International Journal of Pediatric Otorhinolaryngology Extra 8 (2013) e1e4

Contents lists available at SciVerse ScienceDirect

International Journal of Pediatric Otorhinolaryngology Extra

journal homepage: www.elsevier.com/locate/ijporl

Case Report

Conservative management of dentigerous cyst in a child

jo Lima a,*, Conceic o Aparecida Dornelas Monteiro Maia a, Emeline das Neves de Arau a cia Teixeira de Oliveira c, Ana Miryam Costa de Medeiros d Alberto Costa Gurgel b, Patr
a

Department of Oral Pathology, Federal University of Rio Grande do Norte, Natal, RN, Brazil Maxillofacial Surgery, Potiguar University (UNP), Natal, RN, Brazil c Oral Radiology, Federal University of Rio Grande do Norte, Natal, RN, Brazil d Stomatology, Federal University of Rio Grande do Norte, Natal, RN, Brazil
b

A R T I C L E I N F O

A B S T R A C T

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

Dentigerous cysts are odontogenic cysts associated with the crowns of unerupted permanent teeth. This article reports the case of a 10-year-old child who presented permanence of deciduous teeth 51, 52, and 53. Panoramic radiograph showed extensive cystic lesion involving the teeth 11, 12 and 13, which were included. Diagnosis of dentigerous cysts was conrmed 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.

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 inammatory 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 cyst with uid accumulation between the crown and enamel organ of an unerupted tooth [4]. Buccal bony expansion is the most common clinical feature [5] related to epithelial proliferation, release of boneresorbing factors and an increase in cyst uid osmolality [6]. It is the second most common odontogenic cyst, with a frequency ranging from 11.4% to 44% and is more frequently seen in the second decade of life [1]. Few of these cysts have 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 inammatory origin has also been suggested [7]. It has been reported that the progression of inammation in the root apex of a deciduous tooth may be related to the development of a DC in the unerupted 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-dened radiolucency surrounding the crown of an unerupted tooth, which often has a sclerotic border. Histologically, the DC consists of a brous wall lined by nonkeratinized, stratied, squamous epithelium, presenting myxoid tissue, odontogenic remnants and, rarely, sebaceous cells [12]. Removal of the entire cyst associated with the extraction of the impacted tooth is the main treatment to prevent recurrence. 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

* Corresponding author at: Federal University of Rio Grande do 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). 1871-4048/$ see front matter 2012 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.pedex.2012.10.005

e2

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

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

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 teeth 51, 52 and 53 showed wear facets compatible with bruxism. The right central incisor was discolored with evidence of

pulp disturbance. Panoramic and occlusal radiographs showed a large, well-dened radiolucency surrounding the retained permanent teeth 11, 12 and 13. Based on clinical and radiographic ndings, 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 conrmed. After 2 years of follow-up, the impacted teeth are positioning itself spontaneously toward its eruption and the patient was referred for orthodontic assessment (Figs. 15). 3. Discussion The distribution and characteristics of jaw cysts in children are different from those in adults. In children there is a relatively high rate of developmental cysts, whereas in adults the inammatory cysts are more common [14]. The DC is the second most common

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.

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

e3

Fig. 4. Photomicrographs showing a cystic lesion lined by nonkeratinised, stratied, squamous epithelium. Note the intense inammatory inltrate 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.

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 impacted tooth. However, Jones et al. [16] reported a peak incidence between the fth and sixth decades. The difference in prevalence of developmental cysts is probably related to the fact that during the pediatric age period, the jaws are involved in profound developmental processes. 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 ndings in the literature, but differs with respect to the number of teeth involved and the success of a conservative treatment.

Swelling can be experienced if the DC grows too large or lies in a sensitive area. When the cyst occupies the whole maxillary 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 inammation from any source but usually from a nonvital deciduous tooth and spreading to involve the follicles of the unerupted permanent successors. The inammatory 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 inammatory in nature [11,19,20]. In this case, the infection in a primary tooth pulp may have originated the inammatory 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 denitive 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

