Abstract

We report a case of dentigerous cyst of the maxilla and maxillary sinus that caused the ectopic displacement of an unerupted tooth into the orbital rim and floor. After an incisional biopsy, marsupialization of the lesion promoted its involution and stimulated osteogenesis. This in turn simplified the surgical enucleation of the specimen and removal of the unerupted tooth without the excessive loss of the bony contours of the maxilla.

Introduction

Dentigerous cysts surrounding impacted teeth often displace these teeth into ectopic positions. In the mandible, they have been reportedly found in the symphysis, body, angle, coronoid process, and the condylar neck. In the maxilla, these teeth are often displaced into the maxillary sinus. (1-3) They have been reported to be locked into the ostiomeatal complex, (4) piriform wall, and occasionally the orbit itself. (5,6) The sequelae of these cysts and ectopic teeth vary from obstruction of the sinus to blindness. (7)

Dentigerous cysts of the maxillary sinus, and the impacted tooth within, are often easily removed via a Caldwell-Luc procedure. However, removal of large lesions may require extensive surgery, the complications of which can include aesthetic and functional deficits.

Marsupialization is a method of exteriorizing cystic lesions to facilitate decompression and involution, thereby simplifying their removal or in some cases even allowing for the complete resolution of the cyst without the need for secondary surgery. Marsupialization has been reported as a definitive treatment for extensive cysts, including the more aggressive and highly recurrent odontogenic keratocyst. (8) When a secondary surgery is required for the definitive treatment of a lesion, previous marsupialization often affords the luxury of a less invasive surgery, thereby reducing surgical complications such as oroantral fistula, oronasal fistula, nerve injuries (particularly to the inferior alveolar and lingual nerves), excessive bleeding, and mandibular fracture. It also minimizes the surgical defects caused by extensive bone removal, and it provides access for a biopsy specimen for a definitive diagnosis.

In this article, we report a case of dentigerous cyst of the maxilla and maxillary sinus that caused the ectopic displacement of an unerupted tooth into the orbital rim and floor.

Case report

A 57-year-old black woman was referred to us by her dentist on Nov. 25, 2003, for evaluation of an enlarged soft swelling of her right maxilla and face. At the time of her presentation, she was healthy, well-nourished, and in no acute distress. Head and neck examination revealed that her extraocular muscles were intact. Her pupils were equal, round, and reactive to light accommodation, and there was no evidence of diplopia. Her nares were patent bilaterally, and her septum was at midline. An intraoral examination detected an expansile swelling of her right posterior maxilla (figure 1). The swelling was fluctuant to bimanual palpation, indicating buccal and palatal cortex destruction. The patient had no other symptoms, and the results of routine laboratory tests were within normal limits.

[FIGURE 1 OMITTED]

A panoramic radiograph revealed that a large unilocular radiolucency (~10 x 12 cm) had encompassed the right maxilla from the first premolar posteriorly to the tuberosity (figure 2, A). A tooth was visible at the superior aspect of the lesion. According to the radiology report, computed tomography (CT) of the maxilla identified "a large lesion of the right maxilla consistent with a dentigerous cyst" (figure 2, B). Three-dimensional reformatted CT showed the extent of destruction and ectopic displacement of the tooth into the right infraorbital rim and orbital floor (figure 2, C).

The patient was taken to the operating room on Dec. 16, 2003. A 2 x 2-cm opening into the right maxillary antrum was created, and care was taken to not violate the cyst wall (figure 3, A). Aspiration of the cystic contents yielded approximately 20 ml of a brownish fluid. Bimanual palpation and visual inspection suggested a high likelihood of a gross deformity of the maxilla if the lesion were to be enucleated primarily. Therefore, the decision was made to marsupialize the lesion.

Through the antral opening, several biopsy specimens of the cyst wall were obtained. Through the lesion itself, the crown of an impacted tooth, locked solidly into the orbital rim and floor, was palpated. The edges of the cyst opening were sutured outwardly to the buccal window. Following irrigation, the entire lesion was packed with 1/2-inch gauze impregnated with bismuth subnitrate, iodoform, and petrolatum paste.

