Warthin tumor of the upper lip: an unusual location of a benign salivary gland tumor.
ABSTRACT Warthin tumor (papillary cystadenoma lymphomatosum) is a benign salivary gland tumor involving almost exclusively the parotid gland. The lip is a very unusual location for this type of tumor, which develops only rarely in minor salivary glands. The case of 42-year-old woman with Warthin tumor arising in minor salivary glands of the upper lip is reported.
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ABSTRACT: Warthin tumor (WT) accounts for 4% to 13% of all salivary gland tumors. This benign tumor, which commonly arises in the parotid gland, is the second most common tumor of the salivary gland. WT is multicentric in 12% to 20% of patients and is bilateral in 5% to 14%. The mean age at diagnosis is 62 years (range, 12-92 years), and it rarely presents (<6%) before age 40 years. Extraparotid WT, arising from the submandibular gland or cervical lymph node for example, is very infrequent, with corresponding incidences of 0.4% to 6.9% and 8%, respectively. Moreover, WT arising from the minor salivary gland is extremely rare, with a reported incidence of merely 0.1% to 1.2%. We report here WT arising from the minor salivary gland in the buccal mucosa in a 66-year-old woman and review cases of WT of the minor salivary gland reported in the English literature.The Journal of craniofacial surgery 09/2012; 23(5):e374-6. · 0.81 Impact Factor
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ABSTRACT: Bilateral Warthin's tumours of parotid constitute a small proportion of all salivary gland tumours. We present a case report of surgical management of a prototype case.Journal of Maxillofacial and Oral Surgery 12/2012; 11(4):483-486.
Warthin Tumor of the Upper Lip:
An Unusual Location
Aroldo dos Santos Almeida, DDS, João Adolfo Costa
Hanemann, DDS, PhD, and Denise Tostes Oliveira, DDS, PhD
which has been present for 2 years. The typically
asymptomatic white nodule had a smooth surface,
well-defined borders, and was slightly raised from
surrounding oral mucosa. The patient described epi-
sodes of recurrent swelling, with periodic ruptures
and release of its fluid contents. A clinical diagnosis
of mucocele versus benign tumor arising in the
minor salivary gland was made, and the lesion was
surgically excised under local anesthesia.
Gross examination of the formalin-fixed speci-
men revealed 2 pieces of smooth brown tissue meas-
uring on the aggregate 0.9 × 0.6 × 0.4 cm. Histological
examination with hematoxylin and eosin stain
showed a normal minor salivary glands and a papil-
lary cystic area composed of a double layer of colum-
nar and cuboidal eosinophilic epithelial cells
associated with a prominent lymphoid component
(Figures 1 and 2). Focal mucous cells in the epithe-
lium of the cyst stained positively with periodic acid
Schiff (Figure 3). The lymphoid infiltrate was
arranged in a follicular pattern with germinal cent-
ers formation (Figure 1). Microscopic examination
of multiple sections revealed a transition from
smaller to larger tortuous duct-like spaces. The con-
nective tissue adjacent to the lymphoid area was
noninflammatory and consisted of collagen fibers,
fibroblasts, vessels, and normal minor salivary glands.
The content of the cystic cavity was a homogeneous
eosinophilic material. There was neither nuclear
atypia nor mitotic activity of the luminal cells. No
in the parotid gland. Few cases have been reported
arising from minor salivary gland.1,2 This tumor
shows a predilection for males, with a peak of inci-
dence during the sixth decade of life.2,3
According to literature review, 8 cases of Warthin
tumor located exclusively in the lips region have
been described, 6 of them in the lower lip and 2 in
the upper lip.1 An uncommon case of Warthin tumor
arising in minor salivary glands of the upper lip in a
42-year-old woman is reported.
arthin tumor (papillary cystadenoma lym-
phomatosum) is a benign salivary gland
neoplasm that occurs almost exclusively
A 42-year-old woman was evaluated for a small
enlargement of the upper lip mucosa. The lesion
appeared as a nonulcerated small circumscribed
mass measuring approximately 1 cm in diameter,
Warthin tumor (papillary cystadenoma lymphomato-
sum) is a benign salivary gland tumor involving almost
exclusively the parotid gland. The lip is a very unusual
location for this type of tumor, which develops only
rarely in minor salivary glands. The case of 42-year-old
woman with Warthin tumor arising in minor salivary
glands of the upper lip is reported.
Keywords: Warthin tumor; papillary cystadenoma
lymphomatosum; minor salivary gland
From the Department of Stomatology, Division of Pathology,
Bauru School of Dentistry, University of São Paulo, Bauru, São
Paulo (AdSA, DTO) and Department of Clinics and Surgery,
Division of Stomatology, Federal University of Alfenas, Alfenas,
Minas Gerais (JACH), Brazil.
