- Case Report
- Open access
- Published:
Bilateral localization of necrotizing sialometaplasia: a case report
Cases Journal volume 2, Article number: 9068 (2009)
Abstract
Introduction
Necrotizing sialometaplasia is a rare disease first described in 1973. It involves salivary gland in the oral cavity.
Case presentation
We present a 32-year-old, white, male with rare bilateral localization of necrotizing sialometaplasia.
Conclusion
Clinically and histologically necrotizing sialometaplasia may mimic malignant lesions. In the above presented case, the patient's history was suggestive of a malignant process. Thus, knowledge and experience of both clinicians and histopathologists are essential to establish a correct diagnosis, which helps avoid radical surgery.
Introduction
Necrotizing sialometaplasia is an inflammatory process first described by Abrams et al. in 1973. The condition most frequently involves minor salivary glands in the hard palate region [1]; other anatomical locations, i.e., the oral cavity, minor salivary glands, and upper respiratory tract have also been observed [2]-[5]. An incidence of 0.03 to 0.063% has been reported [6, 7]. Although the ethiopathogenesis has not been fully elucidated, the lesion is usually regarded as resulting from an ischemic event [1, 8]. Predisposing factors include trauma, smoking, alcohol abuse [2, 8], and recurrent vomiting [9]. A patient usually presents with ulceration, and, although more rarely, swelling of the mucous membrane [2]. Histologically, there is necrosis of the minor salivary gland structures; however, lobular architecture is maintained. Squamous metaplasia is seen within the mucinous acini and ducts, which makes the condition similar to malignancy; mucin extravasation and inflammatory reaction are also present [1]. Necrotizing sialometaplasia is a self-healing disease usually resolving within a period of 3 to 12 weeks [2].
Case presentation
We report a case of 32-year-old white male who was seen in the Outpatient Clinic of the Maxillofacial Surgery Department in Katowice in April 2007 with a recurrent bilateral palate swelling which had appeared three years before. On the day of the examination the patient also complained of jelly-like nasal discharge and general malaise. The history was suggestive of neoplastic disease including a nondiagnosed palpable anal tumour and recent diagnostic procedures of lung lesions (no medical documentation available).
The clinical examination revealed a soft-elastic symmetrical affection in the area of the maxillary tuberosities; the covering mucous membrane was of normal appearance (Figure 1). The results of laboratory blood and urine tests were within normal range. Pantomogram and sinus X-ray did not detect any abnormalities. Facial CT showed a fluid-filled polypoid mucous membrane thickening of the medial maxillary sinus wall. The chest X-ray was normal. Rectoscopy revealed an anal varix to be first treated with anti-inflammatory agents, and then a surgical procedure. Fine needle aspiration biopsy showed 'epithelial nests and myxoid connective tissue suggesting a mixed tumor' (44035/BC Właszczuk). Therefore an incisional biopsy was performed with a diagnosis of necrotizing sialometaplasia (34685/K Pająk). The patient was hospitalized, and the lesion excised on the left side. Histologic features of the lesion were consistent with necrotizing sialometaplasia.
Conclusion
The location of necrotizing sialometaplasia was typical as most of the lesions were seen on the palate [2, 10]. However, bilateral symmetrical affection occurs rarely; Brannon et al. reported a 12% incidence [2]. The patient gave a 3-year history of symptoms recurrence, which is in accordance with the observations of Arguelles et al. [11]; however, our follow-up examinations did not confirm the patient's complaints.
Clinically and histologically necrotizing sialometaplasia may mimic malignant lesions. In the above presented case, the patient's history was suggestive of a malignant process. Thus, knowledge and experience of both clinicians and histopathologists are essential to establish a correct diagnosis which helps avoid radical surgery.
Consent
Written informed consent was obtained from the patient for publication of this case report and accompanying image. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
Abbreviations
- CT:
-
computed tomography.
References
Abrams AM, Melrose RJ, Howell FV: Necrotizing sialometaplasia. A disease simulating malignancy. Cancer. 1973, 32: 130-135. 10.1002/1097-0142(197307)32:1<130::AID-CNCR2820320118>3.0.CO;2-8.
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: 317-325. 10.1016/0030-4220(91)90225-2.
Prabhakaran VC, Flora RS, Kendall C: Pressure-induced necrotizing sialometaplasia of the parotid gland. Histopathology. 2006, 48: 464-465. 10.1111/j.1365-2559.2005.02253.x.
Pulse CL, Lebovics RS, Zagarelli DJ: Necrotizing sialometaplasia: Report of a case after lower lip mucocele excision. J Oral Maxillofac Surg. 2000, 58: 1419-1421. 10.1053/joms.2000.18280.
Romagossa V, Bella MR, Truchero C, Moya J: Necrotizing sialometaplasia (adenometaplasia) of the trachea. Histopathology. 1992, 21: 280-282. 10.1111/j.1365-2559.1992.tb00389.x.
Messa IM, Gertler RS, Schneider IC: Necrotizing sialometaplasia: frequency of histologic misdiagnosis. Oral Surg Oral Med Oral Pathol. 1984, 57: 71-73. 10.1016/0030-4220(84)90264-0.
Pająk J, Niedzielska I, Łangowska-Adamczyk H, Zielińska-Pająk E, Borgiel-Marek H, Mazur L: Necrotizing sialometaplasia in the salivary glands of the oral cavity - a frequent cause of diagnostic error. Czas Stomatol. 2002, 55: 579-585.
Imbery TA, Edwards PA: Necrotizing sialometaplasia: literature review and case reports. J Am Dent Assoc. 1996, 127: 1087-1092.
Solomon LW, Merzianu M, Sullivan M, Rigual NR: Necrotizing sialometaplasia associated with bulimia: case report and literature review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007, 103: 39-42. 10.1016/j.tripleo.2006.08.005.
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: 600-609. 10.1067/moe.2000.105943.
Arguelles MT, Viloria JB, Talens MC, McCory TP: Necrotizing sialometaplasia. Oral Surg Oral Med Oral Pathol. 1976, 42: 86-90. 10.1016/0030-4220(76)90034-7.
Author information
Authors and Affiliations
Corresponding author
Additional information
Competing interests
The authors declare that they have no competing interests.
Authors' contributions
IN involved in clinical examination, fine needle aspiration biopsy, writing the manuscript. TJ involved in literature review, writing the manuscript, clinical examination. JM involved. in writing the manuscript, clinical examination. All authors read and approved the final manuscript.
Rights and permissions
This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
About this article
Cite this article
Niedzielska, I., Janic, T. & Markowski, J. Bilateral localization of necrotizing sialometaplasia: a case report. Cases Journal 2, 9068 (2009). https://doi.org/10.4076/1757-1626-2-9068
Received:
Accepted:
Published:
DOI: https://doi.org/10.4076/1757-1626-2-9068