/home/opencode/cpanel/stomaeduj_hacked/uploads/2019/10/art-Al-Kabuli-188-192.pdf MAXILLOFACIAL SURGERY AGGRESSIVE DENTIGEROUS CYST IN A 9-YEAR CHILD: Case Rreports A CASE REPORT AND REVIEW OF LITERATURE Juma Omran Al Khabuli1a* , Sally Mohamed1b, Huda Abutayyem1c , Said A. Abdelnabi2d 1 RAK College of Dental Sciences, RAK Medical and Health Sciences University, UAE 2 Al Saqr Hospital, Ras Al Khaimah, UAE a BDS, MDentSci, MFDS RCPS(Glasg), FICD, PhD, Associate Professor, Basic Sciences Department b BDS, MSc, PhD, Assistant Professor, Pediatric Dentistry c DDS, LDS RCS (Eng), MFDS RCS (Edin), MSc, PhD, Assistant Professor, Orthodontics d BDS, HDD, FDSRCS (Edin), Consultant, Oral and Maxillofacial Surgery ABSTRACT DOI: https://doi.org/10.25241/stomaeduj.2019.6(3).art.5 Aim: To report a case of aggressive dentigerous cyst associated with unerupted OPEN ACCESS This is an Open Access article mandibular 2nd premolar. under the CC BY-NC 4.0 license. Summary: Dentigerous cyst (DC) is a developmental odontogenic cyst, commonly Peer-Reviewed Article occurs between the 2nd and 3rd decade and is associated with the crown of Citation: Al Khabuli JO, Mohamed S, Abutayyem unerupted tooth. Unless infected, these cysts usually remain asymptomatic. H, Abdelnabi SA. Aggressive dentigerous cyst in a However, they may develop as a result of apical spread of inflammation from 9-year child: a case report and review of literature. Stoma Edu J. 2019;6(3):188-192. primary teeth causing pain, swelling and bone destruction. Received: July 20, 2019 A 9-year-old child presented to the pediatric clinic with pain and swelling for Revised: August 01, 2019 3 weeks. Clinical examination revealed endodontically treated lower left primary Accepted: August 04, 2019 Published: August 06, 2019 2nd molar with slight mobility. Also, there was an obvious expansion of the buccal *Corresponding author: plate. The radiographs and CBCT revealed large cystic lesion around the crown of Associate Professor Juma Omran Alkhabuli unerupted mandibular left 2nd premolar causing massive destruction of the buccal Basic Medical and Dental Sciences Department, RAK College of Dental Sciences, RAK Medical and Health and lingual plates. The cystic lesion was treated by enucleation and removal of Sciences University, UAE Tel: +97172222593, Fax: +971 7 2269997, the unerupted 2nd premolar tooth. The histopathology confirmed a diagnosis of e-mail: juma@rakmhsu.ac.ae dentigerous cyst. The follow-up demonstrated uneventful healing and good Copyright: © 2019 the Editorial Council for the prognosis. Stomatology Edu Journal. Key learning points: - It is crucial to follow-up any pulpally treated primary teeth. - There is a potential Infection spread from infected primary roots to the follicular tissues of permanent teeth that could instigate unexpected pathology. Keywords: Dentigerous Cyst; Enucleation; Marsupialization; Unerupted premolars 1. Introduction tooth follicle; however, there is a strong association Dentigerous cyst (DC) is the most common between DC development and inflammation developmental odontogenic cyst that invariably spreading from nonvital predecessor teeth [5-7]. occurs between the second and third decade and Generally, DCs are presented with no symptoms and its incidence is second to the radicular cyst [1,2]. The in many occasions are discovered during routine incidence of the DC in young individual is quite low; radiographic examination. Radiographically, they less than 10% within the first 10 years of life [3]. The are presented as a unilocular radiolucency around most common site for DC is the mandibular third the crown of unerupted tooth with a well-defined molar region followed by maxillary canine region. sclerotic border. In other instances, they may be Nevertheless, they may develop in association with symptomatic causing swelling, mobility of teeth, unerupted mandibular premolars or supernumerary and delay in eruption or pain if it is infected [8]. teeth, with a slight male predominance [4]. The Diagnosis of DCs is mostly based on the radiographic pathogenesis of these cystic lesions is not fully examination, however, a set of differential understood. However, it is believed that the diagnosis need to be made in cases with aberrant accumulation of fluids between the enamel surface presentation. Various treatment options of DCs have and the reduced enamel epithelium (epithelial been advocated, including surgical removal of the remnants of tooth-forming organs) leading to cyst and associated tooth, elimination of damage to separation of the latter and cyst formation. The the affected permanent teeth and marsupialization. cyst encloses the crown of the involved tooth and Nevertheless, complete removal of the pathological is attached to the cementoenamel junction. DCs cyst lining along with the involved tooth is preferred are considered developmental in origin, from a to avoid any future recurrence [3,8,9]. The aim of this 188 Stoma Edu J. 2019;6(3): 