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Received: 15 May 2018 | Accepted: 30 August 2018

DOI: 10.1111/jop.12776

ORIGINAL ARTICLE

Histological analysis of 41 dentigerous cysts in a paediatric


population

Gwendolyn Huang1 | Lynette Moore2,3 | Richard M. Logan4 | Sumant Gue1,5

1
Department of Paediatric Dentistry,
Division of Paediatric Surgery, Women's
Background: Dentigerous cysts are usually of developmental nature but may be of
and Children's Hospital, Adelaide, South inflammatory origin especially in paediatric populations. It is important to understand
Australia, Australia
2
the histological features of dentigerous cysts to enable accurate diagnosis. The aim
Surgical Pathology, Women's and
Children's Hospital, Adelaide, South of this study is to present epidemiological, clinical features and histopathological
Australia, Australia features of dentigerous cysts seen in a paediatric tertiary referral hospital.
3
University Discipline of Pathology, The
Method: The medical, radiographic and histopathology records of the Department
University of Adelaide, Adelaide, South
Australia, Australia of Pathology, Women's and Children's Hospital, Adelaide, Australia, during January
4
Department of Oral Pathology, School of 1998 to December 2013 were reviewed for patients with dentigerous cysts. All
Dentistry, The University of Adelaide,
Adelaide, South Australia, Australia cases were re‐examined by a specialist oral pathologist, consultant paediatric pathol-
5
Paediatric Dentistry, School of Dentistry, ogist and paediatric dentistry registrar.
Faculty of Health Sciences, The University
Results: Forty‐one cases of dentigerous cysts were found. Patients in the perma-
of Adelaide, Adelaide, South Australia,
Australia nent dentition were most frequently affected. Male predilection was observed
(male:female 2.42:1). The posterior mandible was the most frequently affected
Correspondence
Gwendolyn Huang, Department of Paediatric region (63.42%) although maxillary canines were the teeth most commonly associ-
Dentistry, Women's and Children's Hospital,
ated with dentigerous cysts (29.27%). The majority of cases were incidental findings.
North Adelaide, SA, Australia.
Email: gwendolyn.huang@sa.gov.au Squamous epithelium showing pseudoepitheliomatous hyperplasia (46%) was fre-
quently observed and was significantly present with thicker epithelium (P < 0.0001)
and an acute and chronic inflammatory infiltrate (P < 0.001). Inflammatory infiltrate
was seen in 75.6% of cases.
Conclusions: The current study provides increased knowledge of the histological
features of dentigerous cysts in a large retrospective series of paediatric patients
and provides further evidence regarding the frequency of inflammatory dentigerous
cysts.

KEYWORDS
Australia, dentigerous cyst, epidemiology, paediatric, treatment

1 | INTRODUCTION between the reduced enamel epithelium and the enamel or within
the enamel organ.4,5 Proliferation of the epithelium may be induced
Dentigerous cysts are a common odontogenic cyst and are associ- by osmotic pressure from the fluid‐filled sac when the tooth is
ated with the crowns of unerupted teeth. In paediatric populations, impacted.6
1-3
the frequency of dentigerous cysts ranges from 3.56%‐8.64%. Dentigerous cysts are usually clinically asymptomatic and are fre-
Dentigerous cysts are thought to result from proliferation of reduced quently detected during routine radiographic examination or from
enamel epithelium after enamel formation of tooth development. delayed eruption of a permanent tooth.7 Occasionally, there may be
The pathogenesis of dentigerous cysts is unclear. It is proposed that acute inflammatory exacerbation leading to clinical symptoms.8
dentigerous cysts develop from abnormal fluid accumulation Subtle facial asymmetry may be observed as cysts increase in size,

