o t o l a r yn go l o g i a p o l s k a 6 8 ( 2 0 1 4 ) 1 5 4 – 1 5 6

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Case report/Kazuistyka

Necrotizing sialometaplasia: A rare lesion that

mimics oral cancer clinically andhistopathologically

Nakul Uppal *, Mohan Baliga

Department of Oral and Maxillofacial Surgery, Manipal College of Dental Sciences, Mangalore (Manipal University),India

a r t i c l e i n f o

Article history:

Received: 19.06.2013

Accepted: 30.07.2013

Available online: 05.08.2013

Keywords:� Oral cancer� Ulcero-proliferative growth� Misdiagnosis� Biopsy� Screening� Risk factors

a b s t r a c t

With screening for oral cancer being increasingly performed by physicians, ulceroprolife-

rative lesions of the oral cavity that resemble carcinoma may present a diagnostic

challenge. We present one such case wherein the lesion resolved spontaneously after

incisional biopsy. A clinical diagnosis of necrotizing sialometaplasia was confirmed by

histopathology. General medical practitioners and otolaryngologists should consider this

rare lesion in the work up of a suspicious oral ulcer. Decisions must be based on biopsy

and clinicians should resist the urge to make diagnoses unsupported by biopsy or at

least toluidine blue staining.

© 2013 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by

Elsevier Urban & Partner Sp. z o.o. All rights reserved.

Introduction

Patients with suspicious lesions in the oral cavity mightreport to physicians instead of dentists for diagnosis [1].Individuals of lower socio-economic status prefer to avoiddental visits due to perceived higher costs. One rare orallesion that closely resembles oral malignancy in clinicalpresentation is necrotizing sialometaplasia. Misleading clini-cal appearance as well as history of tobacco and alcoholabuse might bias the clinical diagnosis toward oral cancerand result in unnecessarily radical excision of the lesion.We report one such case.

* Corresponding author at. Manipal University, Lighthouse Hill Road, KE-mail address: drnakul@gmail.com (N. Uppal).

0030-6657/$ – see front matter © 2013 Polish Otorhinolaryngology - Head and Neck Su

http://dx.doi.org/10.1016/j.otpol.2013.07.004

Report of a case

A 68-year-old male presented with a large palatal ulcer onemonth after a maxillary tooth was extracted. Having abusedtobacco and alcohol for several years, he mentioned appre-hension of having developed oral cancer. The ulcerouslesion, noticed 3 weeks earlier, measured 2 [8_TD$DIFF]cm � 2 cm, hadelevated margins, a necrotic center and was painless (Fig. 1).A clinical diagnosis of oral carcinoma was made based onhistory and physical examination findings. Incisional biopsywas performed to confirm diagnosis. At recall, however, thelesion was noted to have resolved spontaneously and

arnataka 575 001, India. Tel.: +91 984 562 8027.

rgery Society. Published by Elsevier Urban & Partner Sp. z o.o. All rights reserved.

[(Fig._1)TD$FIG]

Fig[1_TD$DIFF]. 1 – Large ulceroproliferative growth on hard palateappears suspiciously like malignancy in this smoker

o t o l a r yn g o l o g i a p o l s k a 6 8 ( 2 0 1 4 ) 1 5 4 – 1 5 6 155

completely (Fig. 2). Histo-pathological diagnosis suggestednecrotizing sialometaplasia, an uncommon but non-neo-plastic ulceration of tissues overlying minor salivary glands.

Discussion

Necrotizing sialometaplasia is a reactive, inflammatory,non-neoplastic process that arises from ischemic necrosis ofminor salivary glands. The hard palate is the commonestoral site affected. The characteristic clinical presentation ofnecrotizing sialometaplasia is due to non-neoplastic ulcera-tion of tissues overlying minor salivary glands, causedpossibly by trauma. Previous maxillary dental extraction inour patient would have necessitated palatal injection oflocal anesthetic containing epinephrine. The resultant ische-mia of palatal vasculature is thought to be the reason forlocalized necrosis of tissues [2]. Patients might fear the

[(Fig._2)TD$FIG]

Fig[2_TD$DIFF]. 2 – At a later recall visit, lesion had disappeared entirely

growth is cancerous while clinicians are misled into assu-ming the lesion is malignant because the margins areelevated, center is ulcerated and onset is spontaneous. Lackof pain – an early feature of malignant ulcers – is commonin necrotizing sialometaplasia and reinforces the clinician'sinadvertent misdiagnosis [3]. History of alcohol and tobaccoabuse reinforces the suspicion of malignancy. However, thecondition is known to occur spontaneously [2].

