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Case Report

Conservative management of dentigerous cyst in a child

Emeline das Neves de Araujo Lima a,*, Conceicao Aparecida Dornelas Monteiro Maia a,Alberto Costa Gurgelb, Patrcia Teixeira de Oliveira c, Ana Miryam Costa de Medeirosd

aDepartment of Oral Pathology, Federal University of Rio Grande do Norte, Natal, RN, BrazilbMaxillofacial Surgery, Potiguar University (UNP), Natal, RN, BrazilcOral Radiology, Federal University of Rio Grande do Norte, Natal, RN, Brazild Stomatology, Federal University of Rio Grande do Norte, Natal, RN, Brazil

1. Introduction

Odontogenic cysts are frequently seen in dental practice and

constitute an important group of oral and maxillofacial pathology

[1]. However, the frequency of odontogenic cysts in children is

relatively low [2]. Developmental and inflammatory odontogenic

cysts are epithelial in origin, exhibiting slow growth and a

tendency toward expansion. However, despite their benign

biological behavior, these lesions can reach a marked size if they

are not diagnosed and treated appropriately [3]. Therefore, the

correct diagnosis is essential for prompt and appropriate surgical

treatment with adequate follow-up [1].

Dentigerous cyst (DC), also known as follicular cyst, is an

odontogenic cystwith fluidaccumulationbetween the crown and

enamelorgan of an unerupted tooth [4]. Buccalbony expansion is

the most common clinical feature [5] related to epithelial

proliferation, release of boneresorbing factors and an increasein cyst fluid osmolality [6]. It is the second most common

odontogenic cyst,with a frequency ranging from11.4% to44%and

is more frequently seen in the second decade of life [1]. Few of

these cystshave been reported in children younger than 15 years

of age [710].

The exact histogenesis of DC is unclear [6] and, apart from its

developmental origin, an inflammatory origin has also been

suggested [7]. It has been reported that the progression of

inflammation in the root apex of a deciduous tooth may be

related to thedevelopment of a DC in theunerupted corresponding

permanent tooth [11]. The most commonly affected teeth are the

mandibular third molar and maxillary canine, being rarely

associated with other teeth [12]. The frequency of DC at these

sites can be explained by the fact that the lower third molars and

upper canines are the most commonly impacted teeth [1].

Radiographically, DC shows a well-defined radiolucency

surrounding the crown of an unerupted tooth, which often has a

sclerotic border. Histologically, the DC consists of a fibrous wall

lined by nonkeratinized, stratified, squamous epithelium, pre-

senting myxoid tissue, odontogenic remnants and, rarely, seba-

ceous cells [12]. Removal of the entire cyst associated with the

extraction of the impacted tooth is the main treatment to preventrecurrence. However, when there is a possibility of preservation

and eruption of those teeth, conservative management should be

the choice [13].

This paper presents a case of DC in a 10-year-old child with

emphasis on conservative surgical treatment and eruption of the

permanent teeth after 2 years of follow-up.

2. Case report

A 10-year-old boy was referred to the Department of Dentistry,

with a chief complaint of painless swelling in the left maxillary

International Journal of Pediatric Otorhinolaryngology Extra 8 (2013) e1e4

A R T I C L E I N F O

Article history:

Received 20 August 2012

Received in revised form 19 October 2012

Accepted 22 October 2012

Available online 26 November 2012

Keywords:

Dentigerous cyst

Odontogenic cysts

Primary dentition

A B S T R A C T

Dentigerous cysts are odontogenic cysts associatedwith the crownsof unerupted permanent teeth. This

article reports the case of a 10-year-old childwho presented permanence of deciduousteeth51, 52, and

53. Panoramic radiograph showed extensive cystic lesion involvingthe teeth 11, 12 and 13, whichwere

included. Diagnosis of dentigerous cysts was confirmed by incisional biopsy and the treatment was

chosen in order to preserve the associated permanent teeth.At 2 years of follow-up, the impacted teeth

are positioning itself spontaneously toward its eruption and the patient was referred for orthodontic

assessment.

