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British Journal of Oral and Maxillofacial Surgery xxx (2015) xxx.e1–xxx.e3

Short communication
Pleomorphic adenoma of an accessory submandibular
salivary gland: a rare entity
Rajiv S. Desai a,∗ , Deepashree Meshram b , Sagar S. Jangam b , Jatinder S. Singh a
a Department of Oral Pathology, Nair Hospital Dental College, Dr. A. L. Nair Road, Mumbai Central, Mumbai, 400008, India
b Department of Oral & Maxillofacial Surgery, Nair Hospital Dental College, Dr. A. L. Nair Road, Mumbai Central, Mumbai, 400008, India

Accepted 9 May 2015

Abstract

An accessory submandibular salivary gland is a rare anatomical variant, and a tumour within one is even rarer. We describe a 54-year-old man
who presented with a slowly-enlarging mass in the right submandibular region which on magnetic resonance imaging (MRI) seemed to be
close to, but not arising from, the right submandibular salivary gland. This was found to be a benign pleomorphic adenoma arising from an
accessory submandibular salivary gland. To our knowledge it is only the second report of a pleomorphic adenoma that developed within an
accessory submandibular salivary gland.
© 2015 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: Acessory; Submandibular gland; Pleomorphic adenoma

Introduction Case report

Any salivary tissue (apart from the major or minor salivary
glands) that is found in the oral cavity, pharynx, or upper air-
way is called heterotopic, and is most common in the neck or
within the mandible.1,2 An accessory submandibular salivary
gland is extremely rare, and one with a mass within it is even
rarer.3,4 Here we describe the case of a benign pleomorphic
adenoma within an accessory submandibular salivary gland.
∗ Corresponding author at: Department of Oral Pathology, Nair Hospital
Dental College, Dr. A. L. Nair Road, Mumbai Central, Mumbai-400008,
India. Tel.: +919821545914.
E-mail addresses: nansrd@hotmail.com
(R.S. Desai), deeps meshram@rediffmail.com (D. Meshram),
sagarjangam19@gmail.com (S.S. Jangam), docjatinder@hotmail.com
(J.S. Singh).
A 54-year-old man presented with a slow-growing, painless
swelling in the right upper neck of 6 months’ duration. There
was no intraoral or extraoral discharge, and the swelling did
not enlarge with eating. His medical, surgical, and personal
history were not relevant.
On extraoral examination there was a 1 cm, mobile, firm
mass in the right submandibular triangle that was not palpable
bimanually. The rest of the neck was within normal limits.
He had no signs of cranial nerve palsy.
Magnetic resonance imaging (MRI) showed a well-
defined, round, exophytic, lobulated soft tissue area of altered
signal intensity measuring 2 x 2.3 x 1.7 cm in the right sub-
mandibular region, which arose from the superior aspect
of the right submandibular salivary gland but not from the
gland itself, and displaced the superficial lobe of the right
parotid gland posteriorly and medially. It looked hypointense
on T1-weighted images and hyperintense on T2-weighted
inages and a STIR sequence, and there was homogeneous

http://dx.doi.org/10.1016/j.bjoms.2015.05.011
0266-4356/© 2015 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Desai RS, et al. Pleomorphic adenoma of an accessory submandibular salivary gland: a rare entity. Br
J Oral Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2015.05.011
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Fig. 1. Magnetic resonance image after contrast showing hyperintense T2-
weighted homogenous enhancement of the right accessory submandibular
salivary gland (arrow).
enhancement on the study taken after constrast. There was
no evidence of calcification or haemorrhage (Figs. 1-2).
Ultrasound-guided, fine-needle aspiration cytology was
consistent with a pleomorphic adenoma, and the tumour was
removed under general anaesthesia. The tumour was found
close to, but completely separate from, the right submandi-
bular salivary gland (Fig. 3).
Fig. 2. Axial view of the lump on magnetic resonance imaging (arrow).
Please cite this article in press as: Desai RS, et al. Pleomorphic adenoma of an accessory submandibular salivary gland: a rare entity. Br
J Oral Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2015.05.011
Fig. 3. A completely separated tumour from the right submandibular salivary
gland during operation (arrow).
As the lesion was benign we did only a conservative exci-
sion of the tumour with a margin of normal tissue, instead
of our usual practice of removal of the entire submandi-
bular gland for a tumour of a submandibular gland. The
excised tumour was encapsulated, measured 2 x 2 x 1 cm,
and on histopathological examination was a benign pleomor-
phic adenoma. A small amount of tissue surrounding the
mass resembled normal submandibular salivary gland. The
patient’s postoperative course was uneventful.
Discussion
Distinction between accessory and heterotopic salivary glan-
dular tissue is clinically important. Heterotopic salivary tissue
is found where a salivary gland is normally neither located
nor expected. It is the result of embryonic malposition and,
rarely, the site of other disease. This contrasts with an acces-
sory salivary gland, which is a different clinical entity, being
a detached part of a major salivary gland that develops along
the external ductal systems.
The anatomical variant of an accessory parotid gland is
well-documented with an estimated incidence of 21% in the
population.3 However, the incidence of an accessory subman-
dibular salivary gland is not known because of its rarity, and
there are few case reports.4–7 It is probable that the accessory
salivary gland results from premature development of an aci-
nar system, with a connecting duct that drains into the main
parotid or submandibular duct.6
Being cross-sectional, MRI provides a 3-dimensional pic-
ture of the submandibular salivary gland ductal system, which
shows the presence and site of any accessory gland and any
underlying disease involving the glandular tissue.5 Histolog-
ical features of the accessory and main salivary glands do not
differ from those of the normal gland, and all disorders of
the major salivary glands may affect the accessory glands.2,8
Apart from the tumour, our case showed no histological
difference in accessory salivary gland tissue when com-
pared with normal submandibular salivary gland. Although
they are rare, diseases that involve an accessory submandi-
bular salivary gland should be considered in the differential
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diagnosis of any condition of the submandibular region.
Appropriate investigations in the form of MRI and fine-
needle aspiration cytology are of great help.
Conflict of Interest
We have no conflict of interest.
Ethics statement/confirmation of patient’s permission
We have the patient’s permission for publication of this case
report.
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Please cite this article in press as: Desai RS, et al. Pleomorphic adenoma of an accessory submandibular salivary gland: a rare entity. Br
J Oral Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2015.05.011
xxx.e3
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