Suppurative sialadenitis of the upper lip: a report of 3 cases of an infrequent lesion

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HAROLD D. BAURMASH 2. Portmann B, Popper H, Neuberger J, et al: Sequential and diagnostic features in primary biliary cirrhosis based on serial histologic study in 209 patients. Gastroenterology 88:1777, 1985 3. Heseltine L, Turner IB, Fussey SM, et al: Primary biliary cirrhosis. Gastroenterology 99:1786, 1990 4. Butler P, Valle F, Burroughs AK: Mitochondrial antigens and antibodies in primary biliary cirrhosis. Postgrad Med J 67:790, 1991 5. Gershwin ME, Ansara AA, Mackay IR, et al: Primary biliary cirrhosis: An orchestrated immune response against epithelial cells. Immunol Rev 174:210, 2000 6. Tanaka A, Borchers AT, Ishibashi H, et al: Genetic and familial considerations in primary biliary cirrhosis. Am J Gastroenterol 96:8, 2001 7. Richards A, Rooney J, Prime S, et al: Primary biliary cirrhosis: Sole presentation with rampant dental caries. Oral Surg Oral Med Oral Pathol 77:16, 1994 8. Kaplan MM: Primary biliary cirrhosis. N Engl J Med 335:1570, 1996 9. Mahl TC, Shockcor W, Boyer JL: Primary biliary cirrhosis: Survival of a large cohort of symptomatic and asymptomatic patients followed for 24 years. J Hepatol 20:707, 1994 10. Crippin JS, Jorgensen RA, Dickson ER, et al: Hepatic osteodystrophy in primary biliary cirrhosis: Effects of medical treatment. Am J Gastroenterol 89:47, 1994 11. Vitali C, Bombardieri S, Moustsopoulos HM, et al: Assessment of the European classification criteria for Sjogren’s. Ann Rheum Dis 55:116, 1996

12. Golding PL, Smith M, Williams R: Multisystem involvement in chronic liver disease: Studies on the incidence and pathogenesis. Am J Med 55:772, 1973 13. Epstein O, Chapman RWG, Lake-Bakaar G: The pancreas in primary biliary cirrhosis and primary sclerosing cholangitis. Gastroenterology 83:1177, 1982 14. Uddenfeldt P, Danielsson A, Forssell A, et al: Features of Sjogren’s syndrome in patients with primary biliary cirrhosis. J Int Med 230:443, 1991 15. Bach N, Schaffner F: Familial primary biliary cirrhosis. J Hepatol 20:698, 1994 16. Pirakh-Patel A, Gold EB, Worman H, et al: Risk factors for primary biliary cirrhosis in a cohort of patients from the United States. Hepatology 33:16, 2001 17. Findor JA, Sorda JA, Darvich JR, et al: Familial association in autoimmune liver disease. Medicina (B Aires) 62:241, 2002 18. Stone J, Wade JA, Cauch-Dudek, et al: Human leucocyte antigen class II associations in serum antimitochondrial antibodies (AMA)-positive and AMA-negative primary biliary cirrhosis. J Hepatol 36:8, 2002 19. Wassmuth R, Depner F, Danielsson A, et al: HLA class II markers and clinical heterogeneity in Swedish patients with primary biliary cirrhosis. Tiss Antigens 59:381, 2002 20. Lindgren S, Manthorpe R, Eriksson S: Autoimmune liver disease in patients with primary Sjogren’s syndrome. J Hepatol 20:354, 1994 21. Hansen BU, Lindgren S, Eriksson S, et al: Clinical and immunologic features of Sjogren’s syndrome in patients with primary biliary cirrhosis with emphasis on focal sialadenitis. Acta Med Scand 224:611, 1988 J Oral Maxillofac Surg 61:1361-1365, 2003