74 | © 2018 John Wiley & Sons A/S. wileyonlinelibrary.com/journal/jop J Oral Pathol Med. 2019;48:74–78.
Published by John Wiley & Sons Ltd
HUANG ET AL. | 75

which may then prompt patients to seek professional assistance. Histological analysis was conducted on all diagnosed dentigerous
Early identification and management of dentigerous cysts are impor- cysts to confirm histopathological diagnosis and to determine any
tant as it may avoid potential negative sequelae including failure of variability within the series. The cyst walls showed a variety of
eruption, impaction of associated and surrounding teeth, displace- epithelial appearances including reduced enamel epithelium (24.39%),
ment of teeth, root resorption of adjacent roots, destruction of bone, transitional epithelium (9.76%) and non‐keratinised stratified squa-
encroachment on vital structures and occasionally pathologic frac- mous epithelium (48.78%) (Figures 1-3). Squamous epithelium show-
ture. Carcinomatous transformation has also been reported, although ing pseudoepitheliomatous hyperplasia (53.66%) was frequently
9
very rare. observed. Ciliated epithelium and calcific deposits within the epithe-
Dentigerous cysts are frequently encountered in paediatric lium were occasionally observed (4.88% and 2.44%, respectively).
patients; however, there have been few large studies assessing their The epithelium merged with hyperplastic non‐keratinised stratified
histopathological features.6 This study investigates the histopatho- squamous epithelium of variable thickness, sometimes with anasto-
logical features of dentigerous cysts in a series of paediatric patients. mosing rete ridges. Particularly where the hyperplastic epithelium
was observed, a significant acute and chronic inflammatory infiltrate
was also noted in the connective tissue (P < 0.001). An inflammatory
2 | METHODS infiltration was seen in 75.6% cases, of which 74.2% cases were in
patients aged 12 years or younger (Figure 4). An acute inflammatory
The study was approved by the Children, Youth and Women's cell infiltrate was observed in 65.85% of cases and all cases of acute
Health Service Human Research Ethics Committee, Women's and inflammation also had signs of chronic inflammation.
Children's Hospital (WCH), Adelaide, South Australia (ethics number Vascular proliferation within the epithelium was occasionally
463A). The medical files of all paediatric patients who were treated observed (19.51%), particularly where pseudoepitheliomatous hyper-
at the WCH from 1998 to 2015 for dentigerous cysts were plasia (P = 0.004) and considerable inflammatory infiltrate were pre-
retrieved. Dentigerous cyst cases included in the study had to meet sent (P = 0.004). Cholesterol clefts, foreign body giant cells and
the following criteria to avoid misclassification: (a) the histopatholog- lymphoid follicles were very occasionally observed (9.76%, 2.44%
ical diagnosis of dentigerous cysts with available tissue slides or and 2.44%, respectively) (Figure 5). Hyaline connective tissue was
blocks; (b) a radiographic record of a pericoronal radiolucency of an observed in 26.83% of patients. Cases where pseudoepitheliomatous
unerupted or impacted tooth; and (c) a description of a cystic lesion hyperplasia was observed were associated with thicker epithelium
in the operation record. All cases were re‐examined by a specialist (P < 0.0001) whereas the presence of reduced enamel epithelium
oral pathologist, consultant paediatric pathologist and paediatric den- was accompanied by thinner cyst epithelium (P = 0.0007). Epithelial
tistry registrar. Demographic data including the age and gender of rests (Figure 6) were also observed in 19.51% of cases. Reduced
patients, presenting symptoms, the involved teeth/region, size, radio- enamel epithelium was not associated with the presence of either
graphic appearance, treatment provided and surgical department acute or chronic inflammation (P < 0.05). The presence of haemor-
were reviewed and recorded. Data are presented as means (with rhagic tissue was also statistically significant in cases where pseu-
standard deviation) and medians (with range) descriptive statistics or doepitheliomatous hyperplasia was observed (P = 0.05).
proportions as required. All analyses were completed using SAS v9.3 Radiographically, the cysts surrounded the crowns of the uner-
(SAS Institute, Cary, NC, USA). Statistical significance was assessed upted teeth as unilocular well‐defined radiolucencies ranging in size.
using Fisher's exact test and set at the 5% alpha level. All tests were
two‐tailed.

