Multiple Dentigerous Cysts

Abstract

Dentigerous cysts are one of the common cysts of the jaws and are associated with the crowns of permanent teeth, most frequently with impacted mandibular third molars. Bilateral dentigerous cysts generally occur in association with a developmental syndrome or systemic diseases. Bilateral dentigerous cysts in absence of a syndrome is a rare condition. There are only 18 cases reported in the literature till date (Dinkar et al. J Indian Soc Pedod Prev Dent 25:56–59, 2007). There are no cases reported with multiple dentigerous cyst involving all the four quadrants.

Introduction

A dentigerous cyst is an epithelial—lined developmental cavity that encloses the crown of an unerupted tooth at the cementoenamel junction [1]. It was reported in 1847, and was known as distended capsule, osseous cyst or serous cyst. These cysts are the second most common odontogenic cysts after radicular cysts [1–4]. They account for approximately 24% of all true cysts in the jaws [1].

Dentigerous cyst develops by accumulation of fluid between reduced enamel epithelium and enamel or within the enamel organ. Pressure exerted by an erupting tooth on an impacted follicle obstructs the venous outflow. This leads to rapid transudation of serum across the capillary walls. Increase in the hydrostatic pressure of the pooling fluid occurs, leading to separation of follicle from the crown with or without reduced enamel epithelium. An intrafollicular spread of periapical inflammation from a deciduous tooth may also result in the development of dentigerous cyst [2].

As noted in Table 1, there have been only 18 cases of multiple non-syndromic cysts reported in literature from 1943 to 2006 [5]. An unusual case of multiple dentigerous cysts is described here.

Table 1.

Review of literature regarding bilateral multiple dentigerous cysts [3]

Authors/year	Sex	Age (in years)	Location	Treatment
Myers, 1943	F	19	Md. third molars	Enucleation
Henefer, 1964	F	52	Mx. third molars	Enucleation
Stanback, 1970	M	09	Md. first molars	Enucleation
Callghan, 1973	M	38	Md. third molars	Enucleation
Burton Scheffer, 1980	F	57	Md. third molars	Enucleation
Swerdloff, Alexander, Ceen Ferguson, 1980	F	07	Md. first molars	Enucleation
Crinzi, 1982	F	15	Md. third molars	Enucleation
Mc Donnel, 1988	M	15	Md. second premolar and second molar	Enucleation
Eidinger, 1989	M	15	Md. first molars	Enucleation
O’ Neil, Mosby, Lowe, 1989	M	05	Md. first molars	Enucleation
Banderas, Gonzalez, Ramirez, Arroyo, 1996	M	38	Md. third molars	Enucleation
Sands, Tocchio, 1998	F	03	Md. central incisors and first molars	Enucleation
Ko, Dover, Jordan, 1999	M	42	Md. third molars	Enucleation
De Biase, Ottolenghi, Polimeni, Benvenuto, Lubrano, Magliocca, 2001	M	08	Md. first molars	Enucleation
Shah, Thuau, Beale, 2002	M	39	Md. third molars	No treatment
Ustuner, Fitoz, Atasoy, Erden, Akyar, 2003	M	06	Mx. canines	Enucleation
Batra, Roychoudhury, Balakrishan, Parkash, 2004	F	15	Md. third molars and second premolar	Enucleation
Frietas, Tempest, 2006	M	14	Mx. third molars and Md. second molar	Enucleation

M male, F female, Mx maxillary, Md mandibular

Case Report

A 17 year old male patient reported to the department of Oral Medicine and Radiology with a chief complaint of unpleasing appearance of upper front teeth. He gives history of retained deciduous teeth in upper front teeth region. There was no relevant past dental, medical or family history and there was no associated syndromes or systemic diseases present.

Intraoral examination revealed that his maxillary central and lateral incisors, maxillary and mandibular canines and mandibular premolars were unerupted (Fig. 1). There were retained primary maxillary central incisors, lateral incisors and right canine and also mandibular canines, first and second molars on the right side and second molar on left side. There was a diffuse swelling involving the attached gingiva and vestibular mucosa, present in relation to the distal aspect of right lower lateral incisor till the mesial aspect of right lower deciduous first molar, measuring approximately 2 × 1 cm. The swelling was soft to firm in consistency and there was expansion of the buccal cortical bone in relation to right lower deciduous canine and second molar.

Fig. 1.