Suppurative Sialadenitis of the Upper Lip: A Report of 3 Cases of an Infrequent Lesion Harold D. Baurmash, DDS* Suppurative sialadenitis involving the upper lip appears to be a very rare occurrence in the absence of trauma. A 32-year review of the literature, conducted under the heading of “sialadenitis of the lips,” revealed only 1 similiar case, reported by Lederman.1 It is the purpose of this article to call attention to this lesion and to describe its clinical manifestations, histopathology, and successful management, with the presentation of 3 cases from a 19-year period. In all cases, the swellings were primary episodes, and the medical and trauma histories were negative. *Retired Clinical Professor, Columbia University, School of Dental and Oral Surgery, New York, NY. Address correspondence and reprint requests to Dr Baurmash: 4666 Hazleton Lane, Lake Worth, FL 33467. © 2003 American Association of Oral and Maxillofacial Surgeons

0278-2391/03/6111-0022$30.00/0 doi:10.1016/S0278-2391(03)00742-0

Report of Cases CASE 1 A 48-year-old black man presented with an acute painful swelling of the upper right lateral lip region that had been present for 48 hours (Fig 1). Intraorally, there was a moderately large diffuse swelling that appeared to extend to the adjacent cheek region. A soft tissue radiograph, with a periapical film placed under the swollen lip, was negative for a sialolith. Incision and drainage was performed under local anesthesia, resulting in a free flow of pus (Fig 2). A drain was placed for 24 hours, and amoxicillin was prescribed for 2 weeks. Although the swelling decreased, there remained a significantly large, well-circumscribed indurated lip mass (Fig 3). With the use of local anesthesia, a horizontal mucosal incision was made through the midportion of the mass, exposing a wide region of inflamed salivary gland tissue. This lesional tissue was removed down to orbicularis muscle. Only after thorough palpation of the region with no evidence of induration was the mucosa closed with interrupted sutures.

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FIGURE 1. Patient 1, showing a moderate-sized extraoral swelling of the upper right lip, including depressed position of the lip and commissure on that side.

The histopathology revealed an admixture of inflamed fibrous connective tissue and skeletal muscle bundles (Fig 4). Also evident were salivary gland structures with dilated ducts containing a combination of mucus and neutrophils. There was ductal squamous metaplasia, salivary gland acinar depletion, and a diffuse infiltrate of acute and chronic inflammatory cells. The diagnosis was acute and chronic sialadenitis. Healing was uneventful, and 2 months postoperatively there was no clinical evidence of swelling or induration either extraorally or intraorally (Fig 5). CASE 2 A 53-year-old edentulous black man developed an acute painful swelling of the upper right lateral lip extending to the commissure that had been present for 24 hours (Fig 6). After a soft tissue radiograph failed to show a sialolith (Fig 7). an incision was performed, resulting in significant suppurative drainage (Fig 8). After a 2-week period on penicillin, the patient returned for excision of the diseased salivary gland. A large horizontal

FIGURE 2. Patient 1, showing the large intraoral swelling and evacuated pus after incision and drainage.

SUPPURATIVE SIALADENITIS OF THE UPPER LIP

FIGURE 3. Patient 1, showing the intraoral swelling reduced after 2 weeks of antibiotic therapy. However, there remains a rather large, well-circumscribed residual region.

incision was made through the mucosal mass, exposing a wide zone of exophytic inflamed salivary gland tissue that was completely excised (Fig 9). The histopathology was essentially the same as that for patient 1, with the same diagnosis. Primary closure was performed, and a drain was placed for 24 hours. Healing progressed satisfactorily with no evidence of swelling or induration after 2 weeks. CASE 3 A 52-year-old white man complained of a moderate swelling of the right upper lip and adjacent cheek that had been present for approximately 4 days. Compression of the swelling resulted in suppurative drainage at the center of the intraoral mass (Fig 10). After a negative soft tissue radiograph, incision and drainage was performed. After 1 week of antibiotic therapy with penicillin, the standard excisional technique was performed, leaving a clean muscle base and margins (Fig 11). The histopathology and diagnosis were the same as for patients 1 and 2.

