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Jonathan Madras, BSc (Hons), DDS; Henry Lapointe, DDS, PhD, FRCD(C)
Dr. Lapointe
Email: hlapoint@uwo.ca
ABSTRACT
The purpose of this paper is to review the features and behaviour of the odontogenic
keratocyst (OKC), now officially known as the keratocystic odontogenic tumour (KCOT);
to analyze a series of histologically confirmed KCOT cases; and to review and discuss the
redesignation of KCOT and the implications for treatment. Based on a literature review,
more aggressive treatment either resection or enucleation supplemented with Carnoys
solution with or without peripheral ostectomy results in a lower recurrence rate than
enucleation alone or marsupialization. However, the recurrence rate after marsupialization followed by enucleation is not significantly higher than that after aggressive modalities. In a case series of 21 patients (27 KCOTs), recurrence rate was 29%, consistent with
published data; all recurrences occurred within 2 years after intervention. The size of most
lesions was 015 cm2 (average 14 cm2) measured radiographically. WHOs reclassification
of this lesion from cyst to tumour underscores its aggressive nature and should motivate
clinicians to manage the disease in a correspondingly aggressive manner. The most
effective treatments are enucleation supplemented with Carnoys solution, or marsupialization with later cystectomy. Future treatment may involve molecular-based modalities,
which may reduce or eliminate the need for aggressive surgical management.
For citation purposes, the electronic version is the definitive version of this article: www.cda-adc.ca/jcda/vol-74/issue-2/165.html
165
Lapointe
Location
Surface
area; cm2
24
1912-08-18
Anterior mandible
83
1918-01-13
6.5 3
1921-11-16
3 1.5
11
1922-07-17
4.5 1.5
1925-07-22
1925-11-27
1927-07-07
Treatment
Follow-up period
Curettage
1 year
Marsupialization
1 year
19.5
4.5
Curettage
Curettage
5.5 years
6.75
Curettage
5 years
5.5 3.5
19.25
Marsupialization
3.5 years
6.5 2.5
16.25
Curettage
2.5 years
At teeth 44 and 45
11
Curettage
Recurrence at 1 year
At teeth 44 and 45
11
Curettage
Recurrence at 2 years
11
Curettage
6 years
6.75
Resection
5 years
At teeth 44 and 45
Size; cm
1929-01-19
4.5 1.5
1959-09-07
43
12
Curettage
2 months
1933-05-15
2.5 1.5
3.75
Curettage
5.5 years
1936-06-08
6.5 3
19.5
Curettage
Recurrence at 8 months
At teeth 4446
2.5 2
Curettage
5 years
22.5
Curettage
1 year
1946-03-03
9 2.5
1949-01-13
10 4.5
45
Resection
6 years
1949-03-22
Anterior mandible
63
18
Curettage
Recurrence at 9 months
Anterior mandible
42
Curettage
7 years
1957-08-28
73
21
Marsupialization
1.5 years
1957-12-01
2.5 3.5
8.75
Curettage
16 months
1960-07-18
4.5 4.5
20.25
Curettage
18 months
1961-04-04
8 4.5
36
Curettage
1 year
1966-09-23
43
12
Curettage
3.5 1.5
5.25
Curettage
2 years
1975-12-26
64
24
Curettage
2.5 years
1986-05-06
Right maxilla
52
10
Curettage
2 years
165a
20
18
16
14
12
10
8
6
4
2
0
Posterior
maxilla
Anterior
maxilla
Posterior
mandible
Anterior
mandible
% of disease-free patients
Number of lesions
100
90
80
70
60
50
40
30
20
10
0
KCOT location
Number of lesions
Recurences
16
14
12
10
8
6
4
2
0
> 30
7
6
5
4
3
2
1
0
1019
2029
3039
4049
5059
6069
7079
80+
Sample Cases
Total lesions
> 1530
015
165b
Lapointe
Figure 5: Partial panoramic radiograph taken (a) pre-operatively and (b) 6 days and (c) 6 years after resection.
a
Figure 6: Partial panoramic radiograph taken (a) pre-operatively, (b) 9 days, (c) 3 months and (d) 3.5 years after marsupialization.
a
Figure 7: (a) Pre-operative radiographic appearance of the lesion. (b) Recurrence at 9 months after curettage; and (c) 16 months and
(d) 7 years after curettage of the recurring tumour.
