Vol. 124 No.

2 August 2017

Cystic lesions of the jaws: a retrospective clinicopathologic

study of 2030 cases
Lorenzo Lo Muzio, MD, DDS, PhD,a Marco Mascitti, DDS,b Andrea Santarelli, DDS,b Corrado Rubini, MD,c
Fabrizio Bambini, MD, DDS,b Maurizio Procaccini, MD, DDS,b Dario Bertossi, MD,d Massimo Albanese, MD,d
Vincenzo Bondì, MD,d and Pier Francesco Nocini, MDd

Objective. The aim of this study was to perform an epidemiologic analysis of cases of jaw cysts treated from 1973 to 2012 at
the Dentistry and Maxillofacial Surgery Unit of the Verona Hospital, Italy, and to compare the data obtained with those
published in the literature.
Study Design. A retrospective survey of 2030 patients treated for jaw cysts from 1973 to 2012 was performed. The lesions
were classified according to the 2005 World Health Organization histologic classification, and the following variables were
analyzed: age, gender, histopathologic diagnosis, and site of onset.
Results. Of 2030 total lesions, there were 1970 odontogenic cysts (97.04%), 50 nonodontogenic cysts (2.46%), and 10
pseudocysts (0.49%). Of the patients, 314 were children (15.47%), and 1716 were adults (84.53%). Mean age was
37.24 years, with a male/female ratio of 1.71:1.
Conclusions. There is a wide variety of cysts, some of which are subject to variations according to gender, localization, and
age. (Oral Surg Oral Med Oral Pathol Oral Radiol 2017;124:128-138)

Jaw cysts are characterized by a pathologic cavity, the epidemiology of these lesions as well as that of
either completely or partially covered by an epithelial odontogenic tumors.8
tissue, and are not caused by accumulation of pus.1 One of the main issues in dental and oral maxillo-
These lesions are classified according to 3 criteria: (1) facial surgical practice is the differential diagnosis of
the presence or absence of lining epithelium (cyst or cystic lesions because few odontogenic cysts (OCs)
pseudocyst); (2) the pathogenic mechanism that exhibit aggressive behavior and the tendency to recur.
causes the formation of cysts (developmental or Furthermore, it is important to perform a differential
inflammatory origin); and (3) the tissues (odontogenic diagnosis of cysts and tumors of the jaw to determine
or nonodontogenic) involved in cyst formation.2 The the most appropriate surgical treatment and follow-up.
importance of this type of lesions arises from the fact The diagnosis of a cyst is based on clinical and radio-
that cysts are the most common cause of chronic logic features, although the final diagnosis depends on
swelling in the jaw with respect to the remaining histopathology.9 Many authors have reported on the
skeletal system.3 jaw cyst; however, very few have presented detailed
During the twentieth century, several classification epidemiologic data which are often based on local
systems of jaw cysts were proposed, and in the 1992 geographic backgrounds.10
edition of World Health Organization (WHO) classifi- The aim of this retrospective study was to perform a
cation “Histologic Typing of Odontogenic Tumors,” comprehensive epidemiologic analysis of all cystic
jaw cysts were extensively described.4 In view of its neoformations of jaws, diagnosed and surgically treated
biological behavior, odontogenic keratocyst (OKC) in the Dentistry and Maxillofacial surgery Unit of
was reclassified in 2005 by the WHO as a neoplastic “Policlinico G. B. Rossi” Hospital in Verona, Italy,
lesion and named keratocystic odontogenic tumor from 1973 to 2012. In this investigation, we focused on
(KCOT).5-7 One of the main effects of this new clas- the analysis of the most frequent cystic lesions: radic-
sification of cysts is the change in the understanding of ular cysts, dentigerous cysts, and residual cysts. In
addition, the collected data were compared with those
a
Department of Clinical and Experimental Medicine, Foggia Uni- of previously published works to provide a better
versity, Foggia, Italy. epidemiologic representation of this kind of lesions.
b
Department of Clinical Specialistic and Dental Sciences, Marche
Polytechnic University, Ancona, Italy.
c
Department of Biomedical Sciences and Public Health, Marche
Polytechnic University, Ancona, Italy.
d
Clinic of Dentistry and Maxillofacial Surgery, Policlinico G. B.
Statement of Clinical Relevance
Rossi, University of Verona, Verona, Italy.
Received for publication Feb 12, 2017; returned for revision Mar 14, This retrospective clinicopathologic study analyzed
2017; accepted for publication Apr 6, 2017. a large number of cystic neoformations of the jaws
Ó 2017 Elsevier Inc. All rights reserved. diagnosed and surgically treated in Verona, Italy,
2212-4403/$ - see front matter over a considerable period (1973-2012).
http://dx.doi.org/10.1016/j.oooo.2017.04.006

128
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Volume 124, Number 2 Lo Muzio et al. 129