FIGURE 4. Patient 1. Histopathology reveals acute and chronic inflammatory infiltrates with dilated salivary gland ducts (hematoxylin and eosin stain, original magnification ⫻100).

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FIGURE 5. Patient 1 at 2 months postsurgery. A, Extraoral appearance is normal. B, Intraoral tissue is normal.

There was normal healing, and the patient was discharged after 1 month with no evidence of swelling or induration.

Discussion The submucosal area of the upper lip is the most common site for the relatively rare minor salivary gland adenoma (monomorphic)2 and the minor salivary gland sialolith,3 which have similiar age incidences (fourth to seventh decades) and male preference, as shown by the patients presented here. Clinical differentiation between suppurative sialadenitis and adenoma should not be a problem because benign tumors of the minor salivary glands present as localized, submucosal, painless, fairly moveable nodular masses with a duration of 1 to 3 years. In the majority of cases, these are the same symptoms as for the minor salivary gland sialolith, although their duration is slightly less. However, in Jensen et al’s4 study of minor salivary gland stones, they noted that 7

FIGURE 6. Patient 2, showing involvement of the extraoral swelling with the upper left lateral lip, commissure, and adjacent cheek region.

patients presented with an inflammatory swelling combined with drainage, similiar to the presenting symptoms reported in this article. For this reason, the standard use of a soft tissue radiograph of most upper lip swellings is recommended. One should also keep in mind that a negative radiograph may not rule out a stone; in the study of Jensen et al, 11 cases were highly mineralized, 23 cases were partially mineralized, and 13 cases were nonmineralized. Lederman1 described the case of a 43-year-old woman who initially presented with a moderately painful diffuse swelling of the upper lip that had been present for 24 hours and that clinically and his-

FIGURE 7. Patient 2. A periapical radiograph was placed under the lip swelling to rule out a sialolith. Penciled arrow corresponds with the center of the mass.

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FIGURE 8. Patient 2: Incision and drainage resulted in a free flow of suppuration.

topathologically resembled these reported cases. However, the similarity ends there, because the patient reported prior recurrent episodes of swelling in this region 3 or 4 times a year for 4 to 5 years. She had been unsuccessfully treated during this period by numerous practitioners who prescribed cephalexin, L-lysine, and tetracycline rinses. Lederman continued the unsuccessful management with intralesional and oral steroids in combination with cephalexin. His eventual recommendation of surgical excision was rejected by the patient, and the recurrences continued during his 1-year follow-up. The key to successful management of these cases after the acute symptoms have subsided is the complete surgical excision of all involved salivary gland tissue. Normal accessory salivary glands appear as discrete soft grape-like structures of light yellow or amber, whereas those with inflammation are diffuse, matted, reddish to purplish indurated masses. All such tissue must be dissected and removed, leaving a clean muscle base with normal soft tissue margins before primary closure.

FIGURE 9. Patient 2. After the acute symptoms subsided, a large horizontal mucosal incision through the center of the mass exposed the inflamed salivary gland tissue.

SUPPURATIVE SIALADENITIS OF THE UPPER LIP

FIGURE 10. Patient 3, showing a moderate-sized intraoral swelling involving the commissure and adjacent cheek region. There is pus drainage at the center of the mass after external compression.

In response to a literature search of “sialadenitis of the lips,” the majority of articles involved cheilitis glandularis (CG)5,7 or the more recent term, stomatitis glandularis,6,8 which appeared to include all nonspecific, nontraumatic, diffuse enlargements of the minor salivary glands of the oral cavity. The lower lip was involved almost exclusively, although there were reports of upper lip,7 palate6 and cheek.8 swellings. Unlike the descriptions reported here, Winchester et al’s7 description of the patient with upper lip involvement was that of multiple firm nodules of the entire lip along with overlying black punctate lesions from which viscous mucus could be expressed. The etiology of CG is unclear and confusing because a myraid of irritants such as actinic (chronic sun and wind exposure), chronic lip biting, mouth breath-

FIGURE 11. Patient 3, showing the intraoral wound after removal of the diseased salivary gland tissue. The muscle surface and wound margins are clean and appear normal.

