Acta Odontologica Scandinavica.

2014; Early Online, 1–6

ORIGINAL ARTICLE

Prevalence of developmental odontogenic cysts in children and

adolescents with emphasis on dentigerous cyst and odontogenic
keratocyst (keratocystic odontogenic tumor)

NANNAN LI1*, XING GAO2*, ZIYUAN XU3, ZHUO CHEN1, LAIKUAN ZHU3,
JINRUI WANG1 & WEI LIU3
1
School of Stomatology, Jilin University, Changchun, Jilin, China, 2Department of Oral and Maxillofacial Surgery,
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XiangYa Hospital, Central South University, Changsha, Hunan, China, and 3Shanghai Key Laboratory of
Stomatology, Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China

Abstract
Objective. To investigate the incidence and prevalence of developmental odontogenic cysts in children and adolescents and
compare the features of the two most common types, dentigerous cyst and keratocystic odontogenic tumor (KCOT). Study
design. A retrospective review in a series of 369 patients with all histological diagnoses of developmental odontogenic cysts in
children (£12 years) and adolescents (13–18 years) was conducted. Results. Among these, 361 (97.8%) patients were
diagnosed as dentigerous cyst (n = 281) and KCOT (n = 80), with the male-to-female ratios of dentigerous cyst and KCOT
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both being 2:1. The average age of the patients with KCOT was older than that of those with dentigerous cyst (14.7 years vs
11.8 years, p < 0.001). Dentigerous cyst (59.1%) was more common in children, but KCOT (78.8%) was more common in
adolescents (p < 0.001). Dentigerous cyst (57.6%) predominantly located on the maxilla, but KCOT (60.3%) predominantly
located on the mandible (p = 0.010). Conclusions. Adolescent patients with lesions located on the mandible would favor
KCOT over dentigerous cyst. This study aids in better knowledge of the prevalence of developmental odontogenic cysts in a
large pediatric population, and shows that a well-supported early diagnosis is indispensable for a more adequate treatment.

Key Words: developmental odontogenic cysts, dentigerous cyst, keratocystic odontogenic tumor, pediatrics

Introduction cyst, eruption cyst, gingival cyst of infant and lateral

periodontal cyst [2,3].
Odontogenic cysts are the common osseous- Cystic lesions in the jaws share similarities in
destructive lesions affecting the jaws and they classified clinical and radiographic as well as symptomatology,
into developmental or inflammatory cysts based on especially between the dentigerous cyst and KCOT
their origin and pathogenesis. In children there is a [4]. Radiographically, a dentigerous cyst usually
relatively high rate of developmental cysts, whereas in presents as a unilocular and pericoronal radiolucent
adults inflammatory cysts are more common [1,2]. lesion of an unerupted or impacted tooth in the jaws.
Dentigerous cyst is the most common developmental The radiographic picture alone is insufficient for
odontogenic cyst of the jaw bones and rarely recurs. definite diagnoses of DCs, because similar radio-
Keratocystic odontogenic tumor (KCOT), formerly graphic pictures can be found in other odontogenic
known as odontogenic keratocyst and primordial cyst, lesions such as KCOT. The radiographic aspect
is classified into the group of developmental odonto- of the KCOT, when involving the crown of an
genic cysts. KCOT is a locally aggressive benign enclosed tooth, can simulate a dentigerous cyst.
odontogenic lesion with a relatively high recurrence Therefore, the final pathologic diagnosis is based
potential. The other uncommon developmental on microscopic examination of biopsy or surgical
odontogenic cysts include glandular odontogenic excision specimens.

Correspondence: Jinrui Wang, School of Stomatology, Jilin University, 1500 Qinghua Road, Changchun 130021, China. E-mail: linannan1734@126.com; or
Wei Liu, Shanghai Key Laboratory of Stomatology, 639 Zhizaoju Road, Shanghai 200011, China. liuweb@hotmail.com
*These authors contributed equally to this work.

(Received 3 August 2013; accepted 15 March 2014)

ISSN 0001-6357 print/ISSN 1502-3850 online  2014 Informa Healthcare
DOI: 10.3109/00016357.2014.913192
 2 N. Li et al.