We used the 2005 WHO classification, which con- gender, age, and localization. OCs account for most
siders OKC as a tumor (now KCOT), and because of the cysts of the jaws, with 1970 cases (97.04%)
many studies reported in the literature used the 1992 and an M/F ratio of 1.69:1 (P < .001). Mean age was
WHO classification, we compared our results with 37.72 years (range 2-92 years). Considering the age at
those previously published and considered the differ- onset, dentigerous cysts affected younger patients
ences between them. more than radicular cysts did, whereas the age at
onset of radicular cysts was significantly lower than
MATERIALS AND METHODS that of residual cysts (P < .001). These lesions were
The present retrospective study considered 2030 pa- more common in the mandible (1124 cases [57.06%])
tients who underwent surgery for jaw cysts from 1973 than in the maxilla (846 cases [42.94%]; P < .001).
to 2012. Data were retrieved and cataloged from clin- Fifty cases of nonodontogenic cysts were reported,
ical records and from the archive of the Institute of accounting for 2.46% of all jaw cysts, with a higher
Pathology by a single operator to ensure uniformity of frequency in males (M/F ratio 5.25:1) and a mean age
the collected data. Because of the 40-year period of 30.32 years. With regard to the site of onset,
considered in this study, histopathologic slides of jaw because of their nature, these cysts are obviously
cysts were re-evaluated by 2 pathologists to confirm the localized in the maxilla. Finally, there were 10 cases
diagnosis, according to the current WHO histopatho- of pseudocysts (0.49%), with an M/F ratio of 0.25:1,
logic criteria.4,5 In case of controversy, a third pathol- mean age of 20 years, and greater occurrence in the
ogist was consulted to ensure that the final diagnosis mandible.
was correct. Tables II and III present data on pediatric and adult
In the present study, the following information was presentations of jaw cysts, respectively. Among
obtained from each case: age, gender, histopathologic pediatric patients, the mean age was 12.56 years (SD
diagnosis, site distribution, and relapses. The regions of 3.54), whereas jaw cysts occurred in adult patients at
onset (mandible and maxilla) were further divided into a mean age of 41.99 years (SD 15.77). In addition, in
3 regions (anterior, premolar, and molar regions). pediatric patients, the age at onset differed between
Furthermore, these data were analyzed considering radicular and dentigerous cysts. In adult patients, the
pediatric (ages 16 years) and adult populations (ages age at onset of residual cysts was significantly higher
17 years), separately. than that of dentigerous and radicular cysts (P < .001).
Data analysis was performed using GraphPad Prism The distribution of cysts between the genders was
software version 6.00 for Windows (GraphPad Soft- found to be more homogeneous in pediatric patients
ware, San Diego, CA; www.graphpad.com). Chi-square than in adults (M/F ratio of 1.24:1 vs 1.82:1), reaching
test and Fisher’s exact test were used for grouped var- statistical significance (P < .01).
iables. One-way analysis of variance (ANOVA) and the Radicular cysts were the most frequently diagnosed,
Bonferroni post hoc test were used for continued vari- with 865 cases (42.61% of all cysts; 43.91% of OCs)
ables, and the Kruskal-Wallis and Dunn post hoc tests and an M/F ratio of 1.77:1 (Table I; P < .001). This
were used for grouped variables. The level of signifi- cyst represents the most common lesion in adults
cance was set at P < .05. This study was conducted in (43.82% of all cysts; 44.71% of OCs), whereas in
accordance with the tenets of the Helsinki Declaration. pediatric patients, radicular cysts were less frequent
(35.99% of all cysts; 39.24% of OCs), but without
RESULTS reaching statistical significance (Tables II and III).
In total, 2030 patients were treated from 1973 to 2012 With regard to the site of onset, radicular cysts were
for cystic neoformations of the jaws, and the cases were significantly more frequent in the maxilla (P < .001),
recorded in the Dentistry and Maxillofacial Surgery with 568 cases localized in this region (65.66% of
Unit of “Policlinico G. B. Rossi” Hospital in Verona, total), especially in the anterior region (402 cases);
Italy. Of these patients, 314 (15.47%) were <17 years 297 cases were found in the mandible (34.34% of
of age, and 1716 (84.53%) ranged from the ages 17 to total), equally distributed among the anterior,
92 years. Mean age was 37.24 years (range 1-92 years; premolar, and molar regions (Table IV). Mean age
SD 18.04). Among all cases, 1281 (63.10%) were was 38.17 years (range 7-92 years; SD 16.44). With
males and 749 (36.90%) were females, with a male/ regard to gender distribution, males showed a peak
female (M/F) ratio of 1.71:1 (P < .001). With regard to frequency in fourth and fifth decades, whereas the
localization, the mandible (1134 cases, 55.86%) was female population presented a more homogeneous
more commonly involved than the maxilla was (896 distribution (Figure 1).
cases [44.14%]; P < .001). The second most frequent lesion was the dentigerous
Table I shows the distribution of OCs, cyst, amounting to 806 cases (39.70% of all cysts;
nonodontogenic cysts, and pseudocysts, in relation to 40.91% of OCs) and an M/F ratio of 1.53:1 (Table I)
ORAL AND MAXILLOFACIAL SURGERY OOOO
130 Lo Muzio et al. August 2017

Table I. Jaw cysts distribution (1973-2012)

Frequency Male Female Age Localization
Lesion No. (%) % total No. (%) No. (%) M/F ratio Mean (range) Mandible Maxilla
Odontogenic cyst
Radicular cyst 865 (43.91) 42.61 553 (63.9) 312 (36.1) 1.77:1 38.17 (10-92) 297 568
Dentigerous cyst 806 (40.91) 39.70 488 (60.5) 318 (39.5) 1.53:1 34.80 (2-81) 649 157
Residual cyst 218 (11.07) 10.74 142 (65.1) 76 (34.9) 1.87:1 48.27 (10-90) 118 100
Paradental cyst 20 (1.01) 0.99 12 (60) 8 (40) 1.5:1 28.90 (8-58) 18 2
Lateral periodontal cyst 14 (0.71) 0.69 12 (85.7) 2 (14.3) 6:1 57.14 (15-78) 10 4
Glandular odontogenic cyst 14 (0.71) 0.69 6 (42.9) 8 (57.1) 0.75:1 33.86 (13-55) 10 4
Eruptive cyst 12 (0.61) 0.59 8 (66.7) 4 (33.3) 2:1 12.67 (6-19) 7 5
Gingival cyst of adult 12 (0.61) 0.59 10 (83.3) 2 (16.7) 5:1 32.67 (23-43) 9 3
Orthokeratized 9 (0.46) 0.44 6 (66.7) 3 (33.3) 2:1 35.56 (20-60) 6 3
odontogenic cyst
1970 (100) 97.04 1237 (62.8) 733 (37.2) 1.69:1 37.72 (2-92) 1124 846
Nonodontogenic cyst
Nasopalatine cyst 38 (76) 1.87 32 (84.2) 6 (15.8) 5.33:1 36.42 (9-78) 0 38
Nasolabial cyst 6 (12) 0.30 4 (66.7) 2 (33.3) 2:1 19 (15-21) 0 6
Epstein pearls 6 (12) 0.30 6 (100) 0 (0) N 3 (1-5) 0 6
50 (100) 2.46 42 (84) 8 (16) 5.25:1 30.32 (1-78) 0 50
Pseudocyst
Aneurismatic cyst 6 (60) 0.30 0 (0) 6 (100) 0 17.67 (12-25) 4 2
Solitary bone cyst 4 (40) 0.20 2 (50) 2 (50) 1:1 23.50 (8-39) 4 0
10 (100) 0.49 2 (20) 8 (80) 0.25:1 20 (8-39) 8 2
Total 2030 100 1281 (63.1) 749 (36.9) 1.71:1 37.24 (1-92) 1132 898