Cysts
Follow-up period
Recurrence
rate; %
29
Enucleation
18 years
24
Chow22
70
5 years
10
49
Enucleation
115 years
35
31
Resection
28 years
Stoelinga
82
Enucleation + Carnoys
125 years
11
63
Enucleation
> 5 years
29
16
Enucleation + cryosurgery
> 5 years
38
> 5 years
> 5 years
50
Resection
> 5 years
23
Marsupialization + enucleation
119 years
10
1.84.8 years
30
Approx. 25 months
14
11
Enucleation
13288 months
55
11
Peripheral ostectomy
13288 months
18
13
13288 months
Enucleation + Carnoys
13288 months
50
Resection
13288 months
44
719 years
18
12
Marsupialization
> 16 months
25
72
Enucleation
> 16 months
23
28
Enucleation in 1 piece
517 years
18
41
517 years
56
Marsupialization
517 years
60
12
Enucleation
1758 months
33
13
Enucleation + cryotherapy
2159 months
38
52
Enucleation
121 years
14
40
Enucleation + Carnoys
110 years
22
Enucleation
19 months to 10 years
18
Resection
19 months to 10 years
20
24
25
26
17
27
5
Jensen and others
29
30
1
Vedtofte and Praetorius
Zachariades and others
Treatment
32
57
Enucleation
5 years
51
33
13
Enucleation
> 5 years
31
Resection
> 5 years
Marsupialization
> 5 years
Decompression + enucleation
> 5 years
165d
Lapointe
Lesions
Enucleation
465
141
Enucleation + Carnoys
122
11
Enucleation +
peripheral ostectomy
11
Enucleation + Carnoys +
peripheral ostectomy
83
Enucleation + cryotherapy
29
11
Marsupialization
Marsupialization +
cystectomy
Resection
Recurrences
18
108
14
39
Recurrence
rate; %
30
Recurrence
A review of the literature suggests that recurrence
rate is relatively low with aggressive
18
treatment, whereas more conservative methods
tend to result in more recurrences (Tables 2
8
and 3). Articles reviewed were required to
meet the following inclusion criteria: histo38
logic diagnosis of OKC, a defined follow-up
33
period and a clear description of treatment.
13
If a difference in recurrence rate between
2 modalities of 15% (arbitrarily chosen) is
considered the threshold for clinical signifi0
cance, a few simple inferences are possible.
First, enucleation plus Carnoys solution, with
or without peripheral ostectomy, results in a significantly
lower rate of recurrence than enucleation alone. Second,
the use of cryotherapy with enucleation appears to have no
significant effect on the recurrence rate compared with
enucleation alone. Third, marsupialization as a definitive
treatment is associated with a significantly higher recurrence rate than when the KCOT is subsequently enucleated. Finally, resection, despite a recurrence rate of 0, is
not significantly better at eliminating recurrences than
enucleation plus Carnoys solution or marsupialization
plus cystectomy. Therefore, to minimize invasiveness and
recurrence, the most effective treatment option appears
to be enucleation of the KCOT and subsequent application of Carnoys solution. Alternatively, marsupialization followed by cystectomy is likewise effective, as this
treatment does not result in a significantly higher rate
of recurrence than enucleation plus Carnoys solution.
However, as the latter option requires a protracted course
of treatment, patient compliance must be considered; lesions treated in this manner require several months of
at-home irrigation by the patient as well as clinical observation before enucleation.
9
and showed promising results (0 recurrences), the followup period ( 4.8 years) and sample size (10 patients) were
inadequate to draw definitive conclusions.
Despite the fact that resection of the jaw results in
the lowest recurrence rate, this procedure is extreme.
Thus, unless resection is deemed necessary, the most appropriate action would be enucleation of the KCOT plus
use of Carnoys solution or marsupialization followed by
enucleation.
In our case series, smaller lesions were more often
associated with recurrence. This contradicts our expectations, as larger lesions should be inherently more difficult to excise in one piece and, therefore, should be
more likely to recur. To date, the literature makes little
mention of recurrence of large versus small lesions. The
largest of our lesions was resected and, as supported by
the literature, this method of treatment was associated
with the lowest rate of recurrence. This could influence
the results, making small lesions appear to recur more
often. Thus, our results regarding lesion size and associated recurrence are inconclusive. Notably, Forssell and
others28 found that lesion size does not affect recurrence
rate, confirming earlier observations.
Regarding a relation between treatment modality and
recurrence, in the case series all recurrences followed
enucleation and curettage.
In our study, the tumours presented primarily in
the posterior mandible, in accordance with findings
described above under clinical features. Likewise, in
agreement with earlier research, the recurrence rate we
observed was 29%.13 Our follow-up interval ranged from
2 months to 7 years, with the variability attributed to
patient compliance and time since surgery. Although all
recurrences took place within 2 years post-intervention,
it remains prudent to suggest at least 5 years follow-up for
KCOTs for reasons stated earlier.
In recent years, studies have hinted at possible new
treatment methods for KCOT. According to Taipale and
colleagues,46 cyclopamine, a plant-based steroidal alka-
165f
Lapointe
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THE AUTHORS
Dr. Madras is a graduate of the Schulich School of Medicine
and Dentistry, University of Western Ontario, London,
Ontario. He is currently a general practice resident at Mount
Sinai Hospital, Toronto, Ontario.
165g
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