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Case Report

Necrotizing sialometaplasia of the palate:

A case report
Ashwarya Trivedi, Mysore K Sunil, Sonam Gupta, Shaveta Garg
Department of Oral Medicine and Radiology, Guru Nanak Dev Dental College and Research Institute, Sunam,
Punjab, India

ABSTRACT

Necrotizing sialometaplasia is a benign, self-limiting, reactive inflammatory disorder of the salivary tissue, which mimics
malignancy both clinically and histopathologically. The etiology is unknown, although it most likely represents a local ischemic
event, infectious process or perhaps an immune response to an unknown allergen. A case of necrotizing sialometaplasia of the
palate in a 40-year-old male patient is presented. Histopathological examination is necessary for the diagnosis of necrotizing
sialometaplasia because the clinical features of this condition can mimic other diseases, particularly malignant neoplasms.
Key words: Minor, necrotizing, palate, saliva, salivary glands, sialometaplasia

Introduction Case Report

N ecrotizing sialometaplasia (NS) was first described

by Abrams and Melrose in 1973.[1] Necrotizing
sialometaplasia is a necrotizing
inflammatory process that largely involves the minor
salivary glands and the major salivary glands in up to
10% of the cases. [2] It may occur in all the regions
where salivary gland tissue is found, such as, the nasal
cavity sinuses, lower lip, tongue, cheek, retromolar
pad, soft palate, and larynx; however, it occurs most
commonly in the palate.[1] Here we report a case of a
40-year-old male patient corresponding to the clinical
and histopathological features of NS in the mid-
palatal region.
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DOI:
10.4103/0972-1363.145032
A 40-year-old male patient, who was a driver by
occupation, reported to the department with a chief
complaint of pain in the palatal region since 15 days. The
history of present illness revealed that the pain was
severe, intermittent, sudden in onset, and radiated to the
left temporal region. There was a history of swelling in
the same region 10-15 days back, which was initially
small in size and gradually increased to the present size.
There was also a history of pus discharge from the
swelling since eight to ten days. There was no history of
trauma, paresthesia or fever. The patient gave a history
of extraction of the upper front teeth six to seven months
back, as the teeth were mobile. The past medical history
revealed no systemic illness. The personal history
revealed that the patient was vegetarian in diet and there
was no history of any deleterious habit like smoking,
tobacco, betel nut chewing, alcohol or the like.
Extraoral examination revealed no abnormality. Intraoral
examination revealed a swelling in the hard palate,
which was approximately 3 × 3 cm, extending posteriorly
from the palatal mucosa behind the anterior teeth, up to
the junction of the hard and soft palate. It extended

Address for correspondence: Dr. Shaveta Garg, PG student, Department of Oral Medicine and Radiology, Guru Nanak Dev Dental
College and Research Institute, Bathinda-Patiala Road, Sunam - 148 028, Punjab, India. E-mail: garg_shaveta33@yahoo.com
Received: 26-06-2014 Accepted: 06-11-2014 Published: 19-11-2014