3 | RESULTS

A total of 41 dentigerous cysts originating from 41 primary, perma-


nent and supernumerary teeth were included in this study. Forty‐
one patients were affected, with the male‐to‐female ratio as 2.42:1.
The mean age of patients was 11.05 years (±2.78 years, range 4‐
16 years). Teeth most commonly associated with dentigerous cysts
were canines (33.33%) followed by second premolars (20.83%). All
dentigerous cysts in this case series were diagnosed either as inci-
dental findings on radiographic examination or associated with
delayed eruption of permanent teeth. The exception was one case
which presented with acute symptoms to the Emergency Depart-
ment, WCH. The acute symptoms included pain, buccal and infraor- F I G U R E 1 Squamous epithelium showing pseudoepitheliomatous
bital facial swelling and strabismus of the ipsilateral eye. hyperplasia, 10× magnification (Patient 23)
76 | HUANG ET AL.

FIGURE 2 Reduced enamel epithelium, 40× magnification FIGURE 5 Cholesterol clefts, 40× magnification (Patient 36)
(Patient 6)

F I G U R E 6 Epithelial rests within the connective tissue, 40×


magnification (Patient 6)
FIGURE 3 Ciliated epithelium, 40× magnification (Patient 25)

The sizes of dentigerous cysts in their largest dimension ranged from


6 to 60 mm (average 18.04 mm). 79.16% of lesions were 20 mm or
smaller, with only 20.83% larger than 20 mm. All lesions were
unilocular.

4 | DISCUSSION

The clinical and radiographic presentations are important in estab-


lishing a clinical differential diagnosis. Differential diagnoses of
dentigerous cysts include enlarged dental follicles, radicular cysts,
inflammatory paradental cysts, odontogenic keratocysts, unicystic
ameloblastomas, mural ameloblastomas, ameloblastic fibromas, Pind-
borg tumours, odontogenic fibromas and adenomatoid odontogenic
tumour.7,10,11
Dentigerous cysts are usually asymptomatic, with the majority of
small cysts identified incidentally through radiographic examination
F I G U R E 4 Inflammatory infiltrate with pseudoepitheliomatous
hyperplasia of the epithelium, 10× magnification (Patient 16) or from delayed eruption of a permanent tooth.7 Dentigerous cysts
HUANG ET AL. | 77