Address correspondence to: Denise Tostes Oliveira, PhD,
Faculdade de Odontologia de Bauru, Área de Patologia, Alameda
Dr. Octávio Pinheiro Brisolla, 9-75, Zip Code 17.012-901,
Bauru, São Paulo, Brazil; e-mail: firstname.lastname@example.org.
International Journal of
Volume XX Number X
Month XXXX xx-xx
© 2009 Sage Publications
2 International Journal of Surgical Pathology / Vol. XX, No. X, Month XXXX
obstruction of the minor salivary duct was detected.
The histopathological diagnosis established was
Warthin tumor arising from minor salivary gland. A
1-year follow up evaluation revealed no recurrence
of the lesions.
Papillary cystadenoma of the upper lip is frequently
included in most large series of benign tumors arising
of minor salivary gland reported in the literature.2,4,5
However, papillary cystdenoma lymphomatosum or
Warthin tumor in the upper lip, as exemplified by our
case, is a very uncommon tumor.2,5,6
Based on the clinical history with periodic epi-
sodes of fluid content release, the leading clinical
diagnosis was mucocele, although this type of
lesion is almost always restricted to the lower lip.5,7
However, histopathological analysis showed find-
ings more in keeping with a benign salivary gland
tumor forming cystic spaces than a mucus-retention
The presence of cystic area into which project
papillae lined by cuboidal to columnar eosinophilic
epithelial cells, surrounded by lymphoid tissue with
germinal follicles (Figures 1 and 2)1,2,5,6 is a distinct
histopathologic pattern of Warthin tumor. The focal
mucous cells interdigitated between epithelial cells
are an additional feature of this entity.2,5,6
The differential microscopic diagnosis also
includes chronic sclerosing sialadenitis of minor
salivary glands. However, no obstruction of the
minor salivary duct or marked sclerosis of the sali-
vary gland (including ductal ectasia, periductal fibro-
sis, and acinar atrophy)6,8 were detected. The minor
salivary glands adjacent to the cystic area had a nor-
mal appearance (Figure 1). It is important to empha-
size that Warthin tumor usually does not show the
histopathological features described above, which
are promoted by ductal ectasia.4 It is likely that the
incidence of cystadenoma arising in the minor sali-
vary gland has been underestimated, with many
cases having been misdiagnosed as reactive or cystic
lesions in the oral cavity.4
No relapse was observed on 1-year follow-up.
Several studies indicate a minimal recurrence rate,2,5,6
Figure 1. Multicystic cavity associated with lymphoid compo-
nents arranged in a follicular pattern with germinal centers. The
minor salivary gland surrounding the lesion shows a normal
appearance (hematoxylin–eosin stain, 2.5×).
Figure 3. Numerous mucous cells stained with periodic acid
Schiff interdigitate between epithelial cells of the cystic cavity
(periodic acid Schiff stain, 40×).
Figure 2. Papillary cystic area composed of a double layer of
columnar and cuboidal eosinophilic epithelial cells (hematoxylin–
eosin stain, 10×).
Warthin Tumor of the Upper Lip / Almeida et al 3
and a simple surgical excision is reported as the
treatment of choice of this lesion.6
Warthin tumor is rare in the lips, with only 8 cases
having been reported in the literature.1 In our opinion, a
bona fide case of Warthin tumor of the upper lip, ade-
quately documented with sufficient clinical information,
strict histopathologic criteria, good photographic docu-
mentation, and adequate follow-up, was described by
Adibfar and Mintz1 in 1994. The current case represents,
to the best of our knowledge, the second documented
instance of Warthin tumor of the upper lip.
1. Adibfar A, Mintz SM. Papillary cystadenoma lymphoma-
tosum of the upper lip. J Oral Maxillofac Surg.
2. Barnes L, Eveson JW, Reichart P, Sidransky D. Pathology
and Genetics: Head and Neck Tumours. Lyon, France:
IARC Press; 2005.
3. Fantasia JE, Miller AS. Papillary cystadenoma lympho-
matosum arising in minor salivary glands. Oral Surg.
4. Pires FR, Pringle G, Almeida OP, Chen SY. Intra-oral
minor salivary gland tumors: a clinicopathological study
of 546 cases. Oral Oncol. 2007;43:463-470.
5. Ellis GL, Auclair PL, Gnepp DR, eds. Surgical Pathology
of the Salivary Glands. Philadelphia, PA: WB Saunders;
6. Ellis GL. Lymphoid lesion of salivary glands: malignant
and benign. Med Oral Patol Oral Cir Bucal. 2007;12:
7. Mustapha IZ, Boucree SA Jr. Mucocele of the upper
lip: case report of an uncommon presentation and its
differential diagnosis. J Can Dent Assoc. 2004;70:
8. Kojima M, Miyawaki S, Takada S, Kashiwabara K,
Igarashi T, Nakamura S. Lymphoplasmacytic infiltrate
of regional lymph nodes in Küttner’s tumor (chronic
sclerosing sialadenitis: a report of 3 cases. Int J Surg