Anterior Lingual Mandibular Bone Cavity as a Diagnostic Challenge: Two

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J Oral Maxillofac Surg 68:201-204, 2010

Anterior Lingual Mandibular Bone Cavity as a Diagnostic Challenge: Two

Case Reports

Pit J. Voss, MD, DDS,* Marc C. Metzger, MD, DDS,†

Dirk Schulze, PhD, MD, DDS,‡ Denys J. Loeffelbein, MD, DDS,§

Christoph Pautke, MD, DDS,储 and Bettina Hohlweg-Majert, MD, DDS¶

In 1942, Stafne was the first to report the presence of bone cavities in 35 patients.¹ These usually create a well-demarcated radiolucency in the posterior mandibular angle and are synonymously known under different names in the literature: Stafne bone cyst, static bone cavity, latent bone cyst, lingual mandibular bone cavity, mandibular embryonic defect, idiopathic bone concavity of the mandible, and developmental submandibular gland defect of the mandible.

These cavities occur in the area between the mandibular first molar and the mandibular angle below the mandibular canal outline. Other than this radiographic presence, they have no real clinical presentation. They are innocuous convexities in the lingual cortex of the mandible that would be of no conse- quence if they did not appear as radiolucency. It may be difficult to distinguish them from more serious conditions such as cysts and benign or malignant tumors. Surgical exploration has shown that this hard

tissue configuration represents a focal concavity of the cortical bone on the lingual side of the mandible.

In most cases, regular salivary gland tissue was the histologic finding.² This suggested a developmental origin: 1 theory was that part of the submandibular gland was entrapped in the lingual mandibular cortex.

Muscle, fibrous connective tissue, blood vessels, fat, or lymphoid tissue were also found in some cases.³ Stafne bone cavity is not considered extremely rare.

The incidence ranges from 0.1% to 1.3%.⁴

The first report of Stafne bone cavity in the premo- lar region was issued by Richard and Ziskind in 1957.⁵ The sublingual gland or aberrant salivary gland tissue has also been associated with this bony cavity in the anterior mandible.⁶

In the anterior mandible, the mandibular canal as an anatomic landmark for identifying latent bone cysts is absent. Thus, a diagnosis of Stafne bone cavity is difficult on plain radiographs; and endodontic treatment, bone trephining, and bone exploration may result from an incorrect diagnosis.^7,8 Cone-beam computed tomography, a fast developing 3-dimensional imaging technique, is becoming increasingly available in dental practice, being used most frequently in the planning of dental implants. Even with a low radiation dose, the quality of the hard tissue image is comparable to spiral computed tomograms in the bone window. However, soft tissues cannot be distinguished from each other.

Our objective is to describe 2 new cases of Stafne bone cavity in the anterior mandible and to discuss the differential diagnosis process. We introduce cone- beam computed tomography as a suitable noninvasive diagnostic and follow-up modality for this bony configuration in the anterior mandible.

Case 1

A 58-year-old male patient consulted our outpatient clinic after his dentist noticed a sharply bordered unilateral radi-

*Resident, Department of Oral and Maxillofacial Surgery, Univer- sity Hospital Freiburg, Freiburg, Germany.

†Faculty, Department of Oral and Maxillofacial Surgery, Univer- sity Hospital Freiburg, Freiburg, Germany.

‡Assistant Professor, Department of Head and Neck Diagnosing, University Hospital Freiburg, Freiburg, Germany.

§Resident, Department of Oral and Maxillofacial Surgery, Tech- nische Universität München, Munich, Germany.

储Faculty, Department of Oral and Maxillofacial Surgery, Tech- nische Universität München, Munich, Germany.

¶Consultant, Department of Oral and Maxillofacial Surgery, Tech- nische Universität München, Munich, Germany.

Address correspondence and reprint requests to Dr Hohlweg- Majert: Department of Oral and Maxillofacial Surgery, Technische Universität München Ismaninger Str 22, 81675 Munich, Germany;

e-mail:majert@mkg.med.tum.de

doi:10.1016/j.joms.2009.03.017

VOSS ET AL 201

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olucency in the mandible. The patient’s history was not significant and the lesion was asymptomatic. The lesion was located in an edentulous zone between the lower right canine and the first molar and appeared to have no relation to the adjacent teeth (Fig 1). However, the 1.5- ⫻ 3-cm lesion in the projection of the mandibular canal seemed to be associated with the same. On cone-beam computed tomogram a lingual osteolysis was suspected, interrupting the lingual compact bone and affecting the mandibular canal with only poorly defined margins (Fig 2). Because a neoplasm could not be completely excluded, a biopsy was taken under general anesthesia. Histologic examinations showed a mixed salivary gland tissue with a slight chronic infection in the area of the ducts and periductal fibrosis.