A review of the literature reveals that limited infor- logistic regression analysis of dentigerous cyst and
mation regarding pediatric patients hidden away in KCOT was performed to evaluate the parameters
several studies on developmental odontogenic cysts of that would favor KCOT over a dentigerous cyst.
all age-range patients were reported [5–23]. The issue This study was approved by the local institutional
of the prevalence and distribution of developmental review board.
odontogenic cysts has seldom been addressed in the A descriptive analysis was performed on demo-
Chinese population. Also, hardly any reports are graphic parameters. Statistical analysis was carried
available in the literature focusing on investigating out with the Chi-square test and Fisher’s exact test
the relationship between dentigerous cyst and KCOT among qualitative variables and the Student’s t-test
in children and adolescents. among quantitative variables. Logistic regression was
We, therefore, retrospectively reviewed a large applied to evaluate odds ratio (OR), indicative of
series of 369 patients with all histological diagnoses being KCOT over dentigerous cyst. OR with 95%
of developmental odontogenic cysts in children and confidence interval (CI) and p-values were reported.
adolescents from eastern China to investigate the All the tests were two-sided and p-values < 0.05 were
incidence and prevalence of developmental odonto- considered statistically significant.
genic cysts and compare the features of dentigerous
cyst and KCOT in the current hospital-based study
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over a 20-year period.

Results

Materials and methods Distribution of developmental odontogenic cysts

All the medical records of patients with the clinical Table I shows the diagnosis and the relative frequency
and pathologic diagnosis of developmental odonto- of developmental odontogenic cysts in pediatric
genic cyst from 1993–2012 in a computerized populations between the current study and selected
diagnostic index database of Ninth People’s Hospital, references from different countries.
Shanghai Jiao Tong University School of Medicine, A total of 369 patients in children and adolescents
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were retrieved and reviewed retrospectively. Records with developmental odontogenic cysts were identified
of children (£12 years) and adolescents (13–18 years) for this study during a 20-year period. Among these,
were selected. The tissue specimen of developmental 361 (97.8%) patients were diagnosed as dentigerous
odontogenic cyst was fixed in formalin, embedded in cyst (n = 281) and KCOT (n = 80). Figures 1 and 2
paraffin, and processed for routine histopathological show representative radiographic features of dentiger-
examination from hematoxylin-eosin staining sec- ous cyst and KCOT, respectively. Figure 3 shows
tions. The WHO criteria [1,2] were used when representative histological features of dentigerous cyst
examining the histopathology of the sections. The and KCOT.
patients with nevoid basal cell carcinoma syndrome In addition, six cases of glandular odontogenic cyst
were excluded in this study. If a patient had recurrent were males with an average age of 12.8 years
cysts, just the first diagnosis was included in the study. (range = 6–18), one case (a 1-year-old male) with
Information regarding patient’s age, gender and eruption cyst and one case (a 2-year-old female) with
location of lesion at the time of the initial diagnosis gingival cyst of infant were found; and no case of
were documented in detail. The comparative and lateral periodontal cyst was found in this study.

Table I. The relative frequency of developmental odontogenic cysts in pediatric populations between the current study and selected references
from different countries.

Country China, current study UK [6], 2006 Chile [7], 2007 Brazil [8], 2010 Turkey [9], 2012

Age range, years 1–18 1–16 0–15 0–16 2–17

Diagnosis (n, %)
Dentigerous cyst 281 (76.2) 160 (59.7) 240 (68.2) 96 (89.7) 128 (66.0)
KCOT 80 (21.7) 100 (37.3) 98 (27.8) 6 (5.6) 64 (33.0)
Glandular odontogenic cyst 6 (1.6) 0 3 (0.9) 0 0
Eruption cyst 1 (0.3) 8 (3.0) 10 (2.8) 3 (2.8) 1 (0.5)
Gingival cyst of infant 1 (0.3) 0 0 1 (0.9) 0
Lateral periodontal cyst 0 0 1 (0.3) 1 (0.9) 1 (0.5)
Total 369 268 352 107 194

KCOT, keratocystic odontogenic tumor.

 Pediatric developmental odontogenic cyst 3

A B

C D E

Figure 1. Representative radiographic features of a dentigerous cyst. Panoramic radiograph shows the dentigerous cysts in the (A) left maxilla
and (B) right mandible. (C) Three-dimensional and (D) axial section of computed tomographic scan. (E) Dental radiograph.
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Comparison of dentigerous cyst and KCOT To define the differences in demographic para-
meters between dentigerous cyst and KCOT, a
Table II shows the demographic features and location comparative analysis was performed. The average
of dentigerous cyst and KCOT. Among the 281 age of the patients with dentigerous cyst was 11.8 years
patients with a dentigerous cyst, there were 166 compared with those with KCOT at 14.7 years
children and 115 adolescents. These patients were (Student’s t-test, p < 0.001). Dentigerous cysts
187 males and 94 females (ratio M:F = 2:1). The were more common in children (59.1%) and KCOTs
maxilla (160 cases) was more commonly involved were more common (78.8%) in adolescents (Fisher’s
than the mandible (118 cases) by dentigerous cysts. exact test, p < 0.001). A significant difference in
Of the 80 patients with KCOT, there were 17 children
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location was also observed (Fisher’s exact test,