188-192. http://www.stomaeduj.com AGGRESSIVE DENTIGEROUS CYST IN A 9-YEAR CHILD: A CASE REPORT AND REVIEW OF LITERATURE Case Rreports Figure 1. Shows expansion of the buccal cortex. Figure 2. Shows oval-shaped radiolucency and mesial root resorption of the left mandibular 2nd primary molar. Figure 3. CBCT images (3D reconstruction) demonstrating massive destruction of the buccal cortex (A) and lingual cortex (B). CBCT axial view (C) and coronal view (D) demonstrating expansion of the buccal cortex and thinning of the lingual cortex. report is to present a case of aggressive DC involving knowledge dissemination without any details that unerupted lower left second premolar causing may identify individuals. massive bone resorption in a young female child. As a routine procedure, all patients or their 2. Case report guardians attending RAKCODS clinic are required to A 9-year-old female patient presented to the Pediatric electronically sign a consent form before launching Dentistry Department, RAKCODS clinic complaining any treatment procedure. Also, they are aware that of severe pain and swelling on the left side of the any tissues taken from patients or a performed mandible for the last 4 weeks. On examination, the procedure may be utilized for teaching and patient was healthy and the past medical history Stomatology Edu Journal 189 AGGRESSIVE DENTIGEROUS CYST IN A 9-YEAR CHILD: A CASE REPORT AND REVIEW OF LITERATURE Case Rreports Figure 4. A - shows reflection of the buccal flab and exposure of the lesion; B - shows the attachment of the cystic lining to the cervical margin of the tooth. revealed no relevant illnesses and the routine blood investigations were within the normal ranges. There was no history of hospitalization or trauma to the jaw. Extra-oral examination revealed a single diffuse swelling on the left side of the face with no sinus or active discharges. Intra-oral examination, showed a hard swelling in the 74, 75 regions with obliteration of the buccal vestibule (Fig. 1). The swelling was bony hard with expansion of the buccal cortex, with no evidence of the lingual cortex expansion. The primary left 2nd mandibular molar tooth was nonvisual, showing evidence of pulp therapy and composite filling. The tooth showed slight mobility and the adjacent soft tissues were normal with no signs of inflammation. The permanent first molar (36) was sound and the pulp vitality was not Figure 5. The radiograph demonstrates the regeneration of bone and compromised. Orthopantamograph (OPG) revealed the temporary space maintainer in place. an oval-shaped unilocular radiolucency around the propria showed heavy infiltrates of acute and developing second premolar with partial sclerotic chronic inflammatory cells. These appearances are border. The mesial root of 74 showed resorption consistent with DC. The sutures were removed after with loss of bone in the bifurcation area (Fig. 2). The one week, and the healing was uneventful. Soon cone beam computed tomography (CBCT) images after full eruption of 34 crown a space maintainer revealed thinning of the buccal and lingual cortex (band and loop) was fitted in place until further (Fig. 3 A-D). Based on the clinical, radiographical treatment (Fig. 5). Three months radiographic follow and CBCT examination, a provisional diagnosis of up demonstrated progressive bone regeneration dentigerous or bifurcation cyst was made. After filling the cavity and excellent soft tissue healing. consultation with the oral surgeon, it was decided to enucleate the cystic lesion surgically. A surgical 3. Discussion incision extending from the distal aspect of 32 to DC is a benign developmental cyst associated with the mesial aspect of 36 was established to expose the crown of un-erupted tooth and is the second most the involved area. The primary molars (74 & 75) were common odontogenic cyst [10]. Radiographically, it removed and the area was explored. The cystic lining appears as a solitary, well demarcated radiolucency of the lesion was found attached to the cervical margin of the 2nd premolar crown revealing a DC enclosing a crown of impacted tooth. The hallmark of (Fig. 4 A, B). Enucleation of the DC was established the cyst is the attachment of the follicular epithelium with extraction of the unerupted mandibular 2nd to the cemento-enamel junction. According to premolar, followed by primary closure of the wound. Zhang, et al. [11] the peak incidence is in the second The whole specimen was kept in10% buffered and third decade. In contrast to this finding, Shibata, formalin and sent for histopathology examination. et al. [12] showed that the age range of discovery of The histopathology examination reported a cystic the DC was 9-11 years. In the current case the child fibrous wall lined by non-keratinized stratified age was also 9-year-old. The noticed discrepancy squamous epithelium. Epithelial hyperplasia was may be attributed to the various studied ethnic noticed in many areas of the lining. The lamina groups of population. 