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: [email protected]

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

adenoma in the upper lip (particularity: bilayered
duct-like structures with conspicuous outer layer of
Fig. 1. Painless non-ulcerative, submucosal swelling clear myoepithelial cells simulating a myoepithelial
in the upper lip, right half 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.
Fig. 2. Intraoperative view of dissection in benign
tumor of minor salivary gland originating in the
The signs and symptoms of tumors associated
upper lip 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
Fig. 3. The operative specimen may be present during its evolution. Up to 26%
of patients presented local pain (1).

Journal of Romanian Medical Dentistry 253

PLEOMORPHIC ADENOMA OF THE UPPER LIP, ORIGINATING IN MINOR SALIVARY GLANDS. CASE REPORT

Physical examination and awareness that a majority of cases) (11). We presented in this pa-
clinically „benign” submucosal swelling at any per a rare case of a pleomorphic adenoma in the
place in the head and neck may be a tumor of upper lip, which microscopically looks like a
minor salivary gland origin and that, statisti- myoepithelial carcinoma.
cally, the pathology of that tumor is more likely Surgical excision is the treatment of choice for
to be malignant rather than benign, is the most benign tumors of minor salivary glands in the
important clinical information that will improve oral cavity. The treatment will depend on the
accurate diagnosis, and allow for a rational plan size and location of the tumor. Recurrence de-
of management of these tumors. pends upon the accuracy of local excision, and
Currently, imaging using computed tomog- most patients with recurrence will have it within
raphy and/or magnetic resonance imaging may 18 months. Long-term follow-up is recom-
help to the delineation of the tumor, an accurate mended, as the risk of reccurence may remain
staging of the disease, and also for a correct plan- life long for such patients (12).
ning of a surgical procedure.
The use of fine-needle aspiration cytology CONCLUSIONS
(FNAC) in tumors of minor salivary gland ori-
gin may be helpful in correctly classifying the
• Tumors of minor salivary gland origin are
tumor as benign or malignant, however, the use
uncommon and are more likely to be
of incisional biopsy or punch-biopsy may create
histologically malignant than benign.
a better and more representative specimen, thus
• Clinical presentation for a benign tumor of
revealing the correct histological type.
minor salivary gland origin in the oral cav-
The current incidence of a benign tumor of
ity is most commonly a non-ulcerative,
salivary gland origin is estimated to be 60-80/
submucosal swelling.
million people per year. The incidence of malig-
• Incisonal or punch-biopsy should be per-
nant salivary tumors in the United States of
formed prior to planning radiological
America is 10/1 million people per year and in
imaging and/or final treatment.
the United Kingdom is 0.6/1million per year
• The variety of microscopic configurations
(10).
of pleomorphic adenomas of minor sali-
Benign tumors of minor salivary gland origin
vary glands is remarkable, but stroma rep-
are most frequently pleomorphic adenoma and
resents the bulk of the tumor in the major-
the most common sites are the hard palate and
ity of cases.
the upper lip. Other benign neoplasms of minor
• Complete surgical excision is the preferred
salivary gland origin have been reported in the
treatment.
oral cavity (11). Benign epithelial tumors also
• Pleomorphic adenoma remains the most
include: Warthin tumor, monomorphic ad-
common benign neoplasm for minor sali-
enoma, intraductal papilloma, oncocytoma, and
vary glands and the treatment is local exci-
sebaceous neoplasms. Benign nonepithelial
sion with a safe margin.
tumors (mesenchimal origin) include:
hemangioma, angioma, lymphangioma (cystic
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