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ing, smoking, and poor oral hygiene have been listed as causative agents. Even congenital predisposition or an autosomal dominant pattern of inheritance has been offered as an explanation.9 CG is considered a progressive disease if untreated, beginning with the simple type, consisting of lip enlargement with dilated duct openings. This is followed by the superficial suppurative type with clinical signs of swellings, induration, and crusting. It has been suggested that the latter 2 types are merely progressive stages of the disorder leading to the final stage,7 which is the deep suppurative type with symptoms of enlarged everted lips, a deep seated infection with abscess formation, and fistulous tracts. Histopathologically, the progressive nature and degree of inflammation appear to bear this out. In the absence of any clinical resemblance to the patients presented, the minimal symptoms of pain and suppuration, the chronicity of CG, and its propensity for lower lip involvement, I believe that suppurative sialadenitis should not be included under this broad heading. The one thing shared is the recommendation of surgical excision as the primary treatment option. Anneroth and Hansen,3 in their review of the development of minor salivary gland stones stated that the pathologic changes observed in the glandular lobules surrounding the salivary calculi in the form of diffuse chronic inflammatory cell infiltration, ductal dilatation, acinar atrophy with squamous metaplasia of acini and ducts, and fibrosis are characteristic morphologic changes for longstanding retention phenomenon in both major and minor salivary glands. Ho et al10 also described similiar characteristics in their histopathologic examination of minor salivary gland sialoliths.

It was not the purpose of this article to propose a rational causation for suppurative sialadenitis but rather to describe the condition and suggest a method of management. However, if one were to compare the histopathology of these cases with those described by Anneroth and Hansen3 and Ho et al,10 could one suspect that conditions, before the onset of suppurative sialadenitis, might have been the precursor of stone development but one acute episode interrupted the sequence? Acknowledgment The author wishes to express his appreciation to David Zegarelli, DDS, Professor of Oral Pathology, Columbia Presbyterian Medical Center, for his review and histopathologic report.

References 1. Lederman DA: Suppurative stomatitis glandularis. Oral Surg Oral Med Oral Pathol 78:319, 1994 2. Levine L, Krutchkoff DJ, Eisenberg E: Monomorphic adenoma of minor salivary glands: A reappraisal and report of 9 new cases. J Oral Surg 39:101, 1981 3. Anneroth G, Hansen LS: Minor salivary gland calculi: A clinical and histopatholgical study of 49 cases. Int J Oral Surg 12:80, 1983 4. Jensen JL, Howell FV, Rick GM, et al: Minor salivary gland calculi: A clinicopathologic study of 47 new cases. Oral Surg Oral Med Oral Pathol 47:44, 1979 5. Swerlick RA, Cooper PH: Cheilitis glandularis: A re-evaluation. J Am Acad Dermatol 10:466, 1984 6. Williams HK, Williams DM: Persistent sialadenitis of the mucous glands stomatitis glandularis. Br J Oral Maxillofac Surg 27:212, 1989 7. Winchester L, Scully C, Prune SS, et al: Cheilitis glandularis: A case affecting the upper lip. Oral Surg Oral Med Oral Pathol 62:654, 1986 8. Connell H, Kuawala C, Farthing P: Stomatitis glandularis: Two confirmed cases of a rare condition. Br Dent J 182:22, 1997 9. Weir TW, Johnson WC: Cheilitis glandularis. Arch Dermatol 103:433, 1971 10. Ho V, Currie WJR, Walker A: Sialolithiasis of minor salivary glands. Br J Maxillofac Surg 30:273, 1992