Bilateral Dentigerous Cysts - Report of an Unusual Case and Review of the Literature 

K. S.C. Ko, DDS
D.G. Dover, DDS, MRCD(C)
R.C.K. Jordan, DDS, M.Sc., PhD, FRCD(C)

ABSTRACT
Dentigerous cysts are the most common developmental cysts of the jaws, most frequently associated with impacted mandibular third molar teeth. Bilateral dentigerous cysts are rare and occur typically in association with a developmental syndrome. The reported occurrence of bilateral dentigerous cysts in the absence of a syndrome is rare and, to date, only 11 cases have been described. Here, we report a case of bilateral nonsyndromic, dentigerous cysts and review the literature for this unusual finding.

MeSH Key Words:  
case report; dentigerous cyst/pathology; mandibular diseases/pathology.
© J Can Dent Assoc 1999; 65:49-51 
This article has been peer reviewed. 

 [ Top ]

[Introduction | Case Report | Radiographic Findings | Treatment | Pathology | Discussion | Reference] 

Introduction 

[ Top ]

N/A= Not available                 M = male
C = Caucasian                         F = female
Bl = Black                               Md. = mandibular

 [ Top ]

Case Report

Radiographic Findings

Fig.1   Preoperative panoramic radiograph showing bilbateral well-defined radiolucencies involving both un-erupted mandibular third molar teeth.	Fig.2  Photomicrograph showing cyst wall composed of fibrous tissue and lined by nonkeratinised stratified sqaumour epithelium.(H& E X 40)

[ Top ]

Treatment

The clinical diagnosis was bilateral dentigerous cysts. Under general anesthesia, the buccal flaps were raised and the cysts enucleated together with the associated third molar teeth. Healing was uneventful, and one week after the operation, the surgical sites showed good healing. There was partial paresthesia on the left side affecting the mental nerve distribution of the inferior alveolar nerve, but after three months, there had been complete healing. There was also no evidence of recurrence of the cysts.

Pathology

[ Top ]

Discussion

Although dentigerous cysts are common developmental cysts, reported bilateral dentigerous cysts are extremely rare. Bilateral or multiple dentigerous cysts are usually associated with the Maroteaux-Lamy (mucopolysaccharidosis, type VI) syndrome¹⁶ and cleidocranial dysplasia.¹⁷ Both are developmental conditions that are detected in young individuals with stigmata of the syndromes.

Maroteaux-Lamy syndrome is one of the mucopolysaccharidoses (MPS), a group of diseases resulting from a genetic defect in the degradation of specific mucopolysaccharides. With this syndrome, there is a deficiency of N-acetyl-4-sulphatase that results in impaired degradation of dermatan sulphate, which accumulates in tissues and is excreted in the urine. Dental features include unerupted dentition, dentigerous cysts, malocclusions, condylar defects, and gingival hyperplasia.¹⁶ Cleidocranial dysplasia is an autosomal dominantly inherited disorder that results in a partial or complete absence of clavicles, short stature, frontal and parietal bossing, maxillary micrognathia, prolonged retention of the primary dentition, delayed eruption of the permanent dentition, and unerupted supernumerary teeth.¹⁷ Multiple dentigerous cyst formation occurs in both conditions and can develop at any site in the upper or lower jaws.

Bilateral dentigerous cysts are rare in the absence of an underlying syndrome or systemic disease. An extensive search of the English language literature has identified only the 11 cases reported in Table I. Although this finding may reflect the true rarity of the condition, it is conceivable that bilateral dentigerous cysts are either under-recognized or under-reported.

The age range for the reported cases varies widely, from 5 to 57 years of age. Four of the cysts occurred in children under the age of 12. All but three cases^6,12,13 were identified at ages corresponding to the normal eruption times of the affected teeth. As with our case, these three cases occurred in asymptomatic individuals, which accounts for the delayed diagnosis. All but two of the 11 cases^5,9 have been associated with mandibular molar teeth, with five of these associated with the third molar teeth.^6,10-13 There have been no reported cases of nonsyndromic, bilateral dentigerous cysts occurring in all four dental quadrants.

Since cysts can attain considerable size with minimal or no symptoms, early detection and removal of the cysts is important to reduce morbidity. Moreover, all but one¹¹ of the reported cases, including ours, presented without pain. And, all but one were discovered during investigation of asymptomatic slow-growing swellings.¹¹

It is therefore important to perform radiographic examination of all unerupted teeth. While bite-wing and periapical radiography is typically performed in the routine examination of patients with a healthy dentition, this series of radiographs may occasionally fail to delineate the full extent of a lesion if present. A panoramic radiograph supplemented with skull series or more advanced imaging such as tomography may permit a better delineation of the extent of the lesion and its relationship to adjacent anatomical structures.

Dr. Ko was a dental resident, department of dentistry, Sunnybrook Health Science Centre, Toronto, Ont.

Dr. Dover is an oral and maxillofacial surgeon, department of dentistry, Sunnybrook Health Science Centre, Toronto, Ont.

Dr. Jordan is an oral pathologist, department of dentistry, Sunnybrook Health Science Centre, and in the department of laboratory medicine, Sunnybrook Health Science Centre, Toronto, Ont.

Reprint requests to: Dr. Richard Jordan, H-126, Sunnybrook Health Sciences Centre, 2075 Bayview Ave., Toronto ON M4N 3M5.

 [ Top ]

References

 [ Top ]