Case 2

A 50-year-old female patient was referred to the outpatient clinic of the department of oral and maxillofacial surgery with a suspect radiolucency in the region of the left lower canine. A few weeks previously, the radiolucency was found on a panoramic radiograph during routine dental

examination (Fig 3). The patient’s dental and medical his- tories were uneventful. No older radiographs were available for comparison. A periapical radiograph was obtained. The 20-⫻ 15-mm lesion appeared to relate to the adjacent tooth, which was asymptomatic. There were no abnormal findings and all teeth in the region responded within normal limits to the temperature pulp test. The differential diagnosis was osteoporotic focal defect of the marrow, apical granuloma, residual cyst, or latent bone cyst. An occlusal radiograph and ultrasound did not provide further information, so cone-beam computed tomography was performed (Fig 4).

The radiologic examination showed a 20-mm-wide and 15-mm-long bony impression on the lingual side of the anterior mandible, which did not have any relation to an adjacent tooth. A follow-up radiograph taken 5 months later did not show any alteration of the radiolucency. Therefore, no invasive therapy was performed on this patient.

Discussion

Ectopic salivary gland tissue has been described on many sites within the head and neck region including the lateral and posterior neck, tongue, middle ear

FIGURE 1. Panoramic radiograph demonstrating a sharply bor- dered unilateral radiolucency in the mandible.

Voss et al. Anterior Lingual Mandibular Bone Cavity. J Oral Maxillofac Surg 2010.

FIGURE 2. Lingual defect demonstrated in the axial view on cone beam computed tomogram.

FIGURE 3. Panoramic radiograph illustrating another anterior radiolucency.

FIGURE 4. Coronal plane of the Stafne cavity.

202 ANTERIOR LINGUAL MANDIBULAR BONE CAVITY

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thyroid, pituitary gland, and mandible. The inclusion of salivary gland tissue in the mandible is a rare phenome- non, which is most commonly seen in the posterior region and more unusually in the anterior mandible.

According to de Courten et al the incidence in the anterior part of the mandible is approximately 0.009% to 0.03%.²Male patients seem to be more often affected (approximately 80%), with a peak in their fifth and sixth decades.^9-11 Only 40 cases have been reported in the English-language literature and 15 in other languages to this date. Thirty-two of the reported cases are of arche- ological dried skull specimens.^2,4,11,12

The pathogenesis of these lesions is not fully un- derstood. Hypotheses put forward suggest that the defect is congenital or develops through pressure resorption. The first theory suggests that part of the salivary gland becomes entrapped during the devel- opment and ossification of the mandible.^12,13Because Stafne lesions are much more common in adults, it also seems possible that they develop later in life due to ossification of the mandible. This hypothesis is supported by the fact that histologic studies have shown disruptions of the mandible’s increment lines and the inferior border of the mandible in some cases.¹⁴ Most lesions are asymptomatic. On rare oc- casions, the lingual defect can be clinically palpated.

It has been established that such lesions are nonpro- gressive.¹²

Clinically and radiographically these entities can present diagnostic difficulties.¹¹ They are mostly asymptomatic. However, not all bone cavities will be well-demarcated impressions with a sclerotic border.

They may be more irregular in their borders and correctly suggest a neoplasm. A sublingual impression may be represented as an ovoid or round radiolucency without a sclerotic border as seen in the present cases. Because a relation to the dental roots on plain radiographs is frequent in the anterior mandible, the differential diagnosis includes a periapical granuloma or radicular cyst; equally a keratocyst, an odontogenic tumor, or an idiopathic bone cavity may be unusual but possible considerations.⁹

A Stafne cavity found coincidentally in the posterior mandible is usually easy to diagnose because the lo- calization and radiographic appearance are characteristic.¹⁵ Any doubts can be eliminated with sialo- graphic examinations of the submandibular gland.