(P < .001). Dentigerous cysts occurred relatively more Furthermore, male patients showed a peak frequency
in childhood (48.41% of all cysts; 52.78% of OCs) than in the sixth decade, whereas females presented a first
in adulthood (38.11% of all cysts; 38.88% of OC), peak frequency in the fourth and fifth decades and a
reaching statistical significance (Tables II and III; second peak in the seventh decade (Figure 1).
P < .001). With regard to the site of onset, All other OCsdnamely, eruptive cyst, gingival cyst
dentigerous cysts occurred mainly in the mandible, of the adult, lateral periodontal cyst, glandular OC,
with 609 cases (83.82% of total; P < .001). This cyst paradental cyst, and orthokeratinized OC (OOC)d
predominantly affected the posterior region of the showed a very low frequency, reaching a total of 81
mandible (544 cases), whereas in the maxilla (157 cases (4.11% of all cysts; 3.99% of OCs). More detailed
cases; 16.18% of total), the anterior region was the information is presented in Tables I to III.
most commonly affected site (Table IV) (P < .05). Among nonodontogenic cysts, the most diagnosed
Mean age was 34.80 years (range 2-81 years; SD was the nasopalatine cyst, with 38 cases (1.87% of all
18.21). With regard to gender distribution, females cysts; 76.0% of nonodontogenic cysts). These lesions
showed a peak in frequency in the second and third showed a greater frequency in males, with an M/F ratio
decades, whereas males presented 2 distinct peaks in of 5.33:1 (P < .01). Other nonodontogenic cysts were
frequency (third and fifth decades) (Figure 1). Epstein pearls and nasolabial cysts, each type with 6
Residual cysts represented the third most common cases (0.30% of all cysts; 12.0% of nonodontogenic
cyst, with 218 cases (10.74% of all cysts; 11.07% of cysts).
OCs), and an M/F ratio of 1.87:1 (Table I; P < .01). With regard to pseudocysts, there were 6 cases of
These cysts almost exclusively affected adult patients aneurysmal bone cyst (0.30% of all cysts), with the
(12.30% of all cysts; 12.54% of OCs), whereas in mean age of patients being 17.67 years (SD 5.96); these
pediatric patients, they were infrequent (P < .001; cysts occurred only in female patients. Solitary bone
Tables II and III). Both the mandible and the maxilla cysts occurred in 4 patients (0.20% of all cysts), and the
were seen to be equally involved, with 118 and 100 mean age at onset was 23.50 years (SD of 17.90).
cases, respectively. With regard to the sites of onset,
residual cysts were more frequently localized in the DISCUSSION
posterior mandible (68 cases) and the anterior maxilla Jaw cysts are the most common bone cysts and one of
(62 cases; Table IV; P < .001). Mean age was the most frequent lesions affecting the jaws. Most of
48.27 years (range 10-90 years; SD 17.90). The age these cysts are OCs that originate from odontogenic
at onset was higher with respect to other groups epithelial remnants in oral tissues. Jaw cysts share
because 211 of 218 cases occurred in adult patients. similar features; therefore, the differential diagnosis
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Volume 124, Number 2 Lo Muzio et al. 131

Table II. Jaw cysts distribution in childhood (1973-2012)

Frequency Male Female Age
Lesion No. (%) % total No. (%) No. (%) M/F ratio Mean (range)
Odontogenic cyst
Dentigerous cyst 152 (52.8) 48.4 78 (51.3) 74 (48.7) 1.1:1 11.8 (2-16)
Radicular cyst 113 (39.2) 35.9 65 (57.5) 48 (42.5) 1.3:1 14.4 (7-16)
Eruptive cyst 8 (2.8) 2.5 8 (100) 0 (0) N 9.7 (6-15)
Residual cyst 7 (2.4) 2.2 3 (42.9) 4 (57.1) 0.7:1 13.7 (10-16)
Paradental cyst 4 (1.4) 1.3 2 (50) 2 (50) 1:1 11 (8-14)
Lateral periodontal cyst 2 (0.7) 0.6 0 (0) 2 (100) 0 15 (15)
Glandular odontogenic cyst 2 (0.7) 0.6 0 (0) 2 (100) 0 13 (13)
288 (100) 91.7 156 (54.2) 132 (45.8) 1.2:1 12.8 (2-16)
Non-odontogenic cyst
Nasopalatine cyst 12 (60.0) 3.8 10 (83.3) 2 (16.7) 5:1 11.5 (9-16)
Nasolabial cyst 2 (10.0) 0.6 2 (100) 0 (0) N 15 (15)
Epstein pearls 6 (30.0) 1.9 6 (100) 0 (0) N 3 (1-5)
20 (100) 6.4 18 (90) 2 (10) 9:1 9.32 (1-16)
Pseudocyst
Aneurismatic cyst 4 (66.67) 1.3 0 (0) 4 (100) 0 14 (12-16)
Solitary bone cyst 2 (33.33) 0.6 0 (0) 2 (100) 0 8 (8)
6 (100) 1.9 0 (0) 6 (100) 0 12 (8-16)
Total 314 100 174 (55.4) 140 (44.6) 1.2:1 12.6 (1-16)

Table III. Jaw cysts distribution in adulthood (1973-2012)

Frequency Male Female Age
Lesion No. (%) % total No. (%) No. (%) M/F ratio Mean (range)
Odontogenic cyst
Radicular cyst 752 (44.7) 43.8 488 (64.9) 264 (35.1) 1.8:1 41.7 (17-92)
Dentigerous cyst 654 (38.9) 38.1 410 (62.7) 244 (37.3) 1.7:1 40.1 (17-81)
Residual cyst 211 (12.6) 12.3 139 (65.9) 72 (34.1) 1.9:1 49.4 (17-90)
Paradental cyst 16 (0.9) 0.9 10 (62.5) 6 (37.5) 1.7:1 33.4 (17-58)
Lateral periodontal cyst 12 (0.7) 0.7 12 (100) 0 (0) N 64.2 (22-78)
Glandular odontogenic cyst 12 (0.7) 0.7 6 (50) 6 (50) 1:1 37.3 (24-55)
Gingival cyst of adult 12 (0.7) 0.7 10 (83.3) 2 (16.7) 5:1 32.7 (23-43)
Orthokeratized odontogenic cyst 9 (0.5) 0.5 6 (66.7) 3 (33.3) 2:1 35.6 (20-60)
Eruptive cyst 4 (0.2) 0.2 0 (0) 4 (100) 0 18.5 (17-19)
1682 (100) 98.0 1081 (64.3) 601 (35.7) 1.8:1 42.0 (17-92)
Nonodontogenic cyst
Nasopalatine cyst 26 (86.7) 1.5 22 (84.6) 4 (15.4) 5.5:1 47.9 (22-78)
Nasolabial cyst 4 (13.3) 0.2 2 (50) 2 (50) 1:1 21 (21)
30 (100) 1.7 24 (80) 6 (20) 4:1 44.3 (21-78)
Pseudocyst
Aneurismatic cyst 2 (50) 0.1 0 (0) 2 (100) 0 25 (25)
Solitary bone cyst 2 (50) 0.1 2 (100) 0 (0) N 39 (39)
4 (100) 0.2 2 (50) 2 (50) 1:1 32 (25-39)
Total 1716 100 1107 (64.5) 609 (35.5) 1.8:1 42.0 (18-92)

