Maxilo-facial surgery

PLEOMORPHIC ADENOMA OF THE UPPER LIP, ORIGINATING IN

MINOR SALIVARY GLANDS. CASE REPORT

Daniela Trandafir1, D. Gog\lniceanu2, Violeta Trandafir3 ,

P. Gog\lniceanu4, V. Burlui5

1. Assist. Prof. PhD, specialist registrar maxillofacial surgery, Faculty of Med Dent - „Gr. T. Popa” U.M.Ph Iasi
2. Prof. PhD, Faculty of Med Dent - „Apollonia” University, Iasi
3. Assist Prof. PhD, senior registrar maxillofacial surgery, Faculty of Med Dent - „Gr. T. Popa” U.MPh Iasi
4. Medicine Doctor, University College of London UK, member of Royal College of Surgeons of England
5. Prof, PhD, Faculty of Medical Dentistry – „Apollonia” University, Ia[i
Corresponding author: Daniela Trandafir: e-mail: trandafir.daniela@gmail.com

Abstract
Pleomorphic adenoma, also known as benign mixed
tumor, is the most common benign tumor of salivary
glands that mostly occurs in the parotid or submandibu-
lar glands, but may also be found in the minor salivary
glands that are distributed throughout the oral cavity.
Surgical removal with adequate margins is the principal
treatment. Due to its microscopic projections, this tumor
requires a wide resection to avoid recurrence. We report
a case of a pleomorphic adenoma in the upper lip, the
second site for frequency for benign tumors of minor sali-
vary glands, after the hard and soft palate.
Key words: minor salivary glands, pleomorphic ad-
enoma, oral cavity
INTRODUCTION
Tumors of the minor salivary glands repre-
sent 10-15% of all salivary gland neoplasms [1).
An estimation of the incidence of salivary gland
neoplasms in the population, is that, for every
100 tumors of the parotid gland, there are likely
to be 10 of the submandibular gland, one of the
sublingual gland, and 10 of the minor salivary
glands. The probability of a malignant diagnosis
is less than 25% in patients with a tumor of the
parotid gland, about 50% in those with a tumor
of the submandibular gland, more than 80% in
patients with a tumor of minor salivary gland
origin, and virtually 100% in those few with a
tumor of the sublingual gland (2).
Benign tumors of minor salivary gland origin
are most frequently pleomorphic adenoma and
have been located in areas as diverse as tongue
(3), posterior part of the tongue (4), nasal cavity
and septum (5), larynx (6), and trachea (7). The
most common site for pleomorphic adenoma is
the hard palate, followed by the upper lip. Some
of these pleomorphic adenomas can become
massive with malignant degeneration before
presentation (8).
CASE REPORT
A 57 year-old female presented with a pain-
less submucosal swelling in the right half of the
upper lip, with a history of 1 year, in the Depart-
ment of Oral and Maxillofacial Surgery Iaºi, in
May 2010. Her past medical history was un-
eventful and there was no previous history of
regional trauma. On clinical examination there
was a 2 cm/1.5 cm firm, painless, circumscribed
lession in the right half of the upper lip, adher-
ent to the underlying structures and the mucosal
surface was non-ulcerated (figure 1). There was
no regional lymphadenopathy and her general
condition was normal. A clinical differential di-
agnosis of squamous cell carcinoma with nodu-
lous outset and a tumor of minor salivary gland
orgin (benign or malignant) were considered.
The entire tumor was excised with a wide mar-
gin, on local anaesthesia (figures 2, 3). The result
of histopathological exam was: pleomorphic ad-
enoma in the upper lip, with a particularity: it
microscopically simulated a myoepithelial car-
cinoma (figure 4). There was no recurrence at
three month follow-up.