Intraoral photograph showing retained upper anterior deciduous teeth

Patient was advised for intraoral periapical radiographs in relation to 51, 52, 61, 62, 73, 83, 84 and panoramic radiograph, which revealed a well defined unilocular radiolucency with sclerotic borders associated with the crowns of right maxillary central and lateral incisors, measuring approximately 2 × 2 cm. In the second quadrant, radiolucency was associated with left maxillary central, lateral incisors and canine, measuring 3 × 2 cm. Bilateral heart-shaped radiolucencies were present in the third and fourth quadrants associated with canine and second premolar in the third quadrant and first and second premolars in fourth quadrant measuring 3 × 2 and 4 × 2 cm, respectively. There was radicular resorption of the retained deciduous maxillary and mandibular teeth (Fig. 2). Computed Tomography revealed, crowding and multiple unerupted teeth and 1.5 × 1 cm sized, cyst in right and left halves of maxilla and 2 × 1 cm sized cysts in right and left halves of mandible. CT imaging also showed the integrity and expansion of the buccal cortex (Figs. 3, 4, 5). The clinical and radiographic findings were suggestive of dentigerous cysts. Under general anaesthesia the dentigerous cysts in maxilla and mandible were enucleated and the unerupted permanent teeth were removed along with the cystic lining and the specimen was sent for histopathological evaluation. Microscopic examination of all the sections revealed that odontogenic epithelium surrounding the cystic cavity composed of flattened cells about three to five cells thickness. Connective tissue stroma showed interlacing collagen fibres, fibroblasts and vascularity. Severe inflammatory cell infiltration and cholesterol clefts were seen in the stroma, suggestive of infected dentigerous cysts (Figs. 6, 7).

Fig. 2.

Panoramic radiograph is showing bilateral radiolucencies in maxilla and mandible

Fig. 3.

Coronal CT image shows multiple unerupted teeth and radiolucencies in maxilla

Fig. 4.

3D volume of mandible showing multiple unerupted teeth

Fig. 5.

Axial CT image shows the buccal and lingual cortical plates of the mandible to be intact

Fig. 6.

Photomicrograph of the lesion showing that the odontogenic epithelium composed of flattened cells. (haematoxylin and eosin, magnification ×40)

Fig. 7.

Connective tissue stroma is showing severe chronic inflammatory cells and cholesterol clefts. (haematoxylin and eosin, magnification ×40)

Discussion

Dentigerous cysts are benign odontogenic cysts associated with the crowns of permanent teeth. It may involve impacted, unerupted permanent teeth, supernumerary teeth, odontomas and rarely deciduous teeth [4]. They are usually present in the second or third decades of life and are rarely seen during childhood. The mandibular third molar and maxillary canines are involved most frequently [4, 5].

The review of literature (Table 1) reveals the involvement of mandibular third molars in 8 cases and involvement of mandibular second premolar in 2 cases [6, 7]. Only one case has been reported to occur in association with lower central incisor [8]. In the present case, there is rare involvement of upper right and left central and lateral incisors and lower right first premolar tooth. The age range for reported cases varies widely, from 3 to 57 years [5]. In all reported cases, including the present case, radiographic examination showed a unilocular radiolucent lesion associated with the crown of an unerupted tooth and well defined sclerotic margins.

The earliest case of multiple dentigerous cysts was recorded by Glaswald in 1844 [9]. Bilateral and multiple cysts have been reported in Basal cell nevus syndrome, Mucopolysaccharidosis, Cleidocranial dysplasia and after prolonged concurrent use of cyclosporine A and calcium channel blockers [4].

Dentigerous cyst may cause displacement of adjacent teeth and resorption of roots [10]. In the present case resorption of roots of the deciduous maxillary and mandibular teeth was present. It may displace or obliterate the maxillary sinus and nasal cavity, and cause paraesthesia of inferior alveolar nerve. The cyst’s lining may contain areas of ortho-keratinization, ciliated cells, or mucin secreting cells. Because of this inherent ability for metaplastic change, some dentigerous cyst appear to progress to more aggressive lesions such as an odontogenic keratocyst, ameloblastoma, mucoepidermoid carcinoma or squamous cell carcinoma [2]. It is important to perform radiographic examinations in case of unerupted teeth. CT imaging gives information about origin, size, content, expansion of cortical plates and relationship of the lesion to adjacent anatomical structures [5]. Enucleation was the line of treatment in 17 of the 18 reported cases, although larger lesions may be surgically drained and marsupialized to relieve the pressure within the cysts and to prevent damage to the involved teeth.

This case report emphasizes the importance of radiographic examination of all unerupted teeth, using panoramic radiography supplemented with computer tomography for a better delineation of the extent of the lesion and its relationship to adjacent anatomical structures.