Although sialography is helpful in doubtful posterior cases, it is impracticable and rarely effective in anterior cases due to the presence of many accessory Bartholin ducts adjoining the sublingual gland in the anterior mouth floor so that it gives little information on the submandibular gland.¹⁶ Associated risks are duct trauma, acinar rupture, and a high failure rate.

Several investigators have described the use of computed tomography or magnetic resonance imaging

to specify the diagnosis, especially in the anterior case.^3,12,17A computed tomogram offers a fast alterna- tive because it can show the bony structure, soft tissue, and extension. The advantage of magnetic resonance imaging is better visualization of the soft tissue; however, its use is limited due to the high cost.

We recommend cone-beam computed tomography—a rapidly developing radiographic 3-dimensional imaging technique—to confirm the diagnosis.

In contrast to conventional spiral computed tomography, the radiation dose ranges from 68 to 1,073␮Sv, whereas computed tomography with a similar field of view produces 860␮Sv.¹⁸The image quality of bony structures is comparable. Metal artifacts in particular are less intense. Admittedly, the volume of the examination is limited and soft tissues are not distinguish- able from each other.

If a lingual impression in the anterior mandible produces a characteristic radiographic pattern with no clinical symptoms or other pathology, it may be diagnosed by an evaluation of radiographs. Inflamma- tory dental disease should be excluded. In most cases, no histopathology should be required and no treatment is indicated if 3-dimensional radiologic screen- ing shows a Stafne cavity. Nevertheless, regular follow-ups are recommended to identify any changes.

References

1. Stafne EC: Bone cavities situated near the angle of the mandible. J Am Dent Assoc 29:1969, 1942

2. de Courten A, Kuffer R, Samson J, et al: Anterior lingual mandibular salivary gland defect (Stafne defect) presented as a residual cyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 94:460, 2002

3. Shimizu M, Osa N, Okamura K, et al: CT: Analysis of Stafne’s bone defects of the mandible. Dentomaxillofac Radiol 35:95, 2006

4. Correll RW, Jensen JL, Rhyne RR: Lingual cortical mandibular defects: A radiographic incidence study. Oral Surg Oral Med Oral Pathol 50:287, 1980

5. Richard EL, Ziskind: Aberrant salivary gland tissue in the mandible. Oral Surg Oral Med Oral Pathol 10:1086, 1957 6. Dorman M, Pierse D: Ectopic salivary gland tissue in the ante-

rior mandible: A case report. Br Dent J 193:571, 2002 7. Apruzzese D, Longoni S: Stafne cyst in an anterior location.

J Oral Maxillofac Surg 57:333, 1999

8. Queiroz LM, Rocha RS, de Medeiros KB, et al: Anterior bilateral presentation of Stafne defect: An unusual case report. J Oral Maxillofac Surg 62:613, 2004

9. Belmonte-Caro R, Velez-Gutierrez MJ, Garcia De La Vega-Sosa FJ, et al: A Stafne’s cavity with unusual location in the mandibular anterior area. Med Oral Patol Oral Cir Bucal 10:173, 2005 10. Langlais RP, Cottone J, Kasle MJ: Anterior and posterior lingual

depressions of the mandible. J Oral Surg 34:502, 1976 11. Philipsen HP, Takata T, Reichart PA, et al: Lingual and buccal

mandibular bone depressions: A review based on 583 cases from a worldwide literature survey, including 69 new cases from Japan. Dentomaxillofac Radiol 31:281, 2002

12. Smith MH, Brooks SL, Eldevik OP, et al: Anterior mandibular lingual salivary gland defect: A report of a case diagnosed with cone-beam computed tomography and magnetic resonance imaging. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 103:71, 2007

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13. Choukas NC, Toto PD: Etiology of static bone defects of the mandible. J Oral Surg Anesth Hosp Dent Serv 18:16, 1960 14. Sandy JR, Williams DM: Anterior salivary gland inclusion in the

mandible: Pathological entity or anatomical variant? Br J Oral Surg 19:223, 1981

15. Kursoglu P, Ari N, Calikkocaoglu S: Use of Stafne’s mandibular defect in improving retention of mandibular complete den- tures. N Y State Dent J 73:52, 2007

