CASE REPORT

Two well demarcated ovoid radiolucent lesions (right, 3.9×1.5 cm and left, 2.9×1.1 cm) were found incidentally in the anterior mandible of a 44-year-old female patient on her panoramic dental X-ray (Fig. 1). The patient did not show any symptom of radiolucencies. The radiological findings were interpreted as radicular cysts. Two fragments of tissue from the cavities were submitted for histological diagnosis. The biopsied tissue fragments showed normal salivary gland tissue with mixed serous and mucinous cells. These findings were otherwise unremarkable, except for the small amount of lymphoid cell infiltration that was discovered (Fig. 2). The clinical, radiological, and pathological findings were consistent with Stafne bone cavity.

DISCUSSION

Since Stafne bone cavities were first reported in 1942, most cases have been found in the posterior mandible.3 However, these cavities were found also in the anterior portion of mandible in 1957 by Richard and Ziskind.4 Until now, only approximately fifty cases of anterior Stafne bone cavities have been reported.2

Similar to Stafne bone cavities located elsewhere in the mandible, anterior lesions show a wide age range (18 to 68 years) and higher prevalence in males (3:1, males:females).2 Most Stafne bone cavities are diagnosed on radiology, and only clinical follow-up with additional radiographic examination is sufficient unless there are symptoms or progression. The rarity of anterior Stafne bone cavities causes confusion and can hinder accurate and prompt diagnosis of this condition. Because the cavities resemble other periapical radiolucencies, they can appear similar to other cysts or tumors.2,5,6 The differential diagnosis of bilateral lesions includes, salivary gland tumors, fibro-osseous lesions, traumatic bone cysts, central giant cell lesions, hyperparathyroidism, ameloblastoma, eosinophilic granuloma, hemangioma, myxoma aneurysmal bone cyst, multiple myeloma, benign neurogenic tumors and arteriovenous fistula. Unlike tumors or the cysts, most Stafne bone cavities are asymptomatic and are therefore found accidentally.

In most cases, the content of the cavities was salivary gland tissue, and these lesions were sometimes referred to as heterotopic salivary glands. The salivary gland tissue contents suggeseted congenital malformation theory for the pathogenesis of the cavities,1 which states that the salivary gland tissue is congenitally entrapped within the mandible. Pressure from adjacent structures, including the hypertrophied salivary glands and facial arteries, has also been suggested as a possible pathogenic cause of Stafne bone cavities.7 A large-scale study of mandibular marrow tissue revealed the presence of intraosseous salivary gland tissue.8 As the authors of the article pointed out, such intraosseous heterotopias can serve as a pathogenic explanation and this finding can be interpreted as evidence supporting the former pathogenic theory of Stafne bone cavity formation. Our case lacks previous history and therefore, we were not able to determine the cavity's pathogenic cause. However, lymphocytic infiltration, which has been suggested as one of the causes of hypertrophied salivary glands in the pressure-induced acquired pathogenesis theory, is present in our case and therefore might be interpreted as a backup evidence for the theory.3

Although most Stafne bone cavities generally do not present any complications, there is one report of a Stafne bone cavity harboring pleomorphic adenoma.9 If a surgical specimen of the Stafne bone cavity is acquired, histological examination of the entire specimen is preferable for the detection of a possible salivary gland tumor within the specimen.

Because anterior Stafne bone cavity is very rare, accurate diagnosis of the lesion is challenging for clinicians.10 Pathologists should be aware of this lesion, because biopsies are performed in some cases and the cavity can harbor a salivary gland tumor.

REFERENCES

• 1. Apruzzese D, Longoni S. Stafne cyst in an anterior location. J Oral Maxillofac Surg 1999; 57: 333-338. ArticlePubMed
• 2. Turkoglu K, Orhan K. Stafne bone cavity in the anterior mandible. J Craniofac Surg 2010; 21: 1769-1775. ArticlePubMed
• 3. Sisman Y, Miloglu O, Sekerci AE, Yilmaz AB, Demirtas O, Tokmak TT. Radiographic evaluation on prevalence of Stafne bone defect: a study from two centres in Turkey. Dentomaxillofac Radiol 2012; 41: 152-158. ArticlePubMedPMC
• 4. Richard EL, Ziskind J. Aberrant salivary gland tissue in mandible. Oral Surg Oral Med Oral Pathol 1957; 10: 1086-1090. ArticlePubMed
• 5. Dorman M, Pierse D. Ectopic salivary gland tissue in the anterior mandible: a case report. Br Dent J 2002; 193: 571-572. ArticlePubMed
• 6. Sisman Y, Etoz OA, Mavili E, Sahman H, Tarim Ertas E. Anterior Stafne bone defect mimicking a residual cyst: a case report. Dentomaxillofac Radiol 2010; 39: 124-126. ArticlePubMedPMC
• 7. Buchner A, Carpenter WM, Merrell PW, Leider AS. Anterior lingual mandibular salivary gland defect: evaluation of twenty-four cases. Oral Surg Oral Med Oral Pathol 1991; 71: 131-136. ArticlePubMed
• 8. Bouquot JE, Gnepp DR, Dardick I, Hietanen JH. Intraosseous salivary tissue: jawbone examples of choristomas, hamartomas, embryonic rests, and inflammatory entrapment: another histogenetic source for intraosseous adenocarcinoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000; 90: 205-217. ArticlePubMed
• 9. Simpson W. A Stafne's mandibular defect containing a pleomorphic adenoma: report of case. J Oral Surg 1965; 23: 553-556. ArticlePubMed
• 10. Katz J, Chaushu G, Rotstein I. Stafne's bone cavity in the anterior mandible: a possible diagnostic challenge. J Endod 2001; 27: 304-307. ArticlePubMed

Fig. 1

Panoramic dental X-ray shows two radiolucencies beneath the apices of the canine and premolars in the right and left anterior mandible.

Fig. 2

Biopsied cavity contents are composed of mixed seromucinous salivary gland tissue with lymphocytic infiltration.

Figure & Data

References

Citations

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