requires clinical and radiographic analyses, in combi- give distorted results.12 In addition, comparison with
nation with histopathologic findings.3,11 This retro- other studies would be difficult, given that several
spective study focused on evaluation of the relative studies have evaluated data from oral pathology
frequency of all cystic neoformations diagnosed and services, which receive numerous specimens from
surgically treated over a 40-year period. public health structures and private dental
We have not included data about the frequency of practitioners.13
jaw cysts in comparison with that of all oral pathologies Another aspect to be taken into account is the
treated in this Dentistry and Maxillofacial Surgery Unit redefinition of OKC as a tumor (now KCOT), which led
over the 40-year period. The reason is that the analysis us to consider only its orthokeratinized variant (OOC)
of data from a single institution in this study and the as a cyst.5 Therefore, comparison of the results of this
extension of these results to the entire population would study with those reported in the literature excluded
ORAL AND MAXILLOFACIAL SURGERY OOOO
132 Lo Muzio et al. August 2017

Table IV. Site distribution of dentigerous, radicular, and residual cysts

Mandible Maxilla
Lesion Anterior Premolar Posterior Total Anterior Premolar Posterior Total
Dentigerous cyst 34 71 544 649 84 34 39 157
Radicular cyst 126 83 88 297 402 71 95 568
Residual cyst 28 22 68 118 62 25 13 100

data about OKCs; however, OOCs were included in the 2 authors found a substantial equality between the cases
analysis only when explicitly mentioned. in the mandible and those in the maxilla.14,24 Finally,
With regard to all of the cysts considered, our results Meningaud et al.18 reported a higher percentage of
showed mean age of 37.24 years, with 314 cases mandibular cysts in comparison with that found in our
occurring in childhood and 1716 in adulthood, a male study (Table V; P < .05).
predominance (M/F ratio of 1.71:1), and localization Radicular cysts develop as consequence of pulpitis and
slightly more frequent in the maxilla. These results pulpal necrosis. These lesions arise from proliferation of
depend largely on the data on OCs, which represent the epithelial rests of Malassez, induced by periapical
97.04% of all cystic lesions. inflammatory response.27 In this study, radicular cysts
When the age at onset was considered, OCs showed accounted for 43.91% of all OCs, representing the most
mean age of 37.72 years, considerably higher than that common lesion (Table I). Although this result is in
found by Sharifian et al.14 (28.60 years), Ramachandra agreement with all of the studies reported in the
et al.15 (30.29 years), de Souza et al.16 (31 years), and literature, a higher proportion of these cysts have been
Tortorici et al.17 (35.1 years) but significantly lower observed by other authors3,10,15-27 (P < .05). In fact,
than the mean age reported by Meningaud et al.18 only Ledesma-Montes et al.,9 Johnson et al.,12 and
(41.8 years) and Nùñez-Urrutia et al.19 (42 years) Sharifian et al.14 found a similar frequency of radicular
(P < .05). Furthermore, other authors such as cysts (Table VII). There are several explanations for
Selvamani et al.20 (28 years), Avelar et al.21 these results: First of all, some authors analyzed
(28.9 years), Demirkol et al.22 (32.70 years), specimens from oral pathology services, which had
Varinauskas et al.23 (35.8 years), and Tekkesin received samples from biopsy procedures performed by
et al.3 (36.33 years) reported a lower mean age at general dental practitioners. This may have increased
onset; a higher mean age was reported by Johnson the number of simple lesions, such as radicular cysts,
et al.12 (43.40 years), but we could not demonstrate and reduced the number of more complex cysts, which
a statistically significant difference because of lack were more easily treated by oral and maxillofacial
of data (Table V). surgeons.27 Another reason is that several studies have
The analysis of gender distribution of OCs showed a been conducted in countries that have a high incidence
male predominance, with an M/F ratio of 1.69:1. These of caries. Caries increase the risk of pulp necrosis and
results are in agreement with those reported by Menin- chronic periapical inflammation, which are related to the
gaud et al.,18 Nùñez-Urrutia et al.,19 Ramachandra et al.,15 onset of radicular cysts.9,24 Finally, as suggested by
and Demirkol et al.22 However, many other studies found other authors, socioeconomic conditions may influence
a more balanced M/F ratio,3,9,10,12,14,17,20,23-26 but Prockt the relative frequency of radicular cysts.16,26 However,
et al.27 and de Souza et al.16 reported a female some pathology laboratories have reported a similar
predominance (Table V; P < .05). The preponderance relative frequency of radicular cysts; the possible expla-
of male patients encountered in the present study was nation is that many clinicians do not perform biopsy
emphasized in the adult population (M/F ratio of procedures and arrive at their diagnosis only on the basis
1.82:1). In fact, when considering only pediatric of clinical and radiologic data.14
patients, there was a slight majority of males (M/F ratio When we considered only adult patients, the fre-
of 1.24:1), which did not differ significantly from other quency of radicular cysts was significantly lower
studies3,16,25,28,29 (Table VI). compared with other studies3,16,25,30,31 (Table VI;
With regard to the anatomic localization of the le- P < .05). With regard to the pediatric population, our
sions, the mandible was the site most often affected by results are in agreement with several studies in which
OCs (1124 cases [57.06%]). These results were radicular cysts were found to be the second most
consistent with those reported by Avelar et al.,21 Nùñez- common lesions (after dentigerous cysts),16,25,29
Urrutia et al.,19 and Johnson et al.12 In contrast, several whereas, other studies showed a preponderance of
studies reported a higher frequency of OCs in the radicular cysts3,28 (Table VI) (P < .05). As pointed out
maxilla (range 52.39%-64.05%),3,15,16,20,22,23,25,27 and by Jones et al., the reason for this difference may be
OOOO ORIGINAL ARTICLE
Volume 124, Number 2 Lo Muzio et al. 133