16. Barak S, Katz J, Mintz S: Anterior lingual mandibular salivary gland defect—a dilemma in diagnosis. Br J Oral Maxillofac Surg 31:318, 1993

17. Katz J, Chaushu G, Rotstein I: Stafne’s bone cavity in the anterior mandible: A possible diagnostic challenge. J Endod 27:304, 2001 18. Ludlow JB, Ivanovic M: Comparative dosimetry of dental CBCT

devices and 64-slice CT for oral and maxillofacial radiology. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 106:106, 2008

J Oral Maxillofac Surg 68:204-207, 2010

Glomangioma: A Case Presentation

Per Dérand III, DDS,* Gunnar Warfvinge, DDS, PhD,† and Andreas Thor, DDS, PhD‡

The glomus body is an apparatus between the arterial and venous system located in the skin. It is encapsu- lated and works as a shunt between the arterioles and the venous blood. This is called the Sucquet-Hoyer canal.¹Its purpose is to regulate the temperature in the skin. Occasionally, the glomus apparatus develops neoplasms. The glomus tumor is an entity that often appears as a slow growing, small, blue-red nodule that, clinically, might be reminiscent of a hemangioma. They are often approximately 5 mm in diameter, painful, and, in the fingertips, associated with a triad of symptoms: hypersensitivity to cold, paroxysmal pain, and pinpoint pain.¹ In the oral cavity, these symptoms seem to be more or less absent. Most glomus tumors are solitary but can also present as multifocal familial lesions. The glomus tumor is a rare neoplasm that is mostly found in the distal part of the extremities.

To our knowledge, only 21 cases in the oral cavity have been reported.^2-22No more than 20 cases of malignancy in the whole body have been reported,²³ with only 1 oral case representing a metastasis.¹⁰

Typical glomus tumors have varying proportions of glomus cells, vascular structures, and smooth muscle tissue, according to which they are grouped as solid

glomus tumors, glomangiomas, and glomangiomyo- mas.^23,24The presented case, to date, occurred in one of the youngest patients and is one of only 4 glomangiomas convincingly presented in published studies.

Report of a Case

An 11-year-old girl was referred to the Department of Oral and Maxillofacial Surgery at the Central Hospital, Eskilstuna, Sweden, in October 2000. The patient’s parents had noted a well-defined, painless discoloration of the lower lip mea- suring 3 mm in diameter (Fig 1). The lesion was soft on palpation. The patient denied any other similar lesions throughout the body and was otherwise in good health.

There was no record of heredity. Clinically, the lesion was reminiscent of a hemangioma. The lesion was removed with the patient under local anesthesia (Xylocaine with adrena- line 20 mg/mL, Astra, Södertälje, Sweden) with minimal margin, and the wound was sutured. The postoperative period was uneventful, and at 7 years postoperatively, no signs of recurrence had been seen (Fig 2).

Histopathologic Findings

The specimen was fixed in 10% formalin and sec- tioned in 3-␮m sections that were stained with hema- toxylin and eosin. The sections were also immuno- stained with antibodies to epithelial membrane antigen, pan-cytokeratin, vimentin, smooth muscle actin, and S-100.

The sections showed oral mucosa with several di- lated vascular structures in the lamina propria and extending into the submucosa. The venous vessels were lined by a nondescript endothelium surrounded by layers of glomus cells (Figs 3,4). The glomus cells were roundish with lightly stained cytoplasm and cytologically uniform, oval nuclei (Fig 5). Mitoses were virtually absent. Immunostaining showed a dis- tinct expression of smooth muscle actin in the tumor

*Consultant, Department of Oral and Maxillofacial Surgery, Spe- cialist Tandvården, Mälarsjukhuset, Eskilstuna, Sweden.

†Associate Professor, Department of Oral Pathology, Malmö Uni- versity, Malmö, Sweden.

‡Assistant Professor, Department of Surgical Sciences, Oral and Maxillofacial Surgery, Uppsala University, Uppsala, Sweden.

Address correspondence and reprint requests to Dr Dérand:

Department of Oral and Maxillofacial Surgery, Mälarsjukhuset, Es- kilstuna S-631 88 Sweden; e-mail:per.derand@dll.se

doi:10.1016/j.joms.2009.07.023

204 GLOMANGIOMA