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Trivedi A et al.: Necrotizing sialometaplasia of the palate
mediolaterally from the 13, 14, 15, and 16 teeth region on the
patient’s right side to the 23, 24, 25, and 26 teeth region on
the left side, crossing the mid palatine raphe. The surface of
the swelling was lobulated. The overlying mucosa was
erythematous when compared with the surrounding
mucosa. There was a palatal perforation in the midpalatal
region. On palpation, the swelling was soft-to-firm in
consistency, non-compressible, non-reducible, and tender.
There was exudation of pus from the opening [Figure 1].
Teeth numbers 11, 12, 21, and 22 were missing and
generalized calculus and stains were present.
With the above clinical findings, a provisional
diagnosis of generalized chronic periodontitis with
suppurative osteomyelitis was made and differential
diagnoses of deep mycotic infection, squamous cell
carcinoma, mucoepidermoid carcinoma, and midline
lethal granuloma were considered.
Intraoral periapical radiographs (IOPA) were taken,
which showed vertical loss of alveolar bone in relation
Figure 1: Intraoral picture showing swelling with perforation in the mid-
palatal region
Figure 3: Occlusal radiograph showing a diffuse rarefied area in the
anterior mid-palatal region
356 Journal of Indian Academy of Oral Medicine & Radiology | Jul-Sep 2014 | Vol 26 | Issue 3
to 14, 15, 16, and 17, and horizontal loss of alveolar
bone in relation to 26, 27, and 28 [Figure 2a and b].
A maxillary cross-sectional occlusal radiograph was
taken, which revealed normal anatomic landmarks
and missing teeth, namely, 11, 12, 21, and 22. A diffuse
rarefied area was seen in the anterior mid-palatal
region [Figure 3].
The patient was subjected to a complete hemogram,
which revealed normal values except a raised erythrocyte
sedimentation rate (ESR), which was 22 mm/hour. A
culture test was advised, to rule out fungal infection,
which gave a negative result for fungal growth. Under
local anesthesia, an incisional biopsy was performed and
the specimen was sent for histopathological examination.
The histopathological examination revealed areas of
necrosis and inflammatory granulation tissue along with
scattered lymphoid cells. There was
a diffuse mixed inflammatory infiltrate, consisting of
neutrophils, plasma cells, occasional eosinophils,
foamy macrophages, and extensive squamous
metaplasia of the salivary ducts and acini, with
prominent stromal mucin. No evidence of
granulomatous pathology or malignancy was found in
the biopsy tissue [Figure 4]. A final diagnosis of
necrotizing sialometaplasia was made. The patient
was advised symptomatic treatment and referred to
the Department of Oral Surgery for follow-up.
a b
Figure 2: (a) IOPA radiograph of 14, 15, 16, and 17; (b) IOPA radiograph
of 26, 27, and 28
Figure 4: Photomicrograph showing a mixed infl ammatory exudate
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Trivedi A et al.: Necrotizing sialometaplasia of the palate
Discussion
Necrotizing sialometaplasia is a self-limiting, variably
ulcerated, benign process affecting the minor salivary
glands. The most commonly proposed and generally
accepted etiology for NS relates to ischemia. [3] The
most widely accepted theory explaining the etiology is
ischemia of the blood vessels, leading to infarction of
the gland tissues.[4] The Armed Forces Institute of
Pathology has reported that 25 of 69 NS cases have
occurred following a surgical procedure. Other
predisposing factors include traumatic injuries, such
as a dental injection, blunt force trauma, denture wear,
alcohol and tobacco use, and upper respiratory
infections.[3] In the case reported here, the cause of the
lesion could not be identified. In 1996, Shigematsu et
al. found a relationship between the repeated
application of local anesthesia to a rat palate and
histological changes similar to those observed in NS.[5]
Necrotizing sialometaplasia shows a male predeliction,
with a male-to-female ratio of 2:1. According to literature
NS is diagnosed at an average age of 46 years. In some
cases, it may be seen in much younger individuals, as
mentioned in one case report, describing NS in a two-
year-old girl.[5] In the present case, the disease was
diagnosed in a 40-year-old male patient.
Clinically, necrotizing sialometaplasia is characterized
by a seemingly spontaneous presentation, most
commonly at the junction of the hard and soft palates.
Early in its evolution, the patient may note a tender
swelling. Subsequently, the mucosa breaks down as a
sharply demarcated deep ulcer with a yellowish-gray
lobular base. In the palate, the lesion may be unilateral
or bilateral; the diameter of an individual lesion
ranges from 1 to 3 cm. Most patients indicate mild
complaints of tenderness or dull pain. Healing is
generally slow and protracted, ranging from six to ten
weeks.[6] In the present case, the swelling was present
on the hard palate with a perforation and the swelling
was tender to palpation. Deviating from the common,
the present patient complained of pain, which was
severe, intermittent, sudden in onset, and radiating to
the left temporal region. The severity of the pain could
be related to the severity of the lesion in the present
case. Necrotizing sialometaplasia characteristically
shows a deep ulcer. Hence, the differential diagnosis
should include granulomatous diseases. Also
opportunistic infections in immunocompromised
patients may imitate NS.[7] In the present case, a
culture test was advised to rule out fungal infection,
which revealed negative results.
The histopathological appearance of NS shows coagulation
necrosis of the glandular acini, an inflammatory response,
Journal of Indian Academy of Oral Medicine & Radiology | Jul-Sep 2014 | Vol 26 | Issue 3
and pseudoepitheliomatous hyperplasia of the overlying
epithelium, with preservation of the lobular structure of
the gland.[7] On the basis of the histopathology, Anneroth
and Hansen et al. have classified NS into five stages:
Infarction, sequestration, ulceration, the reparative stage,
and the healed stage. During infarction, the glandular
acini undergo necrosis, resulting in the formation of an
ulcer. As the lesion heals there is proliferation of the
overlying epithelium, which is seen microscopically as
pseudoepitheliomatous hyperplasia. No sequestration
occurs if there is limited infarction. As healing takes
place, there is phagocytic activity of the histiocytes and
neutrophils, and appearance of granulation tissue. [4,8] On
the basis of the histopathological findings, the present
case appeared to be in the ulcerative stage at the time the
patient reported to us.
The classical histopathological differential diagnosis of
NS includes squamous cell and mucoepidermoid
carcinomas. However, there are certain benign lesions
requiring differentiation from NS. Necrotizing
sialometaplasia–like changes, without a lobular
configuration, have been reported in herpetic tracheitis
following intubation and have been called necrotizing
squamous metaplasia. Another rare benign disease that
has to be taken into account in the differential diagnosis
of NS is a disorder termed subacute necrotizing
sialadenitis.[9] Subacute necrotizing sialadenitis (SANS) is
a self-limiting inflammatory lesion of the minor salivary
glands, of unknown cause. There is a varied opinion by
authors on this entity. Although some consider it to be a
distinct entity, others consider it as a variant of NS. [10]
According to Lombardi et al. (2003), 22 cases of SANS
have been reported in literature (up to 2003); most of
them in the palatal salivary glands. They have reported
that SANS is more (77%) commonly seen in young men.
The clinical features include a unilateral, erythematous,
non-ulcerated swelling, which is mostly painful. Most
often the posterior hard palate and rarely the soft palate
is involved. The lesion is also self-limiting, which heals in
a few days or weeks.[10]
The management of NS includes symptomatic treatment
and the lesions undergo spontaneous healing within two
to three months. Surgical excision is not necessary. The
recurrence rate of NS is low. Reassessment of the lesions
that fail to resolve is important. [9]
Conclusion
As the clinical appearance of necrotizing sialometaplasia
can imitate other diseases, particularly malignant
neoplasms, histopathological examination is necessary to
confirm its diagnosis. The histopathological findings also
suggest the stage of the lesion.
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Trivedi A et al.: Necrotizing sialometaplasia of the palate

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How to cite this article: Trivedi A, Sunil MK, Gupta S, Garg S.
Necrotizing sialometaplasia of the palate: A case report. J Indian Acad
Oral Med Radiol 2014;26:355-8.
Source of Support: Nil, Conflict of Interest: Nil.

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