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4 October 2013
Objectives. The objective of this study was to evaluate the most up-to-date treatment modalities and respective recurrence
rates for keratocystic odontogenic tumor (KCOT).
Study Design. A systematic review of the literature from 1999 to 2010 was undertaken examining treatment and recurrence
rates for KCOT. Four inclusion criteria were defined for articles to then be analyzed against 8 standards.
Results. Of the 2736 published articles, 8 met the inclusion criteria. When merging the data, enucleation and enucleation
with adjunctive measures (other than Carnoys solution) had recurrence rates of 25.6% and 30.3%, respectively.
Marsupialization with adjunctive measures produced a recurrence rate of 15.8%, whereas enucleation with Carnoys solution
presented a recurrence rate of 7.9%. Only one resection case had recurrence (6.3%).
Conclusions. The enucleation technique with the use of adjunctive procedures (other than Carnoys solution) provides a
higher recurrence rate than any other treatment modality. (Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:e271-e276)
vides the lowest recurrence rate. A review of the literature is the best available technique without the ethical
dilemma inherent in a clinical trial, to determine recurrence rates for the contrasting surgical options and the
most appropriate management of this lesion. A systematic review by Blanas et al.,9 published in 2000, included studies from 1970 to 1998. The aim of this
current systematic review was to provide an update on
the management and recurrence rates of KCOT, building on the work from Blanas et al.9 The data from 1999
to 2010 were analyzed and compared with the data
from Blanas et al.9 The data were then combined to
provide a current consensus on management and up-todate recurrence rates for the different treatment modalities.
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8 standards of inclusion
1-4. Inclusion criteria.
RESULTS
Using the keywords described in the preceding paragraph, 2736 articles were identified. Limiting the search
between the 1999 and 2010 produced 206 articles, with
28 articles having consecutive cases. Eight articles (Table II) met the inclusion criteria, which were then
analyzed using the 8 standards.
All studies confirmed that histopathology was used
to diagnose the KCOTs (Criterion 1). Most cases were
reviewed retrospectively, with 1 study prospective, but
all were consecutive (Criterion 2). Follow-up periods
were provided in all studies (Criterion 3); however, the
reporting of this varied between authors. Some stated a
range for follow-up, whereas others provided an average length of time. Treatment modalities were described in adequate detail so that they could be repeated
if necessary, with recurrence rates provided for each
treatment option (Criterion 4). The different treatment
modalities located in the literature and described by
Blanas et al.9 are presented as follows:
1. Curettage is the method where the wall of the cyst
cavity is surgically scraped with the removal of its
contents.14
2. Enucleation is the removal of a lesion intact.15 As
the lining of the cyst may be friable and thin, removal of the cyst in one piece is difficult.16 To
combat this feature, a number of studies suggest that
the general treatment of the primary KCOT should
include enucleation of the cyst, followed by mechanical curettage using methylene blue as a marking agent, followed by a 3-minute application of
Carnoys solution (a tissue fixative).1,3,8,15-19 This
treatment option has the advantage of preservation
of the adjacent bone, soft tissue, and dental structures. This results in reduced morbidity and cost of
treatment.8,16,20
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ORIGINAL ARTICLE
Johnson et al. e273
Table II. Articles included in the systematic review for management and recurrence of KCOT with follow-up
periods noted
Stoelinga
Author
Year
Treatment
Cysts
Recurrence
Follow-up period
2001
E
E CS
E EoM
E EoM CS
M then E
R
M
M for at least 6 mo then E CE
E CE
M for 7-19 mo
E
EO
E CS
E O CS
R
E CE
D CE
M
E
E CS
E CE
R
EO
R
D then E
E
R
33
5
4
38
22
2
5
23
15
10
11
11
2
13
3
10
20
6
15
5
2
7
8
3
11
119
1
6 (18%)
0
1-25 y
Nakamura et al.12
2002
2004
2005
Maurette et al.11
2006
Chirapathomsakul et al.13
2006
Kolokythas et al.6
2007
Pitak-Arnnop et al.1
2010
3 (7.8%)
0
0
6 (26.1%)
3 (20%)
0
6 (54.5%)
2 (18.2%)
1 (50%)
0
0
4 (20%)
1 (16.7%)
2 (13.3%)
1 (20%)
2 (100%)
1 (14.2%)
0
2 (18%)
28 (26%)
0
3-14 y
1.8-4.8 y
1-24 y
Average 2 y
1-14.6 y
1.5-9 y
1.5-3 y
1.5-12.5 y
CE, curettage; CS, Carnoys solution; D, decompression; E, enucleation; EoM, excision of mucosa; M, marsupialization; O, ostectomy; R,
resection.
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Table III. Comparison of data from Blanas et al.9 and current review (1999-2010)
Treatment
type
Total cysts
reported
Blanas et al.9
Total cysts
reported
1999-2010
Total
recurrences
Blanas et al.9
Total
recurrences
1999-2010
Calculated
recurrence rate, %,
Blanas et al.9
Calculated
recurrence rate, %,
1999-2010
26
387
22
60
45
38
156
33
63
21
76
16
5
111
6
1
11
40
10
5
1
12
1
19.2
28.7
27.3
1.6
24.4
25.6
30.3
7.9
4.8
15.8
6.3
CE
E alone
EA
E CS
M alone
MA
R
A, adjunctive measures other than Carnoys solution; CE, curettage; CS, Carnoys solution; D, decompression; E, enucleation; M, marsupialization;
R, resection.