e4

E.N.A. Lima et al. / International Journal of Pediatric Otorhinolaryngology Extra 8 (2013) e1e4 children: a conservative approach and 7-year follow-up, Journal of Applied Oral Science 20 (2012) 268271. Y. Ishihara, H. Kamioka, T. Takano-Yamamoto, T. Yamashirod, Patient with nonsyndromic bilateral and multiple impacted teeth and dentigerous cysts, American Journal of Orthodontics and Dentofacial Orthopedics 141 (2012) 228241. A. Tamgadge, S. Tamgadge, D. Bhatt, S. Bhalerao, T. Pereira, M. Padhye, Bilateral dentigerous cyst in a non-syndromic patient: report of an unusual case with review of the literature, Journal of Oral and Maxillofacial Pathology 15 (2011) 9195. Y.H. Hu, Y.L. Chang, A. Tsai, Conservative treatment of dentigerous cyst associated with primary teeth, Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology and Endodontics 112 (2011) 57. G.Z. Xu, Q. Jiang, C. Yang, C.G. Yu, Z.Y. Zhang, Clinicopathologic features of dentigerous cysts in the maxillary sinus, Journal of Craniofacial Surgery 23 (2012) 226231. D.S. Pramod, J.N. Shukla, Dentigerous cyst of maxilla in a young child, National Journal of Maxillofacical Surgery 2 (2011) 196199. R. Suresh, M. Janardhanan, A.P. Joseph, R.B. Vinodkumar, S. Peter, A rare case of dentigerous cyst in a one year old child: the earliest known reported occurrence, Head and Neck Pathology 5 (2011) 171174. L.S. de Andrade Freitas Oliveira, D.O. Souza, F.S. Neves, J.N. Dos Santos, P.S. -Rebello. Uncommon dentigerous cyst related to a maxillary Campos, I. Crusoe lateral incisor in a 3-year-old boy. Oral and Maxillofacial Surgery, http:// dx.doi.org/10.1007/s10006-011-0301-x, in press. T.N. Tilakraj, N.K. Kiran, K.S. Mukunda, S. Rao, Non syndromic unilateral dentigerous cyst in a 4-year-old child: a rare case report, Contemporary Clinical Dentistry 2 (2011) 398401. R.S. Narang, A.S. Manchanda, P. Arora, K. Randhawa, Dentigerous cyst of inammatory origin a diagnostic dilemma, Annals of Diagnostic Pathology 16 (2012) 119123. K.P. Bharath, V. Revathy, S. Poornima, V.V. Subba Reddy, Dentigerous cyst in an uncommon site: a rare entity, Journal of the Indian Society of Pedodontics and Preventive Dentistry 29 (2011) 99103. E. Bozdogan, B. Cankaya, K. Gencay, O. Aktoren, Conservative management of a large dentigerous cyst in a 6-year-old girl: a case report, Journal of Dentistry for Children (Chicago) 78 (2011) 163167. M. Rohilla, R. Namdev, S. Dutta, Dentigerous cyst containing multiple impacted teeth: a rare case report, Journal of the Indian Society of Pedodontics and Preventive Dentistry 29 (2011) 244247. E. Manor, L. Kachko, M.B. Puterman, G. Szabo, L. Bodner, Cystic lesions of the jaws A clinicopathological study of 322 cases and review of the literature, International Journal of Medical Sciences 9 (2012) 2026. A.V. Jones, G.T. Craig, C.D. Franklin, Range and demographics of odontogenic cysts diagnosed in a UK population over a 30-year period, Journal of Oral Pathology and Medicine 35 (2006) 500507. E.K. Sannomiya, Q. Nogueira Mde, C. Diniz Mde, F.O. Pacca, S. Dalben Gda, Traumainduced dentigerous cyst involving the anterior maxilla, Journal of Dentistry for Children (Chicago) 74 (2007) 161164. R.R. Kalaskar, A.R. Kalaskar, Multidisciplinary management of impacted central incisors due to supernumerary teeth and an associated dentigerous cyst, Contemporary Clinical Dentistry 2 (2011) 5358. A. Benn, M. Altini, Dentigerous cysts of inammatory origin. A clinicopathologic study, Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology and Endodontics 81 (1996) 203209. N.T. Prabhu, J. Rebecca, A.K. Munshi, Dentigerous cyst with inammatory etiology from a deciduous predecessor report of a case, Journal of the Indian Society of Pedodontics and Preventive Dentistry 14 (1996) 4951. ller, R.C. Gugisch, Treatment of a R. Scariot, D.J. da Costa, N.L. Rebellato, P.R. Mu large dentigerous cyst in a child, Journal of Dentistry for Children (Chicago) 78 (2011) 111114. R.R. Kalaskar, A. Tiku, S.G. Damle, Dentigerous cysts of anterior maxilla in a young child: a case report, Journal of the Indian Society of Pedodontics and Preventive Dentistry 25 (2007) 187190. J.S. Thakur, N.K. Mohindroo, D.R. Sharma, R.S. Minhas, A. Thakur, Ectopic canine associated with a dentigerous cyst in the maxilla, Ear, Nose, and Throat Journal 90 (2011) 2527. P.K. Shivaprakash, T. Rizwanulla, D.K. Baweja, H.H. Noorani, Save-a-tooth: conservative surgical management of dentigerous cyst, Journal of the Indian Society of Pedodontics and Preventive Dentistry 27 (2009) 5257.

basic surgical procedures, namely marsupialization (decompression) and enucleation [21]. The rst 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 onestage 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 diagnosis [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 associated deciduous teeth and 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 conservative 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 lesion with converted teeth eruption. Conict of interest statement The authors certify that they have no commercial or associative interest that represents a conict of interest in connection with the manuscript. References

[3]

[4]

[5]

[6]

[7] [8]

[9]

[10]

[11]

[12]

[13]

[14]

[15]

[16]

[17]

[18]

[19]

[20]

[21]

[22]

[23] [1] M. Selvamani, M. Donoghue, P.S. Basandi, Analysis of 153 cases of odontogenic cysts in a South Indian sample population: a retrospective study over a decade, Brazilian Oral Research 26 (2012) 330334. rio-Homem, [2] M.C.Z. Deboni, M.A. Brozoski, A.A. Traina, R.R. Acay, M.daG. Nacle Surgical management of dentigerous cyst and keratocystic odontogenic tumor in

[24]