252 volume 14 • issue 3 July / September 2010 • pp 252-255

 Daniela Trandafir, D. Gog\lniceanu, Violeta Trandafir, P. Gog\lniceanu, V. Burlui
Fig. 1. Painless non-ulcerative, submucosal swelling
in the upper lip, right half
Fig. 2. Intraoperative view of dissection in benign
tumor of minor salivary gland originating in the
upper lip
Fig. 3. The operative specimen
Journal of Romanian Medical Dentistry
Fig. 4. The histopathological result: pleomorphic
adenoma in the upper lip (particularity: bilayered
duct-like structures with conspicuous outer layer of
clear myoepithelial cells simulating a myoepithelial
carcinoma)
DISCUSSION
Minor salivary glands, coming up to 450-1,000
in number, are widely distributed in the head
and neck area (9). In their majority (70-90%), they
are located in the oral cavity and oropharynx,
including the lateral margins of the tongue, lips
and buccal mucosa, palate, glossopharyngeal
area, and the retromolar trigone. The remaining
ones are located in the nose, paranasal sinuses,
pharynx and larynx. Minor salivary glands con-
tribute to about 8-10% of the volume of whole
saliva.
The signs and symptoms of tumors associated
with minor salivary glands vary according to
their different anatomical sites. The majority of
patients were 60 years of age or older. Many of
the larger series had reported a gender distribu-
tion of 66% women. The most frequent site of
origin is the oral cavity and oropharynx and,
within the oral cavity, most tumors develop in
the region of the hard palate because this is the
area with the highest density of minor salivary
glands. Most of patients present a painless non-
ulcerative, submucosal swelling. The mucosal
layer is adherent to the mass and a small ulcer
may be present during its evolution. Up to 26%
of patients presented local pain (1).
253
PLEOMORPHIC ADENOMA OF THE UPPER LIP, ORIGINATING IN MINOR SALIVARY GLANDS. CASE REPORT
Physical examination and awareness that a
clinically „benign” submucosal swelling at any
place in the head and neck may be a tumor of
minor salivary gland origin and that, statisti-
cally, the pathology of that tumor is more likely
to be malignant rather than benign, is the most
important clinical information that will improve
accurate diagnosis, and allow for a rational plan
of management of these tumors.
Currently, imaging using computed tomog-
raphy and/or magnetic resonance imaging may
help to the delineation of the tumor, an accurate
staging of the disease, and also for a correct plan-
ning of a surgical procedure.
The use of fine-needle aspiration cytology
(FNAC) in tumors of minor salivary gland ori-
gin may be helpful in correctly classifying the
tumor as benign or malignant, however, the use
of incisional biopsy or punch-biopsy may create
a better and more representative specimen, thus
revealing the correct histological type.
The current incidence of a benign tumor of
salivary gland origin is estimated to be 60-80/
million people per year. The incidence of malig-
nant salivary tumors in the United States of
America is 10/1 million people per year and in
the United Kingdom is 0.6/1million per year
(10).
Benign tumors of minor salivary gland origin
are most frequently pleomorphic adenoma and
the most common sites are the hard palate and
the upper lip. Other benign neoplasms of minor
salivary gland origin have been reported in the
oral cavity (11). Benign epithelial tumors also
include: Warthin tumor, monomorphic ad-
enoma, intraductal papilloma, oncocytoma, and
sebaceous neoplasms. Benign nonepithelial
tumors (mesenchimal origin) include:
hemangioma, angioma, lymphangioma (cystic
hygroma), lipoma, and neural sheath tumors.
Microscopically, there is a variety of configu-
rations of pleomorphic adenomas, but the essen-
tial components are: capsule (complete or incom-
plete), epithelial cells in a variety of
configurations, myoepithelial cells, stroma
(which may be fibrocollagenous, mucoid, chon-
droid or myxochondroid, in varing proportions,
but which form the bulk of the tumor in the
254
majority of cases) (11). We presented in this pa-
per a rare case of a pleomorphic adenoma in the
upper lip, which microscopically looks like a
myoepithelial carcinoma.
Surgical excision is the treatment of choice for
benign tumors of minor salivary glands in the
oral cavity. The treatment will depend on the
size and location of the tumor. Recurrence de-
pends upon the accuracy of local excision, and
most patients with recurrence will have it within
18 months. Long-term follow-up is recom-
mended, as the risk of reccurence may remain
life long for such patients (12).
CONCLUSIONS
• Tumors of minor salivary gland origin are
uncommon and are more likely to be
histologically malignant than benign.
• Clinical presentation for a benign tumor of
minor salivary gland origin in the oral cav-
ity is most commonly a non-ulcerative,
submucosal swelling.
• Incisonal or punch-biopsy should be per-
formed prior to planning radiological
imaging and/or final treatment.
• The variety of microscopic configurations
of pleomorphic adenomas of minor sali-
vary glands is remarkable, but stroma rep-
resents the bulk of the tumor in the major-
ity of cases.
• Complete surgical excision is the preferred
treatment.
• Pleomorphic adenoma remains the most
common benign neoplasm for minor sali-
vary glands and the treatment is local exci-
sion with a safe margin.
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