Necrotizing sialometaplasia: Report of five cases

FEMOPASE FL, HERNÁNDEZ SL, GENDELMAN H, CRISCUOLO MI, LÓPEZ DE BLANC SA .NECROTIZING SIALOMETAPLASIA: REPORT OF FIVE CASES. MED ORAL 2004;9:304-8.

ABSTRACT

Necrotizing sialometaplasia (NS) is a self-limiting inflammatory disease, that involves salivary glands, more frequently the minor ones. Although its etiopathogenesis remains still unknown some authors suggest that a physico-chemical or biological injury on the blood vessels may produce ischemic changes, leading to infarction of the gland and its further necrosis. Its clinical and histologic feature resemble malignancy. Clinically it may appear like an ulcer with slightly elevated irregular borders and necrotic base. Histologic features are squamous metaplasia of ducts and acini and a pseudoepitheliomatous hyperplasia of the overlying mucosa. These characteristics may induce to an inapropiated diagnosis of malignant neoplasia. A correct diagnosis to avoid mutilant surgical treatments is essential, considering that it is a self-limiting disease. In this report we describe five cases of NS in females, located in minor glands of the palate.

Key words: Oral ulcer, necrotizing sialometaplasia.

INTRODUCTION

Necrotizing sialometaplasia (NS) is a self-limiting, benign, inflammatory disease of the minor salivary glands. It was first reported by Abrams et al. in 1973(1) and Dunlop et al. in 1974(2), like a disease of minor salivary glands of the hard palate, although some cases were later reported in different sites of the oral cavity(3-6), in major salivary glands(7,8), and in sinusal mucosa(9) and larynx(10).

Although the etiology is not clear, many authors suggest that a physico-chemical or biological injury on the blood vessels would produce ischemic changes, leading to infarction of the gland tissues with posterior necrosis(11), inflammation and intent of repairing, inducing metaplasia, changes in ducts and further cicatrization(12). Sharp direct local trauma like those produced by intubation, local anestesia due to either the injection or the vasocontriccion, surgery procedures(13,14), use of unadapted dental prothesis, violent or provocated vomiting like in patients with bulimia(15), infectious processes(16), radiotherapy, use of tobacco(17) and cocaine among others(18,19) could be the factors involved in this vessels alteration resulting in local ischemia. Some authors find a relationship betwen NS and diseases like diabetes(8), chronic alcoholism(20,21); others investigators consider NS as the result of terminal leucoqueratotic condition(22).

Clinically NS may look like an ulcer or as a tumor(23), being the first one the most frequent, presenting a crateriform aspect, with indurated and well-delimitated shapes, commonly located in palate, and sometimes bilateral(24,25). The most common histopathologic features are: squamous metaplasia of ducts and mucous acini, lobular coagulation necrosis with preservation of the lobular architecture(26), granulation tissue with inespecific inflammatory infiltrate and pseudoepitheliomatous hyperplasia of the overlying mucosa(27). No palpable lymph nodes were described but some cases with painless nodules have been reported(8). Although the relapses are not frequent, a case with recurrent lesions was published(16).

The clinical aspect and some histological features resembling oral malignant neoplasia like adenoid cystic carcinoma(11,14,17), mucoepidermoid carcinoma or squamous cell carcinoma, emphasize the importance of this benign inflamma-tory disease. Therefore an incorrect diagnosis may induce to unnecesary mutilant treatment.

REPORT OF CASES

Case 1: A 56-year-old woman was referred for an ulcerated lesion in left posterior hard palate of twenty days duration. The patient informed us that she suddenly felt very sharp pain in the area that slowly decreased with time; after that she did some local treatments with antiseptic washes without any improvement. Clinical examination revealed an ulcer measuring 13 x 10 mm, necrotic base, with violet whitish borders moderately firm and painless to palpation (Fig 1).

A negative to malignancy citology showed inflammatory cells, germs and necrotic tissue. The histopathologic exam revealed: important coagulation necrosis, with the outlines of the acini still visible and a little celular secretion, slight squamous metaplasia in the ductus, surrounded by inflammatory tissue with large amount of polimorfe neutrophils and eosinophils. Vessels with thick walls, some of them with trombosis and slight non neoplasic pseudoepiteliomatous hyperplasia was also observed (Fig 2). Ten days latter she had an important improvement with total resolution of the lesion in 7 weeks.