Fig. 1. Distribution of radicular, dentigerous, and residual cysts according to age.

related to the prevalence of caries and the differences in (65.66%), in agreement with almost all other
oral health status among the populations of various studies14-16,20,22,24,27 Some authors reported a more
countries.28 pronounced difference in the localization of radicular
In this study, the mean age at diagnosis for radicular cysts,10,26 whereas in other studies, radicular cysts in
cysts was 38.17 years, with a frequency peak in the the maxillary bone were only slightly more
fourth and fifth decades of life, more evidently in male frequent12,17 (P < .05). Finally, only Meningaud et al.
patients (Figure 1). These results agree with those of showed a higher percentage of radicular cysts in the
Jones et al.,10 but other studies showed a significantly mandible18 (Table VII; P < .05). The site distribution
lower age at onset, with frequency peaks between the of radicular cysts showed a greater involvement of the
second and fourth decades of life14-17 (P < .05). In anterior maxilla (46.47% of the total; Table IV;
contrast, Meningaud et al. reported a higher mean age18 P < .05), confirming what is reported in the
(Table VII; P < .05). These results may depend on the literature.10,24,25,27 Several authors have suggested that
differences in the rate of prevalence of caries among the reason for the greater involvement of the maxilla
countries. Age at diagnosis of radicular cysts was could be related to aesthetic factors. In fact, patients
greater than that of dentigerous cyst (38.17 years vs may wish to conserve their teeth, even if this means
34.80 years), and the same result was also found in poor endodontic treatment leading to chronic periapical
the pediatric group (14.37 years vs 11.76 years; inflammation and increasing the risk of developing
Tables I and II; P < .05). A possible explanation is radicular cysts.22
that some lesions, such as radicular cyst, may not be The dentigerous cyst is a developmental cystic lesion
submitted to pathology laboratories, especially if that surrounds the crown of an unerupted tooth, and the
associated with deciduous teeth.29 cyst wall is attached to its neck.33 In this study, the
Most radicular cysts occurred in male patients (M/F dentigerous cyst was found to be the second most
ratio of 1.77:1), as confirmed by Meningaud et al.18 and frequent lesion, accounting for 40.91% of all OCs
Ramachandra et al.15 The male preponderance seen (Table I). Although the dentigerous cyst appears to be
among patients with radicular cysts was significantly second most common OC in all other studies, in our
attenuated in other studies,3,10,12,14,17,21,22,24 but some analysis, this lesion was particularly frequent. This
authors reported a female preponderance for these le- result is in agreement with the findings of Ledesma-
sions9,16,20,25-27 (Table VII). The male preponderance is Montes et al.9 and Mosqueda-Taylor et al.,26 and the
explained by the fact that males generally have poorer relative frequency of dentigerous cyst was
oral health and their oral structures are exposed to significantly greater than that reported by most
trauma more frequently.16 In contrast, the greater authors3,10,12,14-25,27 (Table VII; P < .05). This
frequency of radicular cysts in females found in some difference can be explained by the lower frequency of
studies has been explained as consequence of females radicular cysts found in our study, which indirectly
seeking dental care more frequently.32 influences the percentage of dentigerous cysts. When
Among OCs, radicular cysts were the only lesions only pediatric patients were considered, the
that showed a preference for maxillary localization dentigerous cyst was the most frequent lesion,
ORAL AND MAXILLOFACIAL SURGERY OOOO
134 Lo Muzio et al. August 2017

Table V. Distribution of odontogenic cysts and comparison with other studies (without keratocystic odontogenic
tumors)
Author (year) Mean age M/F ratio Dentigerous cyst Radicular cyst Residual cyst Other cysts Maxilla Mandible
Ledesma-Montes et al. (2000)9 d 1.3:1 43.7 47.8 6.07 2.4 d d
Mosqueda-Taylor et al. (2002)26 d 1.1:1 42.1 50.9 2.8 4.2 d d
Jones et al. (2006)10 d 1.3:1 20.5 59.2 9.1 11.2 d d
Meningaud et al. (2006)18 41.8 1.9:1 27.4 66.2 5.7 0.7 26.9 73.1
Varinauskas et al. (2006)23 35.8 1.1:1 13.8 76.9 9.3 d 63.1 36.9
Ochsenius et al. (2007)25 d 1.1:1 21.6 59.2 13.0 6.1 64.0 35.9
Tortorici et al. (2008)17 35.1 1.2:1 11.9 88.1 d d 52.4 47.6
Prockt et al. (2008)27 0.9:1 22.2 72.5 4.3 1.0 56.2 43.8
Avelar et al. (2009)21 28.9 30.8 52.3 5.9 11.0 43.9 56.0
de Souza et al. (2010)16 31.0 0.8:1 21.5 65.6 5.2 7.6 56.6 43.4
Nùñez-Urrutia et al. (2010)19 42.0 1.4:1 22.0 50.7 4.3 22.9 38.9 61.1
Ramachandra et al. (2011)15 30.3 1.7:1 30.8 69.2 d d 58.9 41.1
Sharifian et al. (2011)14 28.6 1.4:1 30.7 47.1 10.0 12.2 49.6 50.4
Açikgöz et al. (2012)24 d 1.2:1 27.9 57.4 14.4 0.2 49.4 50.6
Tekkesin et al. (2012)3 36.3 1.3:1 13.6 72.1 12.4 1.9 52.4 47.6
Selvamani et al. (2012)20 28 1.2:1 20.3 69.3 3.3 7.2 57.5 42.5
Johnson et al. (2013)12 43.4 1.2:1 22.4 45.7 d 31.9 38.9 61.1
Demirkol et al. (2014)22 32.7 1.4:1 28.6 67.1 3.6 0.7 52.9 47.1
Present study 37.7 1.7:1 40.9 43.9 11.1 4.1 42.8 57.2