2012 Elsevier Ireland Ltd. All rights reserved.

* Correspondingauthorat: FederalUniversityofRioGrandedo Norte, Av.Senador

Salgado Filho, 1787 Lagoa Nova, Natal, Rio Grande do Norte, CEP: 59056-000,

Brazil. Tel.: +55 84 3215 4138; fax: +55 84 3215 4138.

E-mail address: emelinelima@hotmail.com (E.d.N.d.A. Lima).

Contents lists available at SciVerse ScienceDirect

International Journal of Pediatric OtorhinolaryngologyExtra

journal homepage: www.elsevier . com/locat e/ i jp or l

1871-4048/$ see front matter 2012 Elsevier Ireland Ltd. All rights reserved.

http://dx.doi.org/10.1016/j.pedex.2012.10.005

http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005mailto:emelinelima@hotmail.commailto:emelinelima@hotmail.comhttp://www.sciencedirect.com/science/journal/18714048http://www.sciencedirect.com/science/journal/18714048http://www.sciencedirect.com/science/journal/18714048http://dx.doi.org/10.1016/j.pedex.2012.10.005http://dx.doi.org/10.1016/j.pedex.2012.10.005http://www.sciencedirect.com/science/journal/18714048mailto:emelinelima@hotmail.comhttp://dx.doi.org/10.1016/j.pedex.2012.10.005

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region for about two weeks. No systemic condition was reported.

Extraoral examination revealed asymmetric face with a diffuse

swelling and effacement of the nasolabial fold. Intraorally, the

deciduous teeth51,52 and53 showedwear facets compatiblewith

bruxism. The right central incisor was discolored with evidence of

pulp disturbance. Panoramic and occlusal radiographs showed a

large, well-defined radiolucency surrounding the retained perma-

nent teeth 11, 12 and 13. Based on clinical and radiographic

findings, a provisional diagnosis of DC has been suggested.

However, adenomatoid odontogenic tumor and odontogenic

keratocyst were also considered in the differential diagnosis.

The treatment was surgical enucleation of the lesion and removal

of deciduous teeth under local anesthesia. The specimen was sent

for histopathologic examination and the diagnosis of DC was

confirmed. After 2 years of follow-up, the impacted teeth arepositioning itself spontaneously toward its eruption and the

patient was referred for orthodontic assessment (Figs. 15).

3. Discussion

The distribution and characteristics ofjaw cysts in children are

different from those in adults. In children there is a relatively high

rate of developmental cysts, whereas in adults the inflammatory

cysts are more common [14]. The DC is the second most common

Fig. 1. Extra and intraoral examination showing volume increase in the right face and permanence of deciduous teeth 51, 52, and 53.

Fig. 2. Panoramic and occlusal radiographs showing a cystic lesion associated with

deciduous

teeth

51,

52

and

53.Fig. 3. Exposure of permanent teeth during the surgery.

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odontogenic cyst in adults and the most common in children [15].

The study of Selvamani et al. [1] determined the prevalence of

odontogenic cysts in a South Indian sample population and found

that the DC was the second more frequent cyst, after the periapical

or radicular cyst. The majority of cases were reported in the second

decade of life and they were more commonly seen in the anterior

maxilla

of

males

associated

with

erupted,

developing,

or

impactedtooth. However,Jones etal. [16] reportedapeak incidence between

the fifth and sixth decades.

The difference in prevalence of developmental cysts is

probably related to the fact that during the pediatric age period,

thejaws areinvolved inprofounddevelopmentalprocesses.These

include growth of the maxillofacial skeleton and development of

the primary and permanent dentition, all of which can be

associated with cyst formation [15]. Regarding the location,

DCs in the anterior maxilla have been reported [17,18], however,

in most of cases, the recommended treatment is usually radical

surgery, with removal of the teeth involved [14]. The case

reported is consistent with findings in the literature, but differs

with respect to the number of teeth involved and the success of a

conservative

treatment.