Case 2: A thirty nine-year-old-woman showed an ulcerated lesion in the palate gum next to the up left first premolar, with ten days evolution that provoqued to her a neuralgic sharp pain. She consulted to the dentist for a painfull swelling of the area; he made the endodontic treatment of the 24 element, and after that, when the mucosa lesion was detected the patient was referred to the Stomatology Service. Clinical examination revealed the presence of an irregular shaped ulcer, with necrotic base and soft eritematous borders. Grade II and III citology was informed. The incisional biopsy confirmed NS diagnosis two weeks later; the base of the lesion was covered by granulation tissue with a strong tendency to cicatrize.

Case 3: A 78-year-old female, was referred by a one month evolution ulcerated lesion in soft palate behind the unadapted prothesis. She had Parkinson's disease and oral parafunctional habits. She informed us to have had initially a painfull throbbing sensation, without pain at the moment of the clinical examination. We observed an erosive lesion, heart shaped, with slightly increased leucoedematous margins, firm to palpation.

The citology was grade II and the incisional biopsy confirmed NS diagnosis; cicatrization of the lesion took place at the 7th week.

Case 4: An insulinodependent diabetic, hypertense medically treated 60-year-old woman consulted us ten days after the appearence of a sudden palate lesion after trauma during feeding. Two years before she had had glomerulonefritis followed by chronic renal failure; she had also a mamma carcinoma surgically and radiotherapically treated. One month before she had stopped the treatment of diabetes. Ten days before consulting us, she noticed the sudden appearence of a lesion on palate after trauma during feeding. The clinical exam revealed substance loss on the left part of the hard palate rougs area; it was painfull to palpation with slightly erithematous borders and necrotic plug in the base. No lymph nodes were palpated. The biopsy was consistent with NS; six weeks later the area was completely epithelized and slightly depressed. During 8 months the patient came for controls and reported to have hypersensibility in the area .

Case 5: A 17-year-old woman was referred by her dentist for a three months evolution lesion with irradiated pain to the ipsilateral ear. The clinical exam revealed a 10x10mm red violet tumoral lesion, tender to palpation and surrounded by erithematous mucosa (fig.3). After the lesion was detected, we knew that the patient was bulimic, she admitted to induce vomiting. Routine analysis indicated normal values. Oclusal and ortopanthomography radiographs did not revealed abnormal findings. Escisional biopsy was performed and NS was confirmed.

DISCUSSION

Although the etiology of NS is still not fully resolved, it is generally accepted that an acute loss of blood supply to the minor salivary glands is the major cause of this lesion. In cases 3 and 4 it could be related to local trauma during feeding and in case 5 to trauma induced by bulimia. Additionally case 4 diabetes could have been as predisposing systemic factor. Acinar necrosis induced by ischemia is the earliest event followed by liberation of mucus and the resulting inflammatory response.

NS has been reported more frequently in middle-age male(27) and less in female(28); nevertheless the cases described in this paper were four females one of them adolescent. Hard palate is the most frequent localization reported(10,27,28) like in three of our cases. Only few cases were described in other locations as lower lip mucosa(5) or submaxillary gland(7), followed by parotid gland, retromolar pad, sublingual region, lower lip, tongue, nasal cavity(3,14) maxillary sinus, soft palate and larynx (14,7,11). (See Table 1)

Although NS may present as a nonulcerated swelling, the most typical presentation is a deep ulcer(29) as in the four first cases of this presentation; in some others cases it may appear as an exophytic lesion(30) like in our last patient. In general NS lesions are uniques, although there are some bilateral lesions(2,11). The ulcers resolve without any treatment in an average of 7 to 10 weeks(18), similar to the evolution observed in our patients. Only one case of NS with an underlying malignancy was reported(31). The differential diagnosis must be done specially with mucoepidermoid carcinoma, adenoid cystic carcinoma and Sutton disease. It is important also to distinguish NS from infectious diseases as syphilis and tuberculosis(32).

Finally it should be considered the differential diagnosis with subacute necrotizing sialadenitis (SANS). This is a nonspecific inflammatory condition of unknown etiology. Microscopically is characterized by focal acinar cell necrosis secondary to the inflammatory process and slight atrophy of ductal cells; neither ductal squamous metaplasia nor seudoepiteliomatous hyperplasia were observed in SANS. Clinically they are nodular lesions non ulcerated located in palatal salivary glands accompanied by an abrupt onset of pain. Patients are most often young men, who have spent several weeks living in close quarters such as military barracks(33,34).

Clinicians and pathologists must be aware to avoid errors in the diagnosis and treatment of these benign pathologic conditions.

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