Bilateral maxillary dentigerous cysts: A case report

Bilateral maxillary dentigerous cysts: A case report Evren Ustuner, MD,a Suat Fitoz, MD,a Cetin Atasoy, MD,a Ilhan Erden, MD,b and Serdar Akyar, MD,b Ankara, Turkey ANKARA UNIVERSITY MEDICAL SCHOOL

Dentigerous cysts are benign odontogenic cysts that are associated with the crowns of permanent teeth. They are usually single in occurrence and located in the mandible. Multiple cysts are reported in patients with conditions such as mucopolysaccharidosis and basal cell nevus syndrome. We present the radiologic findings of bilateral impacted maxillary cuspids with dentigerous cysts displacing the maxillary sinuses in a nonsyndromic patient, a condition that, to our knowledge, has not been previously reported. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:632-5)

Dentigerous cysts are the second most common odontogenic cysts after radicular cysts.1 They usually present in the second or third decades of life and are rarely seen during childhood.2 Dentigerous cysts are usually solitary with multiple cysts reported on occasion in association with syndromes such as mucopolysaccharidosis and basal cell nevus syndrome.3-5 We present the radiologic findings of bilateral maxillary dentigerous cysts involving the cuspid teeth in a nonsyndromic 6-year-old boy. A search of MEDLINE from 2002 to 1967, using the key words dentigerous cyst, bilateral and multiple, was conducted and revealed no examples of bilateral maxillary nonsyndromic dentigerous cysts. To our knowledge, bilateral maxillary dentigerous cysts in a nonsyndromic patient have not been reported previously in the literature. CASE REPORT A six-year-old boy presented with left facial swelling. Except for his facial asymmetry, his physical examination was unremarkable, and routine laboratory tests were within normal limits. Waters view of the paranasal sinuses revealed diffuse opacification of both maxillary sinuses associated with well-defined radiopacities in the superiomedial aspects and prominent displacement of the left maxillary border. The radiopacities were consistent with unerupted teeth (Fig 1). Computed tomography (CT) showed well-defined soft tissue masses displacing and obliterating both maxillary sinuses especially on the left maxillary sinus (Fig 2A). The lesions had thin walls and contained mural teeth structures (Fig 2B). On magnetic resonance imaging (MRI), the lesions were hypointense on T1W (Fig 3A) and hyperintense on T2W (Fig a

Resident, Department of Radiology, Medical School, Ankara University. b Professor of Radiology, Department of Radiology, Medical School, Ankara University. Received for publication Jun 27, 2002; returned for revision Sep 12, 2002; accepted for publication Nov 8, 2002. © 2003, Mosby, Inc. All rights reserved. 1079-2104/2003/$30.00 ⫹ 0 doi:10.1067/moe.2003.123

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Fig 1. Waters radiography of the maxillary sinuses shows bilateral maxillary sinus opacification and impacted teeth in the superiomedial aspects of the sinus walls.

3B) images with the associated teeth hypointense on all sequences. There were no solid components in the lesions on MRI other than the teeth. Examination of the surgical specimen obtained from the lesion on the left revealed a cystic lesion lined by nonkeratinized squamous epithelium associated with the impacted crown of the cuspid. No dysplastic changes were observed (Fig 4).

DISCUSSION Dentigerous cysts are benign odontogenic cysts associated with the crowns of permanent teeth.6 Cysts involve impacted, unerupted permanent teeth, supernumerary teeth, odontomas, and, rarely, decidious teeth.2,6 In 75% of the cases, they are located in the mandible.1 The mandibular third molar and maxillary canine are involved most frequently. Dentigerous cysts are usually painless but may cause facial swelling and delayed

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Fig 2. Coronal soft tissue CT image (A) shows well-defined lesions with soft tissue attenuation containing impacted teeth superiomedially. Both maxillae are expanded laterally. The expansion is more marked in the left maxilla with displacement of the left maxillary sinus wall medially. Bone window CT image (B) reveals the impacted teeth and the maxillary expansion more clearly.