Swelling canbe experienced if theDC grows too large or lies in a

sensitivearea.When the cyst occupies thewholemaxillary sinus or

is infected, other clinical symptoms such as chronic sinusitis

usually occur late in the process [6]. Some authors suggest that DC

can rarely arise as a result of periapical inflammation from any

source but usually from a nonvital deciduous tooth and spreading

to

involve

the

follicles

of

the

unerupted

permanent

successors.

Theinflammatory exudate causes separation of the reduced enamel

epithelium from the enamel with resultant cyst formation. So, they

propose the existence of two types of DC: one developmental and

the other inflammatory in nature [11,19,20]. In this case, the

infection in a primary tooth pulp may have originated the

inflammatory process, producing epithelial cell proliferation and

cystic formation.

In our patient, the panoramic radiograph revealed an

extensive cystic lesion involving the teeth 11, 12 and 13,

suggesting the clinical diagnosis of DC, however the method for

definitive diagnosis of a DC is surgical exploration and

pathologic examination, as was done in the case reported. The

treatment objective for odontogenic cysts is restoring the

morphology and

function

of

the

affected area. There are two

Fig. 4. Photomicrographs showing a cystic lesion lined bynonkeratinised, stratified, squamous epithelium. Note the intense inflammatory infiltrate in the capsule (H & E 40X,

100X).

Fig. 5. Eruption of teeth 11 and 12 two years after surgery. Computed tomography in axial and sagittal planes shows the positioning of impacted teeth.

E.N.A. Lima et al./ International Journal of Pediatric Otorhinolaryngology Extra 8 (2013) e1e4 e3

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basic surgical procedures, namelymarsupialization (decompres-

sion) and enucleation [21]. The first is a relatively simple

procedure and consists of surgically producing a window in the

cystic wall to relieve intra-cystic tension [15]. The notable

disadvantages of the technique are: (a) it is a two-stage surgical

procedure, (b) pathological tissue is left behind and a more

sinister pathological process (i.e., squamous cell carcinoma) may

be overlooked, and (c) in a large cystic cavity, it takes a long

period of time for the bone to regenerate. So, enucleation with

primary closure is the treatment of choice [3,22]. It is a one-

stage surgical treatment followed by periodic radiographic

examinations at regular intervals to observe the progress of

bone regeneration of the defect and it also allows pathologic

examination of the entire specimen for histopathologic diagno-

sis [23]. However, enucleation can be done only when the jaw

bone adjacent to the cyst is intact [5].

Conservative treatment of DC, with maintenance of the

permanent teeth involved requires thorough evaluation of the

case by the professional in order to indicate the correct treatment,

the safety of total tumor removal and the possibility of

rehabilitating without the need for tooth replacement. So,

orthodontic treatment emerges as an important alternative to

the satisfactory conclusion of the case [24]. In the present case, the

enucleation of the cyst,with removal of associateddeciduous teethand maintenance of permanent ones was a good choice with

satisfactory results. The follow-up of patients surgically treated is

yearly panoramic radiograph, at least up to full bony regeneration

of the affected area [3]. In this case, the patient had excellent

recovery with new bone formation and eruption of included teeth.

Finally, an orthodontic evaluation will be performed to detect the

possible need of treatment for complete rehabilitation.

Although DCs have as recommended treatment the removal

of dental elements involved, there are cases in which conserva-

tive treatment can be considered. The present case report is

distinct as it enclosed three permanent maxillary teeth: the

central incisor, lateral incisor and canine, that could be

maintained during enucleation of the cyst and, at 2-year

follow-up, the patient presents good healing of the bony lesionwith converted teeth eruption.

Conflict of interest statement

The authors certify that they have no commercial or associative

interest that represents a conflict of interest in connection with the

manuscript.

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