190 Stoma Edu J. 2019;6(3): 188-192. http://www.stomaeduj.com AGGRESSIVE DENTIGEROUS CYST IN A 9-YEAR CHILD: A CASE REPORT AND REVIEW OF LITERATURE DC is commonly associated with mandibular 3rd examination. In our case, enucleation of the cyst Case Rreports mandibular molar [13]. However, in the current including the unerupted tooth approach was case the cyst was associated with the unerupted chosen. Radiographically, there was substantial mandibular 2nd premolar. Although such cases are bone destruction and examination of the entire relatively rare, a few cases have been reported [14]. cystic tissues was deemed necessary. It was obvious Shibata et al. [12] studied the occurrence of DCs in that the cystic lining was attached to neck of the association with succedaneous teeth during the tooth and the histopathology report showed severe transitional dentition phase and reported 77.1% inflammation masking the classical microscopic prevalence in the premolar region. There have been appearance. Three months post-operative follow-up several explanations for the development of DC. The showed uneventful healing. The radiograph showed experimental and clinical observations propose two that the cystic cavity is completely filled with bone. types of DCs; inflammatory and non-inflammatory, The band and loop space maintainer was provided instigated by different causes and develop at until further orthodontic treatment is instituted. different stages of tooth development [15]. Three pathways were suggested for histogenesis of the 4. Conclusion DC. In the first scenario, the developmental DC Development of DC in association with an unerupted forms from dental follicle and becomes secondarily successor due to inflammatory change at the apex of infected as a result of a non-vital tooth. The second a deciduous tooth is not uncommon. Although, DCs type occurs when a permanent successor erupts are asymptomatic, they may cause pain, swelling into radicular cyst that forms at apex of a non-vital and massive bone destruction. Therefore, close deciduous resulting into a DC that is extra follicular monitoring of pulpally treated primary molars is in origin. Nevertheless, a radicular cyst developing crucial to prevent or reduce the potential morbidity at apex of primary tooth is extremely rare. The associated with the same. third possible cause is due to spread of peri-apical inflammation from a non-vital deciduous tooth to Acknowledgments follicle of permanent successor [7]. We would like to appreciate the help of the of oral Two main surgical approaches are usually followed surgery department team in Saqr hospital, RAK for for management of such cystic lesions; either accommodating the case in spite of the busy oral enucleation or marsupialization. Several factors are surgery schedule. taken into consideration, such as the size and location of the lesion, the amount of bone loss, integrity of Disclosure statement the cystic wall, and its relation to vital structures. The authors report no conflict of interest Conservative approach, the marsupialization has been advocated for management of DC in children Ethical Issue to provide a chance for the unerupted tooth to The guardian of the patient has signed a consent form erupt [16]. 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Mandibular dentigerous cyst in a 10-year-old child. Int J Clin Pediatr Dent. 2016; 9(3): 281-284. [Full text links] [PubMed] Google Scholar Juma Omran AL KHABULI BDS, MDentSci, MFDS RCPS (Glasg), FICD, PhD Associate Professor, Chair Basic and Medical Sciences Department RAK College of Dental Sciences RAK Medical and Health Sciences University RAK, UAE CV Obtained his BDS from Garyounis University, Libya (1985); MDentSci and PhD from Leeds University, UK (2005); MFDS RCPS (Glasgow) and FICD (2014). Worked as GP for 14 years in various health sectors, Libya before specializing in oral pathology. In 2008 joined RAK College of Dental Sciences, RAKMHSU as associate professor and was one of the founding faculty, and contributed massively in its academic accreditation and curriculum development. His main interest is teaching oral biology, oral pathology and oral medicine. In terms of research, his main research theme is directed towards molecular biology of oral cancer. Questions 1. A classical dentigerous cyst is classified as: qa. Inflammatory; qb. Developmental; qc. Hereditary; qd. Reactive. 2. Diagnosis of dentigerous cyst is mainly based on: qa. Clinical examination; qb. Biopsy; qc. Radiographic examination; qd. History taken. 3. Which of the following is a primary differential diagnosis of a dentigerous cyst associated with unerupted premolar tooth? qa. Bifurcation cyst; qb. Radicular cyst; qc. Paradental cyst; qd. Periodontal cyst. 4. A dentigerous cyst can be treated conservatively by: qa. Cyst enucleation; qb. Surgical extraction; qc. Orthodontic; qd. Marsupialization. 192 Stoma Edu J. 2019;6(3): 188-192. http://www.stomaeduj.com