Journal of Oral and Maxillofacial Pathology Vol.

18 Issue 3 Sep - Dec 2014 420

CASE REPORT

Necrotizing sialometaplasia: A diagnostic dilemma!

Samir A. Joshi, Rajshekhar Halli, Vaishali Koranne1, Sarita Singh2
Department of Oral and Maxillofacial Surgery,1Department of Oral Medicine and Radiology, 2Department of Conservative Dentistry and
Endodontics, Bharati Vidyapeeth University Dental College and Hospital, Pune, Maharashtra, India

Address for correspondence: ABSTRACT

Dr. Samir A. Joshi, Necrotizing sialometaplasia (NS) is a benign, self-limiting inflammatory
9B, Vaijayanti Apartments, Kadam Plaza, reaction of salivary gland tissue which may mimic squamous cell carcinoma
Katraj, Pune - 411 046, Maharastra, India.
or mucoepidermoid carcinoma, both clinically and histologically, that creates
E-mail: samirjoshi1671@gmail.com
diagnostic dilemma leading to unwarranted aggressive surgery. Most commonly
Received: 23-06-2014 affected site is the minor salivary glands of the palate. The pathogenesis is
Accepted: 14-01-2015 unknown but is believed to be due to ischemia of vasculature supplying the
salivary gland lobules. A simple incisional biopsy is required to confirm the
histological diagnosis and to rule out more serious disease processes. It is a
self-limiting disease process and requires no treatment. It will be prudent to do
repeat biopsy in case if the lesion does not heal within 3 months.
Key words: Ischemia, necrotizing sialometaplasia, palate, self limiting
disease

INTRODUCTION CASE REPORT

Necrotizing sialometaplasia (NS) is a benign, self-limiting
inflammatory reaction of salivary gland tissue which may
mimic squamous cell carcinoma or mucoepidermoid
carcinoma, both clinically and histologically, that creates
diagnostic dilemma leading to unwarranted aggressive
surgery. [1] This pathological lesion was first reported
in 1973 by Abrams et al. as a reactive necrotizing
inflammatory process involving minor salivary glands of
the hard palate.[2]
A 35-year-old male reported with the history of non- healing
ulcer on left side of the palatal region from 15 days [Figure 1]
with fever and malaise. He had habit of gutkha and tobacco
chewing since 10 years.
On clinical examination there was single ulcerative lesion on
left side of the palate measuring about 1.5 cm in diameter.
Ulcer had indurated margins giving clinical picture of
malignant lesion. There were no palpable neck nodes.

