cell tumor of the soft palate with features of sialadenoma papilliferum. Cancer 42: 1858, 1978 5. Cracker DJ, Christ TF, Cavalaris CJ: Sialadenoma papilliferum: report of case. J Oral Surg 30:520. 1972 6. Jensen JL, Reingold IM: Sialadenoma papilliferum of the oral cavity. Oral Surg 35:521, 1973 7. Drummond JF, Giansanti JS, Sabes WR. et al: Sialadenoma papilliferum of the Oral Cavity. Oral Surgery 45:72, 1978 8. Freedman PD, Lummerman H: Sialadenoma papilliferum of the oral cavity. Oral Sur 45:88, 1978 9. Castigliano SG, Gold L: Intraductal papilloma of the hard palate. Oral Surg 7:232, 1954 10. McCoy JM, Eckert EF: Sialadenoma papilliferum J Oral Surg 38:691, 1980 11. Whitaker JS, Turner EP: Papillary tumors of the minor salivary glands. J Clin Path01 29:798, 1976 12. Gorlin RJ, Peterson WC: Warty dyskeratoma. Arch Dermatol95:292, 1967