Warthin’s tumor Report
of a case
Stephen R. Matteson, and Paul A. Herman, UNIVERSITY
D.D.S.,” Leslie 8. Cutler, D.D.X., Ph.D.,** D.D.S., *++ Parmington and Hartford, Conn.
A case of Warthin’s tumor with an unusual sialographic appearance is reported. The extensive pooling of the contrast media within the tumor was probably the result of communication of the cystic spaces of the tumor with the ductal system of the parotid gland. This case stimulated a re-examination of the incidence of this tumor, with a study of 205 major salivary gland tumors from pathology reports in the files of Hartford Hospital which is affiliated with the University of Connecticut Health Center in Farmington, Connecticut.
arthin’s tumor (papillary cystadenoma lymphomatosum, adenolymphoma) glands. l It was first reported in 1895 by is a distinctive tumor of salivary Hildebrand, who suggested that it was a variant of a congenital neck cyst.* Warthin described the neoplasm in 1927 and thought that it was derived from the Eustachian tube. Warthin’s tumor has been reported to represent 6 per cent of all salivary gland tumors and 10 per cent of all benign major salivary gland neop1asms.4 The tumor occurred most frequently in the fifth or sixth decade, demonstrated a 5 :l male-to-female ratio, and was seen almost exclusively in white populations. *p 5* 6 It has been advanced that “the tumor originates from heterotropic salivary gland tissues entrapped in lymph nodes during embryogenesis.“? Warthin’s tumor has been reported in the lower pole of the parotid region, sometimes within the substance of the gland, but most commonly occupying a position superficial and lateral to the parotid,5* 8 and as an oval, soft, fluctuant, and painless enlargement. *, g Impairment of parotid function usually has not occurred.8l s It is usually a slowly growing lesion which takes several years to reach a size that rarely exceeds 3 cm. Occasionally, the tumor has been reported to have a sudden onset.‘O Seven per cent of these tumors were bilateral, and a
This research was completed with the cooperation of Hartford Hospital, Hartford, Conn. *Assistant Professor, Department of Oral Radiology. **Assistant Professor, Department of Oral Biology. ***Oral Surgeon in private practice in Hartford, Conn.
Matteson and Cutler
1. Lateral no calcifications.
3. Lateral within
and posteroanterior the Warthin’s tumor.
multifocal presentation was not unusual.1’ The tumor was well encapsulated, and has been described histopathologically as epithelial processes projecting into dilated cystic spaces,with these processessupported by lymphoid stroma. Reports of the tumor in minor salivary glands, larynx, buccal mueosa,nasal cavity, lacrimal caruncle, tear sac, palate, and cheek were rare. When reported, they lacked the lymphocytic component and capsule characteristic of the parotid neoplasm.8-I2 Only a few casesof malignant Warthin’s tumor have been reported.‘” CASE
A 45-year-old Caucasian man University of Connecticut Health
was examined Center with
in the Department the chief complaint
of Oral Radiology of the of a swelling in the right
8. Lateral and of the contrast
Fig. (CS), (GC).
4. Left: Histopathologic section (Magnification, x37.5) showing the cystic low columnar epithelium (black arrows), lymphoid stroma (LS), and germinal Right: High-power magnification (x375) showing low columnar epithelium.
cheek. The apple-sized enlargement had been present for 1 month and was centered in the area superficial to the mandibular angle. The swelling was soft, fluctuant, and painless. No other swellings were observed in the head and neck. Radiologic examination revealed a softtissue swelling with no calcifications (Fig. 1). Sialographic examination revealed Lipiodol distributed within the soft-tissue swelling in extensive pooled areas (Fig. 2). The dye was not cleared by the gland when examined 48 hours later (Fig. 3). Following subsequent surgical intervention, histologic examination identified the lesion as a Warthin’s tumor (Fig. 4).