confirming the results of previous studies.16,25,29 In male preponderance was greater in some studies,
contrast, Jones et al.28 and Tekkesin et al.3 reported a reaching statistical significance,10,18,25 but Prockt
lower relative frequency (Table VI; P < .05). As et al.27 reported a slightly greater preponderance of
previously reported, the prevalence of caries may females for these lesions (Table VII; P < .05).
indirectly affect the relative frequency of dentigerous In our study, dentigerous cysts were found to occur
cysts by increasing the number of cases of radicular predominantly in the mandible (80.52%), in agreement
cysts.28 Indeed, current data on adult patients show with some authors.10,12,22 These results differ from
that the frequency of dentigerous cysts is much higher those of many studies reported in the literature, in
than that reported by other authors3,16,25,30,31 which the site predilection of dentigerous cyst was
(Table VI; P < .05). significantly attenuated3,14-17,20,24,25,27 (P < .05). In
The mean age at diagnosis of dentigerous cyst was contrast, only Meningaud et al.18 reported a more
34.80 years, with a frequency peak in the second and pronounced difference in dentigerous cyst localization
third decades of life, and male patients showed two (Table VII; P < .05). With regard to site distribution,
peaks in the third and fifth decades of life (Figure 1). dentigerous cysts showed a greater involvement in the
Results published in the literature are variable, with posterior mandible (67.49% of the total; Table IV;
some authors reporting significantly lower mean age P < .05), in agreement with the findings reported in
at onset14-17 and some others reporting a higher literature.10,24,27 As pointed out by other authors, den-
one10,18 (Table VII; P < .05). The reason for the tigerous cysts occur much more frequently in the pos-
dentigerous cyst being diagnosed at a later age may terior mandible because the lower third molars are the
be the slow and asymptomatic growth of these cysts, most commonly impacted teeth.27
which can lead to delay in surgical treatment.22 In the case of residual cysts, these lesions are simply
Nevertheless, radicular cysts are diagnosed later radicular cysts that remain in the jaws after the causal
compared with dentigerous cysts, and this difference tooth has been extracted. Consequently, the radio-
in age at diagnosis is statistically significant (Tables I graphic and clinical characteristics of residual cysts are
and II; P < .05). The possible explanation is that the same as those of radicular cysts; the only difference
dentigerous cysts are more commonly associated with is that residual cysts are associated with an edentulous
impacted teeth, which are more frequently diagnosed area.2 In this study, residual cysts were the third most
in the first 3 decades of life. common lesions, accounting for 11.07% of all OCs
With regard to gender distribution of dentigerous (Table I). This result is in agreement with those of
cysts, variable results have been reported in the litera- some studies,3,14,24,25 but in some other studies, the
ture. We found that the dentigerous cyst was more relative frequency of residual cysts was significantly
common in male patients (M/F ratio of 1.53:1), in lower9,10,16,18-23,26,27 (Table VII; P < .05).
agreement with several authors.3,9,12,14-17,21,22,24,26 This Furthermore, it must be noted that some authors
OOOO ORIGINAL ARTICLE
Volume 124, Number 2 Lo Muzio et al. 135

Table VI. Distribution of odontogenic cysts in children and adults, and comparison with other studies (without
keratocystic odontogenic tumors)
Children Adults
Author (year) M/F ratio Dentigerous cyst Radicular cyst Residual cyst M/F ratio Dentigerous cyst Radicular cyst Residual cyst
Jones et al. (2006)10 1.3:1 35.4 53.7 0.7 1.3:1 19.8 59.1 10.3
Ochsenius et al. (2007)25 1.2:1 58.4 35.8 0.5 1.1:1 14.5 63.8 15.4
de Souza et al. (2010)16 1.3:1 51.3 45.4 0.5 0.7:1 14.4 73.8 6.5
Tekkesin et al. (2012)3 d 24.5 70.9 3.3 d 11.9 72.2 13.8
Ha et al.29 & Kelloway 1.2:1 56.2 31.0 d 1:1 27.3 63.6 d
et al.31 (2014)
Present study 1.2:1 52.8 39.2 2.4 1.8:1 38.9 44.7 12.5
M/F, male/female.

included residual cysts within their results for radicular to be more common in the posterior mandible and the
cysts, and therefore, our results cannot be compared anterior maxilla (Table IV). There is no agreement in
with the findings of those studies.12,15,17 In pediatric the literature regarding a preferential anatomic site of
patients, this lesion is significantly less common, ac- onset. In fact, some authors reported a greater
counting for 2.43% of all OCs, in agreement with the involvement of the anterior maxilla,10,25 whereas
other studies.3,16,25,28 Radicular cysts were much more some others showed no predilection for any anatomic
common in adult patients, representing 12.54% of all site.19,27
OCs, in agreement with the findings of Tekkesin et al.3 Paradental cyst is an inflammatory cyst arising on the
In contrast, Jones et al.30 and de Souza et al.16 reported root surface of a vital tooth from epithelial remnants as
a lower relative frequency, and Ochsenius et al.25 found a consequence of inflammation associated with peri-
a higher frequency. coronitis.34,35 Our results showed a relative frequency
Mean age at diagnosis for residual cysts was of 1.01% of all OCs, similar to the results of Mosqueda-
48.27 years, with a higher frequency between the fourth Taylor et al.26 and de Souza et al.16 but significantly
and the seventh decades of life (Figure 1). Few results lower10,14,19,21,25 or higher frequency3,27 compared
have been reported in the literature, and some authors with that reported by other studies (P < .05). As sug-
have reported a lower mean age at onset,14,16 whereas gested by Jones et al.,10 the paradental cysts are
some other studies have reported a higher one10,18 probably more common than what is reported in the
(Table VII; P < .05). Age at diagnosis of residual literature because these cysts are commonly
cysts was greater than that of radicular cysts associated with the lower third molar with a history
(48.27 years vs 38.17 years), and this result was of recurrent or persistent pericoronitis. Therefore, it is
confirmed in the adult population as well (49.42 years likely that many paradental cysts are extracted with
vs 41.75 years; Tables I and III; P < .05). The the third molar and not subjected to histopathologic
possible explanation for this difference is that residual analysis. Mean age at diagnosis was 28.9 years, with
cysts usually remain confined within bone and are a peak frequency in the third decade, in agreement
thus asymptomatic and detected only years later with other authors10,14,16 (P < .05). The age distribu-
because of reinfection or incidental radiographic tion is explained by the fact that this lesion is frequently
finding.27 associated with the lower third molar with pericor-
Similar to radicular cysts, we found most residual onitis.36 This lesion was slightly more common in male
cysts occurring in male patients (M/F ratio of 1.87:1), patients (M/F ratio of 1.5:1); the male preponderance
which has been confirmed by almost all other was confirmed by the findings of many
studies,3,10,14,16,18,24,26,27 but Ochsenius et al.25 showed studies,3,10,21,25,26,36 whereas Sharifian et al.14 and de
a significantly attenuated difference in gender Souza et al.16 reported a greater involvement in
distribution, and Avelar et al.21 reported a female female patients. The most common affected site was
preponderance (Table VII; P < .05). the posterior mandible because of the nature of this
Our results revealed no significant difference in re- cyst, and this was found to be in agreement with the
sidual cyst distribution but showed a slightly greater findings of other studies.10,19,21,36
involvement of the mandible. Our results differ signif- Glandular OC, described for the first time by
icantly from those of Ochsenius et al.25 and Tekkesin Padayachee and Van Wyk and classified as an inde-
et al.,3 according to which the maxilla is the most pendent pathologic entity by the WHO in 1992, is a rare
commonly involved area (Table VII; P < .05). With developmental cyst with uncertain histogenesis.37 In
regard to site distribution, residual cysts were found our study, this was found to be a rare cyst,
ORAL AND MAXILLOFACIAL SURGERY OOOO
136 Lo Muzio et al. August 2017