NS occurs in only 0.03% of all biopsied oral lesions with
predominance in Whites.[1] The age prevalence of NS ranges
from 17 to 80 years with male predominance of 2:1.[3]
Most commonly affected site is the minor salivary glands of
the palate (80%). It could also be seen involving other sites
like retromolar pad, gingiva, lip, tongue, cheek, nasal cavity,
sinuses, larynx and trachea where the salivary gland tissue
is located. The lesion has also been reported to occur at sites
other than the salivary glands including lungs, breast and
skin.[4]
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DOI:
10.4103/0973-029X.151336
Incisional biopsy was performed and minimal bleeding was
noticed during the biopsy procedure. Histopathological
examination showed pseudoepitheliomatous hyperplasia of
the overlying epithelium. The underlying connective tissue
showed areas of necrosis, salivary glands showed clear acinar
lumina with squamous metaplasia of the ductal epithelium.
Inflammatory infiltrate was also seen. All these features lead
to the diagnosis of necrotizing sialometaplasia [Figures 2-4]
The ulcerative lesion had healed completely after period of
5 weeks [Figure 5].
DISCUSSION
NS is squamous metaplasia of the salivary gland ducts and acini
with ischemic necrosis of the salivary gland lobules which
occurs most frequently on hard palate. The pathogenesis is
unknown but it is believed to be due to ischemia of vasculature
supplying the salivary gland lobules. There are variety of
factors causing ischemia like direct trauma, administration of
Necrotizing Sialometaplasia
Figure 1: Intraoral view revealing the lesion on the left posterior part
of the palate
Figure 3: Histological photomicrograph showing squamous
metaplasia, necrosis and inflammatory cells. (H&E stain, x100)
local anesthetic, ill-fitting dentures, alcohol, smoking, cocaine
use, radiation, intubation, surgical procedures and upper
respiratory tract infections. The long-term use of salbutamol
might lead to dehydration and thinning of the mucosa, making
it more susceptible to local trauma.[5]
NS has wide range of differential diagnosis which includes
primary adenocarcinoma of the palate, squamous cell
carcinoma, subacute necrotizing sialadenitis (SANS), major
aphthous ulcer, mucoepidermoid carcinoma, secondaries
from adenocarcinoma of rectum, secondary syphilis and
tuberculous ulcer.
NS occurs spontaneously and the initial symptoms may include
fever, chills, malaise or swelling.[3] The posterior hard palate
is the most common site to be affected by NS and junction
of the hard and soft palate being the second most common
site. About two-thirds of the palatal lesions are unilateral;
however, bilateral synchronous and metachronus lesions are
not uncommon. Also the lesion could occur in the midline.[6]
The size may range from 0.7 to 5.0 cm (average 1.8 cm).[7]
Journal of Oral and Maxillofacial Pathology: Vol. 18 Issue 3 Sep - Dec 2014
Joshi, et al. 421
Figure 2: Histological photomicrograph showing pseudoeptheliomatous
hyperplasia (H&E stain, x100)
Figure 4: Histological photomicrograph showing ductal metaplasia.
(H&E stain, x400)
The diagnosis of NS is challenging and is based on a complete
clinical history and a well-oriented biopsy section. Biopsy
taken from base of the ulcer and the edge that is most
indurated and raised, yields the most representative sample.
A combination of histopathological and clinical findings is
often helpful in establishing the confirmatory diagnosis.
The diagnosis can be further supplemented via
immunohistochemistry demonstrating focal to absent
immunoreactivity for p53, low immunoreactivity
for MIB1 (Ki-67); and the presence of 4A4/p63 and
calponin-positive myoepithelial cells. Hematoxylin-eosin
staining remains the gold standard in histopathalogical
diagnosis of NS.[8]
Necrotizing sialometaplasia is characterized by lobular
necrosis and associated squamous metaplasia of ducts and
acini, with the preservation of lobular architecture. The areas
of necrosis consist of small pools of mucin surrounded by
neutrophils located within or adjacent to metaplastic ducts.
An inflammatory background is typically present.
Necrotizing Sialometaplasia
Figure 5: Intraoral view showing complete regression of the lesion
after 5 weeks
The mucosal surface is often ulcerated and
pseudoepitheliomatous hyperplasia and squamous metaplasia
of the excretory ducts are seen.
The pseudoepitheliomatous hyperplasia as well as the deeply
seated islands of metaplastic squamous epithelium often lead
to mistaken diagnosis of squamous cell carcinoma. In addition,
the squamous metaplasia of the ducts and acini juxtaposed
with residual mucous cells may suggest a mucoepidermoid
carcinoma.
In most cases, however, the lobular architecture, areas of
necrosis and mixed inflammatory background, together with
the distinctive epithelial nests, distinguish NS.[9]
As descrided by Anneroth and Hansen, the
histopathogenesis of NS has five histological stages:
Infarction, sequestration, ulceration, repair and healing.
Histological features exhibit a spectrum ranging from
ulceration, lobular necrosis, sequestration of necrotic
acini, pseudoepitheliomatous hyperplasia of adjacent
epithelium, squamous metaplasia of ductal epithelium and
inflammatory changes.[10] As these diagnostic criteria are
quite distinctive, proper care should be taken in diagnosis
of this lesion, so that misdiagnosis and unnecessary
radical treatment can be avoided.
Usually no treatment is required and the lesion heals by
secondary intention within 4-10 weeks (average 5.2 weeks).
Journal of Oral and Maxillofacial Pathology: Vol. 18 Issue 3 Sep - Dec 2014
Joshi, et al. 422
Even a full-thickness palatal lesion communicating with nasal
cavity resolves completely in 6 months.[3]
CONCLUSIONS
NS is a self-limiting disorder of salivary glands mostly affecting
the hard palate giving clinical presentation of malignant
neoplasm. Unfortunately it has been misdiagnosed clinically
and microscopically as a malignant neoplasm, resulting in
inappropriate and aggressive treatment. A simple incisional
biopsy is required to confirm the histological diagnosis and
to rule out more serious disease processes, hence, the role of
oral pathologist is of paramount importance. It is a self-limiting
disease process and requires no treatment. It will be prudent to do
repeat biopsy in case if the lesion does not heal within 3 months.
REFERENCES
1. Mesa ML, Gertler RS, Schneider LC. Necrotizing
sialometaplasia: Frequency of histologic misdiagnosis. Oral
Surg Oral Med Oral Pathol 1984;57:71-3.
2. Abrams AM, Melrose RJ, Howell FV. Necrotizing
sialometaplasia. A disease simulating malignancy. Cancer
1973;32:130-5.
3. Brannon RB, Fowler CB, Hartman KS. Necrotizing
sialometaplasia-A clinicopathologic study of sixty-nine cases
and review of the literature. Oral Surg Oral Med Oral Pathol
1991;72:317-25.
4. Krishna S, BK R. Necrotizing sialometaplasia of palate: A case
report. Imaging Sci Dent. 2011;41 (1):35-38.
5. Fowler CB, Brannon RB. Subacute necrotizing sialadenitis:
Report of 7 cases and a review of the literature. Oral Surg Oral
Med Oral Pathol Oral Radiol Endod 2000;89:600-9.
6. Daudia A, Murty GE. First case of full-thickness palatal
necrotizing sialometaplasia. J Laryngol Otol 2002;116:219-20.
7. Nah KS, Cho BH, Jung YH. Necrotizing sialometaplasia: Report
of 2 cases. Korean J Oral Maxillofac Radiol 2006;36:207-9.
8. Carlson DL. Necrotizing sialometaplasia: A practical approach
to the diagnosis. Arch Pathol Lab Med 2009;133:692-8.
9. Marx RE, Stern D. Oral and maxillofacial pathology a rationale
for diagnosis and treatment. 1st ed. New Delhi: Quintessence
Publishing Co, Inc; 2003. p. 509-10.
10. Anneroth G, Hansen LS. Necrotizing sialometaplasia. The
relationship of its pathogenesis to its clinical characteristics. Int
J Oral Surg 1982;11:283-91.
How to cite this article: Joshi SA, Halli R, Koranne V, Singh S.
Necrotizing sialometaplasia: A diagnostic dilemma!. J Oral Maxillofac
Pathol 2014;18:420-2.
Source of Support: Nil. Conflict of Interest: None declared.