and 63 adolescents. These patients were 53 males and p = 0.010), whereas a difference in gender was not
27 females (ratio M:F = 2:1). The mandible (41 cases) observed between dentigerous cyst and KCOT.
was more commonly affected than the maxilla Considering that dentigerous cyst shares some
(27 cases) by KCOTs and there were nine cases of morphology similarity to KCOT, both clinically
KCOT located at both the maxilla and mandible. and radiographically, distinguishing KCOT from
the cystic lesions has some difficulties. To evaluate
the parameters that would favor KCOT over a denti-
gerous cyst, demographic parameters were analyzed
A by using the logistic regression model.
In univariate analysis, adolescence and location were
significantly indicative for the likelihood of KCOT
over dentigerous cysts. The likelihood of the patient
being adolescent was higher (OR = 5.35; 95%
CI = 2.98–9.61; p < 0.001) than being a child. The
likelihood of lesion located on the mandible was higher
(OR = 2.06; 95% CI = 1.20–3.54; p = 0.009) than that
located on the maxilla. To further assess the influence
B C of each factor, we did multivariate analysis. Adoles-
cence and location retained statistical significance.
The likelihood of the adolescent patient was higher
than the child (adjusted OR = 5.31; 95% CI = 2.83–
9.96; p < 0.001). The likelihood of the lesion being
located on the mandible was higher than it being
located on the maxilla (adjusted OR = 2.18; 95%
CI = 1.24–3.85; p = 0.007).

Figure 2. Representative radiographic features of keratocystic Discussion

odontogenic tumor (KCOT). (A) Panoramic radiograph shows a
KCOT in the right mandible. Computed tomographic scan shows Developmental odontogenic cysts are probably
KCOTs in the right (B) mandible and (C) maxilla. related to the fact that during the pediatric age period
 4 N. Li et al.

A B

x40 x200
C D
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x100 x200

Figure 3. Representative histological features. (A) Dentigerous cyst shows the cystic lesion encloses a dental crown. (B) Dentigerous cyst
shows cystic lining with thin non-keratinized stratified squamous epithelium. (C) Keratocystic odontogenic tumor (KCOT) shows a thin
regular parakeratinized stratified squamous epithelium with basal palisading. (D) Daughter cysts and epithelial budding formation are also
frequently observed in KCOT.

the jaws are involved in profound developmental cyst and KCOT and their more common location of
processes. These include growth of the maxillofacial presentation and age distribution will help practi-
skeleton and development of the primary and perma- tioners to determine a more likely correct clinical
nent dentition, all of which can be associated with cyst early diagnosis. The current study attempts to
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formation. Dentigerous cyst and KCOT were the two perform a descriptive analysis of 369 cases of devel-
most common types, accounting for over 95% of opmental odontogenic cysts and to assess the rela-
developmental odontogenic cysts in pediatric patients tionship between dentigerous cyst and KCOT in a
[5–9]. large series of dentigerous cysts (n = 281) and KCOT
The correct diagnosis of dentigerous cyst and (n = 80) in children and adolescents from eastern
KCOT is necessary and important for correct surgical China during the same 20-year period.
treatment and the institution of adequate follow-up. We found that dentigerous cysts and KCOT were
Therefore, knowledge of the incidence of dentigerous the two most frequent developmental odontogenic

Table II. Comparison of dentigerous cyst and keratocystic odontogenic tumor (KCOT).

Univariate LRA Multivariate LRA

Patients Dentigerous cyst (n = 281) KCOT (n = 80) p-value OR (95% CI) p-value OR (95% CI) p-value

Age (years)
Mean (SD) 11.8 (3.3) 14.7 (2.6) <0.001
Range 1–18 10–18
Age group <0.001
Children 166 (59.1) 17 (21.2) 1.00 (ref) 1.00 (ref)
Adolescents 115 (40.9) 63 (78.8) 5.35 (2.98–9.61) <0.001 5.31 (2.83–9.96) <0.001
Gender 1.00
Female 94 (33.5) 27 (33.8) 1.00 (ref)
Male 187 (66.5) 53 (66.2) 0.99 (0.58–1.67) 0.960
Location* 0.010
Maxilla 160 (57.6) 27 (39.7) 1.00 (ref) 1.00 (ref)
Mandible 118 (42.4) 41 (60.3) 2.06 (1.20–3.54) 0.009 2.18 (1.24–3.85) 0.007
Unknown 3 3

LRA, logistic regression analysis; OR, odds ratios; CI, confidence interval.
*There were nine cases of KCOT located at both maxilla and mandible.
 Pediatric developmental odontogenic cyst 5

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