may present at any age but frequently present in the second or third in 23.8% of 130 sampled dentigerous cysts and ciliated cells in the
decades of life.12,13 The average age of children with dentigerous epithelial linings of 10.8%, indicating the possible mutual presence.26
cysts in this study was 11.05 years, which is when the permanent The type of dentigerous cyst observed may help explain their
premolars and canines have their greatest eruptive potential and aetiology. Developmental cysts are theorised to arise from impaction
widening of the dental follicle is observed as part of the eruptive of mature teeth.24 Comparatively, inflammatory dentigerous cysts
process. An increasing presentation was noted in this study with may result from inflammation from a non‐vital deciduous tooth or
increasing age although dentigerous cysts occurred at younger ages other inflammatory source, which had spread to involve the tooth
in males than females (mean age 10.62 years vs 12.08 years, respec- follicle.27 In 1928, Bloch‐Jorgensen suggested that the origin of all
tively). Allon et al reported an average age of 11.6 years (range 7‐ follicular cysts in his series was from an overlying necrotic deciduous
18 years) at presentation.14 70.16% of dentigerous cysts occurred in tooth.27 Shaw et al reported 13 cases of dentigerous cysts involving
the mixed dentition (6‐12 years) group. The late mixed dentition per- permanent premolars that were associated with infected primary
iod corresponds to the time at which the crowns of permanent teeth teeth.28 Shibata et al retrospectively assessed 47 premolars associ-
have completely developed and have commenced eruption. Thus, ated with dentigerous cysts, with the overlying deciduous molar still
dentigerous cysts in paediatric populations may occur more fre- present.17 93.6% of these cases had possible inflammation at the
quently in the late mixed dentition stage because during this time, deciduous tooth associated with the dentigerous cyst and inflamma-
there is higher probability of impaction of the maxillary canines as tory cellular infiltration in the epithelial lining and subepithelial con-
well as higher probability of periapical inflammation from a non‐vital nective tissue was observed histopathologically in 98.1%. It is highly
deciduous tooth spreading to involve the follicle of the unerupted likely that in younger children, inflammatory dentigerous cysts occur.
permanent succedaneous tooth. In our case series, inflammatory infiltration was seen in 75.6% cases,
Radiographically, dentigerous cysts present as a well‐defined of which 74.2% cases were in patients aged 12 years of younger. It
radiolucent lesion of alveolar bone. The cyst may partially or com- is not possible to know if the presence of a non‐vital deciduous
pletely encircle the crown of an unerupted tooth, contacting the tooth was associated with the dentigerous cyst as, in majority of our
tooth at the cemento‐enamel junction (CEJ).15-17 Depending on cases, the deciduous teeth were not present at the time of diagnosis
the size of the lesion, small dentigerous cysts can present radio- of the cysts. However, the most commonly associated tooth in this
graphically as eruption cysts or enlarged dental follicles. In adult study was the canine, with premolars accounting for 29.3% of cases.
populations, studies have recommended that follicular tissue 53.7% of our cases exhibited pseudoepitheliomatous hyperplasia of
greater than 3‐4 mm should be submitted for histopathological the squamous lining. Given these histological features, it is likely that
analysis as 28%‐46% of unerupted third molars are associated inflammatory dentigerous cysts in paediatric patients may arise
18-21
with dentigerous cysts. There are no recommendations for because of periapical inflammation of a deciduous tooth, which
paediatric populations, whereby enlarged dental follicles are likely spreads to involve the underlying follicle of the unerupted perma-
to be encountered. nent succedaneous tooth. Consequently, it may be prudent to dis-
It may be more likely to encounter inflammatory dentigerous cuss cyst formation as a potential complication of non‐vital primary
cysts in paediatric populations. In this case series, 75.6% of cases teeth. Follow‐up of patients should also be maintained until the suc-
were considered as inflammatory dentigerous cysts with inflamma- cedaneous tooth has erupted.
tory infiltrate present. Unless there are clinical symptoms, histologi- The current study provides information regarding histological
cal analysis is the only way to differentiate between developmental features of dentigerous cysts in a large series of paediatric patients.
and inflammatory dentigerous cysts. Histopathology of developmen- It also provides further evidence supporting the histogenesis of
tal dentigerous cysts typically reveals a fibrous cyst wall with multi- inflammatory dentigerous cysts in paediatric populations and the
ple different cell types including lymphocytes, histiocytes, plasma importance of continued dental follow‐up.
cells and epithelial cells. The lining of the cyst typically consists of
two or three layers of non‐keratinised stratified epithelial cells.7,22 In
ACKNOWLEDGEMENTS
inflamed dentigerous cysts, the cyst wall may be thickened. In
response to inflammation, localised proliferation of the epithelial lin- There are no acknowledgements.
ing has been reported in the literature and observed in this case ser-
ies.23-25 Nests, islands or strands of odontogenic epithelium can also
CONFLICT OF INTERESTS
be seen in the capsule. Discontinuance of the epithelial lining may
be observed where intense inflammatory infiltrate is present in the There are no conflict of interests and the authors have nothing to
adjacent capsule. Aspiration of the cyst reveals a transudate cyst disclose.
fluid, which is thin, watery and clear/straw‐coloured. Inflammatory
infiltration can also be observed. Part of the epithelial lining may
ORCID
contain mucus‐producing cells or ciliated cells, possibly as a result
from metaplasia. Takeda et al observed the presence of mucous cells Gwendolyn Huang http://orcid.org/0000-0002-9009-8309
78 | HUANG ET AL.

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