Table VII. Comparison of clinicopathologic data from our study and others
Localization (%)
Mean age M/F ratio Maxilla Mandible Frequency (% of odontogenic cyst)
Author (year) RC DC ReC RC DC ReC RC DC ReC RC DC ReC RC DC ReC
Jones et al. (2006)10 37.3 40.8 50.7 1.1:1 1.9:1 1.5:1 71.3 18.4 28.7 28.7 59.2 20.5 9.1
Ledesma-Montes et al. (2000)9 0.9:1 1.9:1 0.9:1 43.7 47.8 6.7
Mosqueda-Taylor et al. (2002)26 0.8:1 1.3:1 1.6:1 50.9 42.1 2.8
Meningaud et al. (2006)18 40.8 44.9 50.8 1.7:1 2.3:1 1.7:1 30.9 12.3 50.0 69.1 69.1 50.0 66.2 27.4 5.7
Ochsenius et al. (2007)25 0.9:1 1.7:1 1.1:1 75.5 45.0 67.1 24.5 24.5 32.9 59.2 21.6 13.0
Tortorici et al. (2008)17 35.6 31.0 1.1:1 1.6:1 53.2 40.9 46.8 46.8 88.1 11.9
Prockt et al. (2008)27 0.9:1 0.9:1 1.4:1 63.7 35.1 48.1 36.3 36.3 51.9 72.5 22.2 4.3
Avelar et al. (2009)21 1.3:1 1.7:1 0.8:1 52.3 30.8 5.9
de Souza et al. (2010)16 31.5 21.3 44.5 0.6:1 1.4:1 1.2:1 63.0 42.9 60.0 37.0 37.0 40.0 65.6 21.5 5.2
Ramachandra et al. (2011)15 30.9 29.8 1.8:1 1.1:1 65.3 44.4 34.7 34.7 69.2 30.8
Sharifian et al. (2011)14 28.7 21.5 40.0 1.3:1 1.3:1 1.2:1 61.0 45.4 39.0 39.0 47.1 30.7 10.0
Açikgöz et al. (2012)24 1.1:1 1.2:1 1.6:1 59.0 27.9 54.0 41.0 41.0 46.0 57.4 27.9 14.4
Tekkesin et al. (2012)3 33.7 32.8 45.7 1.2:1 1.5:1 1.7:1 61.0 34.8 55.8 39.0 39.0 44.2 70.8 13.4 12.4
Selvamani et al. (2012)20 0.9:1 2.9:1 4:1 58.5 67.7 20.0 41.5 32.3 80.0 69.3 20.3 3.3
Johnson et al. (2013)12 50.5 36.5 1.2:1 1.1:1 54.4 26.5 45.6 73.5 45.7 22.4
Demirkol et al. (2014)22 31.8 31.7 60.2 1.3:1 1.6:1 4:1 61.0 27.5 100 39.0 72.5 0 67.6 28.8 3.6
Present study 38.2 34.8 48.3 1.8:1 1.5:1 1.9:1 65.7 19.5 45.9 34.3 90.5 54.1 43.9 40.9 11.1
M/F, Male/female; RC, radicular cyst; DC, dentigerous cyst; ReC, residual cyst.

representing 0.71% of all OCs, in agreement with Given their clinical and histologic similarities to lateral
almost all studies3,12,14,16,18,21,26,37 (P < .05). Mean periodontal cysts, a common origin has been proposed.
age at diagnosis was 33.86 years, significantly lower In fact, lateral periodontal cysts are derived from the
than that reported in the literature10,14,16,18 (P < .05). reduced enamel epithelium before the eruption of teeth,
With regard to the gender distribution of glandular whereas gingival cysts of adults are formed after the
OCs, there were no significant differences, whereas a eruption of the dental element.2 Our study reported a
slight preponderance of males has been reported in relative frequency of 0.61%, in agreement with what
literature.37 These cysts were found to predominantly is reported in the literature10,14,21,25 (P < .05). Mean
occur in the mandible, similar to what is reported in age at diagnosis of these lesions was 32.67 years and
the literature.37 appeared to be more frequent in male patients, in
Lateral periodontal cysts are unusual developmental disagreement with the results of Jones et al.10 and
cysts that usually arise in the interradicular region or Sharifian et al.14
lateral to the root surface of a tooth.4 This lesion Eruptive cysts are considered a variant of dentigerous
represented 0.71% of all OCs in our study, cysts within the soft tissues overlying the alveolar ridge,
confirming the rarity of this cyst, as reported by many which covers the crown of an unerupted tooth.2 This is
studies.9,10,14,16,18,25-27 In the study by Tekkesin a rare lesion, representing 0.61% of all OCs, in
et al.,3 lateral periodontal cysts were rarer, whereas agreement with other authors9,10,14,16,21,25,26 Our re-
Avelar et al.21 and Johnson et al.12 reported a higher sults showed mean age at diagnosis of 12.67 years and
frequency. Age at diagnosis was more advanced than that males were more frequently affected, in agreement
that for other cysts (57.14 years) and significantly with the literature.10,14,16 Ledesma-Montes et al.9
higher than that reported in literature10,14,16,18 reported a higher frequency of these lesions in the
(P < .05). The reason for the advanced age at diag- first decade, in agreement with our findings. As
nosis could be that the lateral periodontal cyst often pointed out by Avelar et al., these cysts are
causes no signs or symptoms and is discovered by encountered probably more frequently in the clinic,
chance on routine radiography.27 We found a and the small number of eruptive cysts may be
preponderance of the male gender, in agreement with explained by the fact that these cysts are not
the findings of some authors,10,16,21,26 but differing submitted for histologic examination because of
from those of others.9,14 With regard to site distribution, spontaneous resolution.21
the lateral periodontal cyst occurred more frequently in The OOC is a cystic lesion that was described for the
the mandible, similar to what is reported in the first time by Wright as “odontogenic keratocyst:
literature.25 orthokeratinized variant.”38 As previously mentioned,
Gingival cysts of adults are developmental cysts that in 2005, the WHO reallocated OKCs from cystic to
arise from epithelial remnants within gingival tissues. neoplastic lesions, making the OOC an independent
OOOO ORIGINAL ARTICLE
Volume 124, Number 2 Lo Muzio et al. 137