Matteson and Cutler
I. Classification and occurrence of major salivary gland tumors, Hartford Hospital, Hartford, Conn., 1964-1973 Table
Pleomorphic adenoma Papillary cystadenoma lymphomatosum Oxyphilic adenoma Acmar-cell adenoma Lipoma Malignant
106 32 : 2
Malignant pleomorphic adenoma 1 Adenocarcmoma Adenoid c stic carcinoma Acinar-ccl r carcinoma E Miscellaneous carcinoma Mucoepidermoid carcinoma 1; Epidermoid carcinoma 5 Nonspecific malignancy Miscellaneous nonepithelial malignancy :* Metastatic malignancy 3 *Two lymphomas, one melanoma, one case of Kaposi’s recurred in the opposrte parotid gland 1 year later. SURVEY
: :, 1
1 i i
i ii 6
sarcoma of the parotid
To put this tumor into the perspective of all benign and malignant tumors of the major salivary glands, a retrospective review was conducted of pathology reports from the record room files of a large community hospital affiliated with the University of Connecticut Health Center. Tumors of minor salivary glands were excluded from this review. The pathologic diagnosis was classified according to the sign-out diagnosis of the Department of Pathology, Hartford Hospital. Two hundred five casesof major salivary gland neoplasms were reviewed from the years 1964 to 1973. This series of salivary neoplasms is listed in Table I. Specific data regarding Warthin’s tumor are shown in Table II. Parotid sialograms of two of the thirty-two Warthin’s tumor patients were available. The first of these demonstrated a ductal structure that was well opacified and intact. There was displacement and stretching of the smaller ducts, with no evidence of invasion. The second showed a well-filled ductal system which was displaced by a masswith both deep and superficial components. In both cases,the contrast medium was completely evacuated within 5 minutes. Neither of these demonstrated the pooling effect noted in the case reported here. DISCUSSION
The sialographic appearance of Warthin’s tumor has been described as demonstrating the presence of a space-occupying lesion characterized by ductal displacement and regular borders. 14$I5 In the present ease, the Lipiodol was observed as a pooled mass within the gland; the contrast medium was not cleared by the gland 48 hours following sialography. We could find only one report of this pooling effect associated with Warthin’s tumor in the Englishlanguage literature.16 Suzuki and Kawashima16 suggested that this appearance was caused by leakage of the medium into the cystic spaces of the lesion as a
Volume 41 Number 1
II. Warthin’s tumor data obtained from the 1964-1973 Hartford group of salivary neoplasms
Number of Warthin’s tumors Per cent of benign major salivary gland tumors that were Warthin’s Per cent of all major salivary gland tumors that were Warthin’s Per cent of parotid benign tumors that were Warthin’s Race incidence Age
:021 15:9 29.1 100% Caucasian Range: 39 to 80 Average: 6 1.1 years 23 male : 9 female (2.5: 1)
Sex incidence Table
Ill. Reports of Warthin’s tumor incidence
Foote and Frazell’ Wheelockzl Beahrs Butlera et aL2* FrazelP’
Memorial Hospital, New York City. Norrlttisy University,
1930 to 1949
~o;~o~ vmm, Minnesota Memorial Hospital, New York City
1945 to 1954 1964 1939 to 1959
No. of Warthin’s tumors
No. of benign tumors
Per cent of benign tumors that were Warthin’s
541 195 1113
result of secondary inflammation of the ductal epithelium. Inflammation has not been reported as a common feature of Warthin’s tumoP> 4 and was not seen in the case reported here. Because this neoplasm is a tumor of ductal epithelium,l’r l8 the tumor may communicate with the main duct system of the gland, thereby allowing the contrast medium’s entry into the cystic spaces of the neoplasm. Schall19 reviewed the role of nuclear scanning in the evaluation of salivary gland neoplasms. Of fifteen reported Warthin’s tumors, ten demonstrated increased uptake of gsmTc pertechnetate, three an uptake similar to normal salivary gland, and two a decreased uptake. I9 Other reports also indicated a frequent increased uptake of 9amTcpertechnetate by Warthin’s tumor, with other salivary gland tumors not demonstrating this feature.l”’ Ifi* 2oThis suggests that salivary gland nuclear scanning can aid in the preoperative identification of Warthin’s tumor. The Hartford Hospital series reveals that of all benign major salivary gland tumors, there is a high incidence of Warthin’s tumors, with this neoplasm representing 20.7 per cent. Table III lists various reports of the incidence of this tumor. Only the series reported by Butler contains an incidence comparable to the one reported here. The Hartford Hospital patients with Warthin’s tumor were all Caucasians, which is in agreement with previous reports. (The Hartford Hospital serves patients of varied racial backgrounds.) The age at presentation of the thirty-two Warthin’s patients is also in agreement with other reports. The 2.5:1 male predilection is somewhat lower than the usually given 5 :1 ratio. The current report suggests that Warthin’s tumors may be more common than previously reported and that sialography combined with nuclear scanning may be extremely helpful prior to surgical intervention.