According to the histology report, the biopsy specimens showed "a stroma of delicate bundles of immature collagen fibers interspersed by active fibrocytes and numerous dilated capillaries. Numerous cholesterol crystals with associated giant cells were noted. No malignant features were noted. The specimen appeared to be consistent with a denuded cyst wall, but no lining epithelium was observed." Complete excision of the entire lesion with follow-up examinations was recommended.

[FIGURE 2 OMITTED]

[FIGURE 3 OMITTED]

The patient underwent weekly changes of packing that were eventually replaced by daily irrigations of the lesion until March 23, 2004, at which time the fistula was closing. Repeat CT demonstrated bone deposition in the maxilla, as well as a slight migration of the tooth away from the orbital floor. Bimanual palpation of the maxilla revealed improving firmness along the right posterior palate.

The patient was then returned to the operating room, and the lesion was completely enucleated via a Caldwell-Luc incision. The impacted tooth, with somewhat divergent roots, was carefully removed from the orbital rim and floor with judicious bone removal. The infraorbital nerve was visualized and appeared to be intact. The Caldwell-Luc incision was closed, and attention was then directed to the residual oroantral fistula, which was excised and closed primarily.

The results of surgical pathology were consistent with the biopsy findings obtained at the time of marsupialization. According to the report, "stratified squamous epithelium, foci of lipogranuloma, and extensive hyalinization of fibrous connective tissue" were observed, confirming the diagnosis of a dentigerous cyst.

The patient healed uneventfully, and no oroantral communication was observed (figure 3, B). No complications were encountered.

Discussion

In this case, marsupialization was successfully used to minimize the amount of maxillary destruction and surgical morbidity that might have resulted from the immediate enucleation of the lesion. Although it would have been of interest to allow more time for further involution and to assess whether the tooth would migrate into the oral cavity without a secondary surgery, it can be quite problematic to maintain the patency of these sites intraorally. In this case, marsupialization allowed us to obtain several biopsy specimens for treatment planning and provided some time for osteogenesis, particularly of the palatal aspect of this extensive lesion.

References

(1.) Mody RN, Sathawane RS, Samdani D. Dentigerous cyst: Report of an unusual case. Dent Update 1995;22(3):124-6.

(2.) Gunbay MU, Lomcali G, Ozaksoy D, et al. Ectopic teeth in the maxillary sinus: Diagnosis and treatment. Dent Update 1995;22 (4):146-8.

(3.) Frer AA, Friedman AL, Jarrett WJ. Dentigerous cysts involving the maxillary sinus. Oral Surg Oral Med Oral Pathol 1972;34(3): 378-80.

(4.) Hasbini AS, Hadi U, Ghafari J. Endoscopic removal of an ectopic third molar obstructing the osteomeatal complex. Ear Nose Throat J 2001;80(9):667-70.

(5.) Golden AL, Foote J, Lally E, et al. Dentigerous cyst of the maxillary sinus causing elevation of the orbital floor. Report of a case. Oral Surg Oral Med Oral Pathol 1981;52(2):133-6.

(6.) Ferber EW. Ectopic supernumerary tooth, imbedded in superior wall of left maxillary antrum. J Calif Dent Assoc 1972;48(1):28-9.

(7.) Savundranayagam A. A migratory third molar erupting into the lower border of orbit causing blindness in the left eye. Aust Dent J 1972;17(6):418-20.

(8.) Pogrel MA, Jordan RC.Marsupialization as a definitive treatment for the odontogenic keratocyst. J Oral Maxillofac Surg 2004;62(6):651-5; discussion 655-6.

Morton Litvin, DDS; Domenic Caprice, DMD; Leonard Infranco, DMD

From the Department of Oral and Maxillofacial Surgery, University of Pennsylvania School of Dental Medicine, Philadelphia (Dr. Litvin), and an oral and maxillofacial surgery private practice, Vineland, N.J. (Dr. Caprice and Dr. Infranco).

Corresponding author: Morton Litvin, DDS, Clinical Professor of Oral Surgery and Pharmacology, Department of Oral and Maxillofacial Surgery, University of Pennsylvania School of Dental Medicine, 240 S. 405h St., Philadelphia, Pa 19104. Phone: (856)692-0399; fax: (956) 692-4845; e-mail: L2docs@comcast.net