pathologic entity. As reported in the literature, the 7. Lo Muzio L, Santarelli A, Caltabiano R, et al. p63 expression
relative incidence of OOCs ranged from 5.2% to correlates with pathological features and biological behaviour of
odontogenic tumours. Histopathology. 2006;49:211-214.
16.8% of cases that had been previously reported as 8. Gaitan-Cepeda LA, Quezada-Rivera D, Tenorio-Rocha F, Leyva-
OKCs.39 The re-evaluation of 78 cases previously Huerta ER. Reclassification of odontogenic keratocyst as tumour.
diagnosed as OKCs revealed that 9 were OOCs, cor- Impact on the odontogenic tumours prevalence. Oral Dis.
responding to 11.54% of the total. In contrast to the 2010;16:185-187.
relative frequency of OKCs, OOCs were rare lesions, 9. Ledesma-Montes C, Hernandez-Guerrero JC, Garces-Ortiz M.
Clinico-pathologic study of odontogenic cysts in a Mexican
representing 0.46% of all OCs, in agreement with de sample population. Arch Med Res. 2000;31:373-376.
Souza et al.,16 but differing from Selvamani et al.20 In 10. Jones AV, Craig GT, Franklin CD. Range and demographics of
fact, if we considered all the 78 cases of OKCs in our odontogenic cysts diagnosed in a UK population over a 30-year
study, the relative frequency would be 3.82%, but period. J Oral Pathol Med. 2006;35:500-507.
because these lesions represent only a small number 11. Lo Muzio L, Santarelli A, Caltabiano R, et al. p63 expression in
odontogenic cysts. Int J Oral Maxillofac Surg. 2005;34:668-673.
of OKCs, the percentage of their occurrence was 12. Johnson NR, Savage NW, Kazoullis S, Batstone MD.
significantly low. We found a mean age at diagnosis A prospective epidemiological study for odontogenic and non-
of 35.56 years, with preponderance in male patients odontogenic lesions of the maxilla and mandible in Queensland.
and a greater involvement of the mandible, in Oral Surg Oral Med Oral Pathol Oral Radiol. 2013;115:515-522.
agreement with other studies.16,20,39 13. Daley TD, Wysocki GP, Pringle GA. Relative incidence of
odontogenic tumors and oral and jaw cysts in a Canadian popu-
Nonodontogenic cysts represented 2.46% of all jaw lation. Oral Surg Oral Med Oral Pathol. 1994;77:276-280.
cysts in our study. The nasopalatine cyst was, by far, 14. Sharifian MJ, Khalili M. Odontogenic cysts: a retrospective study
the most frequent lesion, representing 76% of all non- of 1227 cases in an Iranian population from 1987 to 2007. J Oral
odontogenic cysts, in agreement with Daley et al.13 and Sci. 2011;53:361-367.
Tekkesin et al.3 15. Ramachandra P, Maligi P, Raghuveer H. A cumulative analysis of
odontogenic cysts from major dental institutions of Bangalore
city: a study of 252 cases. J Oral Maxillofac Pathol. 2011;15:1-5.
CONCLUSIONS 16. de Souza LB, Gordón-Núñez MA, Nonaka CF, de Medeiros MC,
Our study showed the frequency of jaw cysts as seen in Torres TF, Emiliano GB. Odontogenic cysts: demographic profile
a single institution over a 40-year period. A wide va- in a Brazilian population over a 38-year period. Med Oral Patol
Oral Cir Bucal. 2010;15:e583-e590.
riety of cysts was noted, some of which varied ac- 17. Tortorici S, Amodio E, Massenti MF, Buzzanca ML, Burruano F,
cording to gender, age, and localization. There are some Vitale F. Prevalence and distribution of odontogenic cysts in
differences between our results and those presented in Sicily: 1986e2005. J Oral Sci. 2008;50:15-18.
the literature; the main one is the lower frequency of 18. Meningaud JP, Oprean N, Pitak-Arnnop P, Bertrand JC. Odonto-
radicular cysts. It is yet to be ascertained if this differ- genic cysts: a clinical study of 695 cases. J Oral Sci. 2006;48:59-62.
19. Nùñez-Urrutia S, Figueiredo R, Gay-Escoda C. Retrospective
ence resulted from geographic differences in the inci- clinicopathological study of 418 odontogenic cysts. Med Oral
dence of cysts or several biases in the data collection. Patol Oral Cir Bucal. 2010;15:e767-e773.
Moreover, it is important to point out that some of 20. Selvamani M, Donoghue M, Basandi PS. Analysis of 153 cases of
these cysts tend to recur more than others and have a odontogenic cysts in a South Indian sample population: a retro-
more locally aggressive behavior. This may lead to spective study over a decade. Braz Oral Res. 2012;26:330-334.
21. Avelar RL, Antunes AA, Carvalho RW, Bezerra PG, Oliveira
drastic surgical options, which can be avoided by Neto PJ, Andrade ES. Odontogenic cysts: a clinicopathological
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