Matteson and Cutler
1. Shafer, B., Hine, B., and Levy, B.: A Textbook of Oral Pathology, Philadelphia, 1963, W. B. Saunders Company, pp. 217-218. 2. Chaudhry, A., and Gorlin, R. : Papillary Cystadenoma Lymphomatosum (Adenolymphoma) ; a review of the Literature, Am. J. Surg. 95: 923-929, 1959. Cystadenoma Lymphomatosum; a Rare Teratoid of the Parotid 3. Warthin, A.: Papillary Region, J. Cancer Res. 13: 116-125, 1929. 4. Foote, F., and Frazell, E.: Tumors of the Major Salivary Glands (Atlas of Tumor Pathology, Section IV, Fasciele II), Washington, D. C., 1954, Armed Forces Institute of Pathology. 5. Baum, R., and Perzik, S.: An Evaluation of Warthin’s Tumor: A Clinical Review of 59 Cases. Am. J. Sura. 30: 420-422. 1964. 6. Anderson, W.: PLthology, ed. 6, St. Louis, 1971, The C. V. Mosby Company, pp. 1102. 7. Bernier, S.. and Bhaskar, S.: Lvmphoepithelial Lesions of Salivary Glands, Cancer 11: __ 1156-lf79,
8. Evans, R., and Croickshank, Pathol. 1: 37-57, 1970. 9. Tlie+cke, R.: Oral Pathology,
10. Steiner, F., Eyler, W., Dusault, L., and Block, M.: Identification of Warthin’s Tumors by Scanning of Salivary Glands, Am. J. Surg. 116: 513517, 1968. Tumors of the Major Salivary Glands, Am. J. 11. Turnbull, A., and Frazell, E.: Multiple Surg. 118: 787-789, 1969. 12. Chaudhry, A., Vickers, R., and Gorlin, R.: Intraoral Minor Salivary Gland Tumors ; Analysis of 1,414 Cases, ORAL SURG. 14: 1194-1225, 1961. 13. Assor. D.: Bilateral Carcinoma of the Parotid. One Cancer Arisina in a Warthin’s Tumor, ’ Am. J. Clin. Pathol. 61: 270-274, 1974. 14. Rubin. P.. and Halt. J.: Secretory Sialographv in Diseases of the Major Salivary Glands, Am. Jl Rdentgenol. Radium Ther. “Nuel. Med: ?7: 575598, 1957. ” and Future, CRC Crit. Rev. Radiol. Sci. 4: 15. O’Hara, K. : Sialography : Past, Present, 87-139, 1973. 16. Suzuki, S., and Kawashima, K.: Sialographic Study of Diseases of the Major Salivary Glands, Acta Radiol. [Diagn] (Stockh.) 8: 465478, 1969. Cystadenoma Lymphomatosum, a Developmental 17. Shklar, G., and Chauncey, H.: Papillary Malformation: Histochemical Evidence. J. Oral Sure. 23: 222-230. 1965. and Fu&tional Maldevelopment of 18. Chaudhry, A., Montes, M., and Cutler; L.: Struct&al Salivary Glands, Symposium of Salivary Glands and Their Secretions, Ann Arbor, 1973, Universitv of Michinan Press. DD. 59-93. 19. Schall, G:: The Role of Radionncleotide Scanning in the Evaluation of Neoplasms of the Salivary Glands: A Review, J. Surg. Oncol. 3: 699-714, 1971. 20. Schall? G., and DiChiro, G.: Clinical Usefulness of Salivary Gland Scanning in Nuclear Medicine, Semin. Nucl. Med. 2: 270-277, 1972. 21. Wheelock, M.: Pathologic Features of Neoplasms of the Salivary Glands, Am. J. Surg. 98: 907-911, 1959. 22. Beahrs, O., Woolner, L., Corveth, S., and Devine, K.: Practical Management of Parotid Lesions, Arch. Surg. 8: 890-904, 1960. 23. Butler, C. : Salivary Gland Tumors, J. Laryngol. Otol. 86: 775-784, 1972. 24. Frazell, E.: Observations on the Management of Salivary Gland Tumors, CA 13: 235243, 1968. Reprint requests to : Dr. Stephen R. Matteson Head, Section of Oral Radiology School of Dentistry University of North Carolina Chapel Hill, N. C. 27514