art-Al-Kabuli-188-192

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]. Nevertheless, it has a disadvantage that in his mother’s tongue language with translation
the pathology of the cystic lining is left behind and understood that no identifying details would be
and remains without thorough histopathological declared in any form.

References
1. Suresh R, Janardhanan M, Joseph AP, et al. A rare case of 7. Benn A, Altini M. Dentigerous cysts of inflammatory origin. A
dentigerous cyst in a one-year old child: the earliest known clinicopathologic study. Oral Surg Oral Med Oral Pathol Oral Radiol
reported occurrence. Head Neck Pathol. 2011; 5(2):171-174. Endod. 1996;81(2):203-209.
[Full text links] [CrossRef ] [PubMed] Google Scholar Scopus [Full text links] [PubMed] Google Scholar Scopus
2. Al Sheddi MA. Odontogenic cysts. A clinicopathological study. 8. Şahin O. Conservative management of a dentigerous cyst
Saudi Med J. 2012; 33(3):304-308. associated with eruption of teeth in a 7-year-old girl: a case report.
[PubMed] Google Scholar Scopus J Korean Assoc Oral Maxillofac Surg. 2017; 43(Suppl 1): S1–S5.
3. Ghandour L, Bahmad HF, Bou-Assi S. Conservative treatment [Full text links] [Free PMC Article] [CrossRef ] [PubMed] Google
of dentigerous cyst by marsupialization in a young female Scholar
patient: a case report and review of the literature. Case Rep Dent. 9. Kirtaniya BC, Sachdev V, Singla A, Sharma AK. Marsupialization:
2018;2018:7621363. A conservative approach for treating dentigerous cyst in children
[Full text links] [Free PMC Article] [CrossRef ] [PubMed] Google in the mixed dentition. J Indian Soc Pedod Prev Dent. 2010;
Scholar 28(3):203–208.
4. Manjunatha BS, Chikkaramaiah S, Panja P, Koratagere N. [Full text links] [PubMed] Google Scholar Scopus
Impacted maxillary second premolars: a report of four cases. BMJ 10. Villasis-Sarmiento L, Portilla-Robertson J, Melendez-
Case Rep. 2014;2014:1–4. Ocampo A, et al. Prevalence and distribution of odontogenic
[Full text links] [Free PMC Article] [CrossRef ] [PubMed] Google cysts in a Mexican sample. A 753 cases study. J Clin Exp Dent.
Scholar Scopus 2017;9(4):e531-e538.
5. Gupta P, Jawanda MK, Narula R, Singh J. Inflammatory [Full text links] [CrossRef ] [PubMed] Google Scholar Scopus
dentigerous cyst mimicking a periapical cyst. J Int Clin Dent Res 11. Zhang LL, Yang R, Zhang L, et al. Dentigerous cyst: a
Organ. 2016;8(1):63-66. retrospective clinicopathological analysis of 2082 dentigerous
[Full text links] [CrossRef ] [PubMed] Google Scholar cysts in British Columbia, Canada. Int J Oral Maxillofac Surg.
6. Srividhya SS, Anand B, Mahesh Kumar P. Dentigerous cyst of 2010;39(9):878-882.
inflammatory origin- report of a rare case. WJPPS. 2018;7(9):1064-1069. [Full text links] [CrossRef ] [PubMed] Google Scholar Scopus

Stomatology Edu Journal 191
AGGRESSIVE DENTIGEROUS CYST IN A 9-YEAR CHILD:
A CASE REPORT AND REVIEW OF LITERATURE

12. Shibata Y, Asaumi J, Yanagi Y, et al. Radiographic examination 15. Al-Talabani NG, Smith CJ. Experimental dentigerous cysts and
Case Rreports of dentigerous cysts in the transitional dentition. Dentomaxillofac enamel hypoplasia: their possible significance in explaining the
Radiol. 2004;33(1):17-20. pathogenesis of human dentigerous cysts. J Oral Pathol. 1980;
[Full text links] [PubMed] Google Scholar Scopus 9(2):82-91. [PubMed] Google Scholar Scopus
13. Terauchi M, Akiya S, Kumagai J, et al. An analysis of dentigerous 16. Taysi M, Ozden C, Cankaya AB, et al. Conservative approach to
cysts developed around a mandibular third molar by panoramic a large dentigerous cyst in an 11-year-old patient. J Istanb Univ Fac
radiographs. Dent J (Basel). 2019;7(1).pii:E13. Dent. 2016;50(3):51-56.
[Full text links] [Free PMC Article] [CrossRef ] [PubMed] Google [Full text links] [CrossRef ] [PubMed] Google Scholar
Scholar Scopus
14. Bhardwaj B, Sharma S, Chitlangia P, et al. 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