tooth eruption.4,6,7 The usual presentation is in second or third decade of life. Extensive maxillary involvement and childhood presentation are rare.2,7 Dentigerous cysts are usually single lesions. Bilateral and multiple cysts have been reported in patients with syndromes such as basal cell nevus syndrome, mucopolysaccharidosis, and cleidocranial dysplasia.3,4,5,8 Bilateral mandibular dentigerous cysts have also been reported after prolonged concurrent use of cyclosporine A and calcium channel blockers.9 Gingival hyperplasia

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Fig 3. T1-weighted (A) and T2-weighted (B) axial MR images show T1-hypointense and T2-hyperintense cystic lesions. The impacted teeth are hypointense on T1 and T2.

and impaired dentition are the most common features shared by most of these syndromes.4,9 Up to now, the few reported bilateral dentigerous cysts in nonsyndromic patients have all been located in the mandible.4,2,10,11,12 One case of multiple dentigerous cysts involving both the maxilla and the mandible in a nonsyndromic patient with chickenpox has been reported.3 As with this case, nearly half of the patients were under 12, and most presented with painless facial

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Fig 4. Photomicrography of the specimen shows the cystic lesion lined by nonkeratinized squamous epithelium. There is mild inflammatory cellular infiltration in the subepithelial connective tissue (hematoxylin-eosin, magnification ⫻100).

swelling.4 To our knowledge, bilateral maxillary cysts involving only the cuspids in the maxilla in a nonsyndromic patient have not been previously reported. Our patient was a healthy child with no abnormal physical or laboratory findings suggesting any syndromes. On radiographic examination, dentigerous cysts appear as unilocular lucent cysts of varying sizes, with well-defined sclerotic borders, associated with the crown of an unerupted tooth.1,6 If a follicular space on radiography is more than 5 mm, an odontogenic cyst can be suspected.4 Other odontogenic cysts like radicular cysts, odontogenic keratocysts, and odontogenic tumors such as ameloblastoma, Pindborg tumor, odontoma, odontogenic fibroma, and cemetomas may share the same radiologic features as dentigerous cysts.1,6 Microscopic evaluation is necessary most of the time to define the type of lesion.1 In extensive cases, radiographics alone may not be sufficient to show the full extent of the lesions, and advanced imaging may be needed.4 Of the lesions included in the differential diagnosis of dentigerous cysts, radicular cysts are the most common. Radicular cysts are odontogenic cysts that develop from a periapical granuloma in a carious tooth. Odontogenic keratocysts are often multilocular and most commonly located in the body or the ramus of the mandibula. Ameloblastoma is the most common radiolucent, benign odontogenic tumor that may be unilocular or multilocular. It may cause expansion and destruction of the maxilla and mandibula. Pindborg tumors are rare odontogenic tumors that are radiolucent

with well-defined borders and associated calcified radiopaque foci. Odontomas and cementomas are lytic lesions most often accompanied by amorphous calcification. Odontogenic fibromyxoma usually has multiple radiolucent areas of varying size and bony septations, but unilocular lesions have also been described.1,6 In cases of extensive bony involvement and presence of a complex cystic lesion, CT imaging becomes necessary.1,6 In the maxilla, dentigerous cysts may be destructive and may occupy the maxillary sinus; nasal cavities and even orbital encroachment may be observed.1,7 CT imaging helps to rule out solid and fibroosseous lesions, displays bony detail, and gives exact information about the size, origin, content, and relationships of the lesions involving the maxilla.13 Although mucoceles form the majority of cystic antral lesions that cause expansion of the maxilla, dentigerous cysts, odontogenic keratocysts, residual cysts, and cystic ameloblastomas are included in the differential diagnosis of extra-antral lesions. Observation of the cortical plates and antral bony walls on CT help to distinguish an antral from an extra-antral maxillary lesion.13 MRI may fail to show the bony detail but precisely displays the lesional contents and provides information about the cyst fluid.1,14 The cystic lesions appear homogeneously hypointense on T1-weighted images and hyperintense on T2-weighted images.1 Dentigerous cysts cause expansion and remodeling of sinus walls if they involve the maxillary sinuses, and the impacted tooth lies eccentricly in the cyst wall, appearing hy-