No treatment is required; lesions of necrotizing sialome-taplasia resolve spontaneously a few weeks later [4]. This isrelevant since there is increasing emphasis in modernliterature on the detection of oral cancers [1, 5] by clinicalexamination alone as a screening method.

Microscopic features of necrotizing sialometaplasia canalso mimic oral squamous cell carcinoma and furthercomplicate diagnosis. Squamous metaplasia of salivaryductal epithelium may lead an inexperienced pathologist tomisinterpret the histological section. When squamousmetaplasia are seen along with viable salivary acini, anerroneous diagnosis of mucoepidermoid carcinoma mayresult [3]. However, necrosis along with preservation oflobular architecture of salivary acini help to identify thelesion.

There is no specific treatment required for necrotizingsialometaplasia. Lesions disappear spontaneously, presu-mably after ischemic injury heals. Recurrence does notoccur. Reassuring the patient is probably important andwound irrigation has been suggested [3].

Conclusion

Missing early malignancy is disastrous, but physicians,dentists and surgeons also need to avoid ‘‘overdiagnosing’’lesions; that would serve only to alarm patients and wouldincrease the number of unwarranted excisional biopsieswhere limited incisional biopsies would have sufficed.Necrotizing sialometaplasia is a self-limiting benign lesionthat requires no treatment; arriving at a correct diagnosis isthe difficulty. Lesions are clinically mistaken to be oralsquamous carcinoma whereas histological picture mayresemble mucoepidermoid tumor of minor salivary glands.Localized trauma appears to a risk factor for this uncommonoral lesion. Recent oral surgery, ill-fitting dentures, endotra-cheal tubes[9_TD$DIFF], etc. have been listed as causes [2].

Anneroth and Hansen described five histologic stages ofnecrotizing sialometaplasia [6]. It is necessary for thepathologist to examine histologic sections after a thoroughreview of patient's history and physical examination. Biopsyremains the gold standard for diagnosis. Toluidine bluestaining would be a simple, non-surgical way to screensuspicious lesions but necrotizing sialometaplasia has beenreported to occur as a non-ulcerated swelling in children [3].Clinicians should also bear in mind that each stage mightpresent with different clinical features. A recent studyestimated that nearly one-half of all ‘‘clinical diagnoses’’ oforal lesions are incorrect [7]; it is necessary to correlate allsources of patient information in making clinical diagnosesto prevent unnecessarily mutilating surgery in necrotizingsialometaplasia.

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Authors' contributions/Wkład autorów

NU [10_TD$DIFF]and MB contributed in the study design, acceptance of [11_TD$DIFF]

the final manuscript version literature search.

Conflict of interest/Konflikt interesu

None declared.

Financial support/Finansowanie

None declared.

Ethics/Etyka

Approved by [12_TD$DIFF]Institutional Ethics Committee, ManipalCollege of Dental Sciences, Mangalore[13_TD$DIFF].

r e f e r e n c e s / p i �s m i e n n i c t w o

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[2] Sowmya K, Ramnarayan BK. Necrotizing sialometaplasia ofpalate: a case report. Imaging Sci Dent 2011;41:35–38.

[3] Ylikontiola L, Siponen M, Salo T, Sándor GKB.Sialometaplasia of the soft palate in a 2-year-old girl. J CanDent Assoc 2007;73:333–336.

[4] Neville BD, Damm DD, Allen CM, Bouquot JE. Salivary glandpathology. In: Neville BD, Damm DD, Allen CM, Bouquot JE,editors. Oral and maxillofacial pathology. 2nd ed., Saunders:Pennsylvania; 2002. p. 405–406.

[5] Decuseara G, MacCarthy D, Menezes G. Oral cancer:knowledge, practices and opinions of dentists in Ireland.J Ir Dent Assoc 2011;57:209–214.

[6] Anneroth G, Hansen LS. Necrotizing sialometaplasia: therelationship of its pathogenesis to its clinical characteristics.Int J Oral Surg 1982;11:283–291.

[7] Kondori I, Mottin RW, Laskin DM. Accuracy of dentists in theclinical diagnosis of oral lesions. Quintessence Int2011;42:575–577.