Table IV. Combined data from Blanas et al.9 and current review (1999-2010)
Treatment Combined total Combined total Combined calculated
type
cysts reported
recurrence
recurrence rate, %
CE
E alone
EA
E CS
M alone
MA
R
26
543
52
123
66
76
54
5
151
16
6
12
12
1
19.2
27.8
30.8
4.8
18.2
15.8
1.85
by marsupialization alone (18.2%%). The use of Carnoys solution reduced recurrence to 4.8%, and resection had the lowest recurrence rate (1.85%). Treatment
of recurrence was reported in only half of the articles,1,3,6,13 with management being enucleation with
adjunctive measures for all cases except 1 (patientelected resection1). Curettage alone was not a technique
reported by authors in this systematic review, therefore
only the data from Blanas et al.9 were provided for this
treatment option.
DISCUSSION
The purpose of this investigation was to build on the
foundation made by Blanas et al.9 and update their
systematic review with the research conducted between
1999 and 2010. With 940 KCOTs analyzed, this review
provides the largest, contemporaneous consensus on
the management of this tumor. There are many variables that preclude any uniform quantitative analysis
being undertaken with regard to the management of
KCOT, hence randomized controlled trials have not
been conducted. These variables include size and location of the lesion, presence of infection, and association
of any teeth. It is difficult to control these variables
because of the nature of the KCOT.
As there are no randomized controlled trials available in the literature, a meta-analysis cannot be used to
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Volume 116, Number 4
sular surface with little separation of the lining epithelium from the outer surface.
The average depth of bone penetration depends on
the duration of application (1.54 mm after 5 minutes).15
The Carnoys solution will prevent recurrence caused
by the presence of satellite cysts, budding of the cyst
lining, or remnants of cyst epithelium remaining after
enucleation.10,20,21
Marsupialization results in a considerable reduction
of the cystic volume and lessens the chance of injury to
important anatomical structures (such as the inferior
dental nerve).1,5-7,9,12,24,25 Some authors suggest using
the marsupialization technique to reduce the size of the
KCOT and then proceed to surgical enucleation of the
remaining cyst structure.7,11 This 2-stage technique
does not affect the recurrence rate.12,24,25 The main
advantage of the conservative treatment option is the
preservation of bone and teeth associated with the
KCOT.11 Reducing the size of the cystic cavity by
decompression, promotes new bone formation in a centripetal direction, with filling of the defect and reduction in cyst volume.11,12,24
As most patients with KCOT are young with high
regenerative capacity, these procedures are less traumatic and avoid the need to accomplish reconstruction
through grafts11; however, this technique necessitates
longer treatment (up to 14 months11,24), multiple-staged
procedures, and exceptional patient cooperation.1,5-7,9,24
These are the main causes for abandonment of the
treatment by the patient, along with loss of interest in
proper irrigation protocols.11 Enucleation and curettage
techniques have a clear advantage over marsupialization by providing a complete specimen for histopathologic analysis.1,7
One of 54 KCOTs recurred after resection, with
Chirapathomsakul et al.13 stating that this specific case
was treated with segmental resection. The authors13
hypothesized that the recurrence occurred within the
bone graft as a result of a new KCOT developing from
the overlying mucosa and penetrating the graft.13
Some authors question the acceptability of resection
for a benign condition, as it is a destructive procedure.9 It is suggested that aggressive resection should
be limited to recurrent KCOTs (3 or more times) or
those that have undergone ameloblastic or malignant
degeneration.6,8,9,21,27
Recurrence of KCOT occurs for several reasons.
Incomplete removal of the cystic lesion allows new cyst
formation or epithelial islands in the wall of the original
cyst remain in the surrounding bone or soft tissue.7,28,29
New KCOTs can also develop from the basal layer of
the oral epithelium.30 Patients with NBCCS are more
prone to continuous formation of new cysts.7 Size,
location, and inadequate surgical treatment of the
ORIGINAL ARTICLE
Johnson et al. e275
CONCLUSIONS
This systematic review has analyzed the literature to
provide a balanced comparison among the different
treatment modalities for KCOT. The goals of management include elimination of the pathology and decrease
potential recurrence while minimizing harm to the patient. The issue surgeons encounter is whether to take a
conservative approach, minimizing the morbidity to the
patient, knowing that several operations may be required to eliminate recurrence; or being more aggressive and potentially more destructive, at the same time
establishing the maximum position to avoid recurrence.
The use of adjunctive measures beyond simple enucleation or marsupialization is justified. Enucleation with
Carnoys solution provides the least recurrence (4.8%)
from any of the conservative techniques. Resection
provides the lowest recurrence rate (1.85%), yet causes
the most suffering to the patient.
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October 2013
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Reprint requests:
Nigel Johnson, BDSc (Hons), MBBS
Maxillofacial Department
Royal Brisbane and Womens Hospital
996 Logan Road
Brisbane, Queensland, Australia
nigel.johnson1@uqconnect.edu.au