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pointense on all sequences. Within the sinus, chronic secretions, air, acute hemorrhage, and mycetoma may also appear hypointense, but their central location in the sinus distinguish them from teeth.14 Surgical excision and pathologic analysis of the lesion is essential for the definitive diagnosis.6 As with this case, maxillary cysts may displace and obliterate the maxillary antrum and nasal cavities. The cysts may cause fractures and become secondarily infected.13 Metaplastic and dysplastic changes may occur.6,15 An ameloblastoma, mucoepidermoid carcinoma, or squamous cell carcinoma may develop from the lining epithelium of a dentigerous cyst.1,2,15 Associated aneurysmal bone cysts and hemangiomas have been reported in rare instances.7 Smaller lesions are removed entirely to prevent damage to the involved permanent teeth, whereas larger lesions may be surgically drained and marsupialized to relieve the pressure within the cysts.2 Dentigerous cysts are known to recur very rarely.7 We would like to thank Dr Omer Gunhan for interpreting the pathology specimens and kindly providing the photomicrographs. REFERENCES 1. Weber AL. Imaging of the cysts and odontogenic tumors of the jaw. Definition and classification. Radiol Clin North Am 1993; 31;1:101-20. 2. O’Neil DW, Mosby EL, Lowe JW. Bilateral mandibular dentigerous cysts in a five-year-old child: report of a case. ASDC J Dent Child 1989;56:382-4. 3. Norris L, Piccoli P, Papageorge MB. Multiple dentigerous cysts of the maxilla and the mandible:report of a case. J Oral Maxillofac Surg 1987;45:694-7.

4. Ko KS, Dover DG, Jordan RC. Bilateral dentigerous cysts— report of an unusual case and review of the literature. J Can Dent Assoc 1999;65:49-51. 5. Roberts MW, Barton NW, Constantopoulos G, Butler DP, Donahue AH. Occurance of multiple dentigerous cysts in a patient with the Morateax-Lamy syndrome (mucopolysaccharidosis, type VI). Oral Surg Oral Med Oral Pathol 1984;58:169-75. 6. Miller CS, Bean LR. Pericoronal radiolucencies with and without radiopacities. Dental Clin North Am 1994;38:51-61. 7. Omnell KA, Rohlin M. Case challenge: chronic maxillary inflammation. J Contemp Dent Pract 2000;15:100-5. 8. Trimble LD, West RA, McNeill RW. Cleidocranial dysplasia. Comprehensive treatment of dentofacial abnormalities. J Am Dent Assoc 1982;05:661-6. 9. De Biase A, Ottolenghi L, Polimeni A, Benvenuto A, Lubrano R, Magliocca FM. Bilateral mandibular cysts associated with cyclosporine use: a case report. Pediatr Nephrol 2001;16:993-5. 10. Sands T, Tocchio C. Multiple dentigerous cysts in a child. Oral Health 1998;88:27-29. 11. Banderas JA, Gonzalez M, Ramirez F, Arroyo A. Bilateral mucous cell containing dentigerous cysts of the mandibular third molars: report of an usual case. Arch Med Res 1996;27:327-9. 12. Crinzi RA. Bilateral dentigerous cysts of the mandible. Oral Surg Oral Med Oral Pathol 1982;54:367. 13. Han MH, Chang KH, Lee CH, Na DG, Yeon KM, Han MC. Cystic expansile masses of the maxilla: differential diagnosis with CT and MR. AJNR 1995;16:333-8. 14. Som PM, Dillon WP, Curtin HD, Fullerton GD, Lidov M. Hypointense paranasal sinus foci: differential diagnosis with MR imaging and relation to CT findings. Radiology 1990;176:77781. 15. Yasuoka T, Yonemoto K, Kato Y, Tatematsu N. Squamous cell carcinoma arising in a dentigerous cyst. J Oral Maxillofac Surg 2000;58:900-5. Reprints requests: Evren Ustuner, MD Ilkyerlesim mah. Koklu sit. No:44 Batıkent, 06173 Ankara, Turkey [email protected]