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Necrotizing sialometaplasia: a potential diagnostic pitfall.

Abstract

Necrotizing sialometaplasia is a benign, spontaneously healing inflammatory condition. It usually affects the hard palate, and it can mimic a malignancy both clinically and histologically. We report a new case of this rare disorder.

Introduction

Necrotizing sialometaplasia is a benign inflammatory process that involves the minor salivary glands, primarily those of the hard palate. The major importance of this condition is the danger of misdiagnosis, because it may clinically and histologically mimic a malignancy. (1-3)

Case report

A 52-year-old man was referred to the otolaryngology clinic with a 1-week history of an asymptomatic deep ulcer in the hard palate. He had a history of tobacco use, but no history of overt trauma. He used a total upper dental replacement.

The physical examination was remarkable for the presence of a 45 x 30-mm necrotic ulcer (figure, A). A blood count and measurement of cross-reacting protein did not demonstrate any evidence of pathology. No communication between the ulcer and the nasal cavity was found endoscopically. Ultrasonography detected no enlarged lymph nodes of the neck, and computed tomography of the hard palate and nasal cavity did not demonstrate any bone pathology.

[FIGURE A OMITTED]

A biopsy of the ulcer was taken, and a diagnosis of squamocellular metaplasia was made. Because cancer was clinically suspected, a second incisional biopsy was performed. The second biopsy revealed no evidence of any neoplastic changes; the histologic features of the lesion were consistent with necrotizing sialometaplasia.

At follow-up 10 days later, the lesion showed signs of spontaneous resolution (figure, B). It was completely healed within 8 weeks without the use of any medication. A 35-mm scar persisted in the hard palate for more than 3 years.

[FIGURE B OMITTED]

Discussion

Necrotizing sialometaplasia was first described by Abrams et al in 1973. (4) This condition usually affects the minor salivary glands of the hard palate (77% of cases); it has also been reported in the major salivary glands, nasal cavity, lip, trachea, and larynx. (1,5) It was Daudia and Murty who reported the first case with full-thickness palatal involvement. (6)

According to the most widely accepted pathophysiologic theory, necrotizing sialometaplasia occurs as a result of ischemia of the vasculature that supplies the salivary gland lobules. (1,2,5) Our patient presented with two possible etiologic factors: a history of smoking and the possibility of hard palate trauma caused by the dental prosthesis.

Patients with necrotizing sialometaplasia exhibit a spectrum of histologic findings, ranging from coagulation necrosis of the salivary gland acini in early lesions to squamous metaplasia of ducts and reactive fibrosis in late lesions. (2,4,5) Features that distinguish necrotizing sialometaplasia from a malignant process are its overall lobular morphology, the generally bland appearance of the squamous nests, and evidence of residual ductal lumina in the nests. (1)

Clinical management involves adequate biopsy, observation, and reassurance. Awareness of this potential diagnostic pitfall is of great importance because an inaccurate histopathologic diagnosis can result in inappropriate or unnecessary treatment, ranging from conservative excision to total maxillectomy. (1,2,7,8) Lesions typically resolve in 4 to 10 weeks. No treatment is required other than an analgesic for a patient whose lesion is painful. (2,7) Repeat biopsy is indicated for a patient whose lesion fails to resolve. (7)

This case report illustrates the importance of a careful correlation between clinical and pathologic findings in order to avoid misdiagnosis. (8)

References

(1.) Sandmeier D, Bouzourene H. Necrotizing sialometaplasia: A potential diagnostic pitfall [letter]. Histopathology 2002;40:200-1.

(2.) Imbery TA, Edwards PA. Necrotizing sialometaplasia: Literature review and case reports. J Am Dent Assoc 1996;127:1087-92.

(3.) 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.

(4.) Abrams AM, Melrose RJ, Howell FV. Necrotizing sialometaplasia. A disease simulating malignancy. Cancer 1973;32:130-5.

(5.) Seifert G. [Diagnostic pitfalls in benign and malignant salivary gland diseases. Their significance for prognosis and therapy]. Mund Kiefer Gesichtschir 1998;2:62-9.

(6.) Daudia A, Murty GE. First case of full-thickness palatal necrotizing sialometaplasia. J Laryngol Otol 2002;116:219-20.

(7.) Schroeder WA Jr. Necrotizing sialometaplasia. Otolaryngol Head Neck Surg 1994;111:328-9.

(8.) Kyrmizakis DE, Doxas PG, Hajiioannou JK, Papadakis CE. Palate ulcer due to mucormycosis. J Laryngol Otol 2002;116:146-7. Pavel Kominek, MD, PhD; Petr Blasch, MD

From the ENT Department (Dr. Kominek) and the Department of Pathology (Dr. Blasch), City Hospital, Frydek-Mistek, the Czech Republic.

Reprint requests: Pavel Kominek, MD, PhD, ENT Department, City Hospital, El. Krasnohorske 321, 738 01 Frydek-Mistek, Czech Republic. Phone: 420-558-415-770; fax: 420-558-415-010; e-mail: kominek@nemfm.cz
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Article Details
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Author:Blasch, Petr
Publication:Ear, Nose and Throat Journal
Geographic Code:4EXCZ
Date:Sep 1, 2006
Words:773
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