Int. J. Oral Maxillofac. Surg.

2015; 44: 15651568

http://dx.doi.org/10.1016/j.ijom.2015.03.008, available online at http://www.sciencedirect.com

A Personal View

The keratocystic odontogenic

tumour (KCOT)an odyssey

M. A. Pogrel
Oral and Maxillofacial Surgery, University of
California San Francisco, San Francisco, CA,
USA

M.A. Pogrel: The keratocystic odontogenic tumour (KCOT)an odyssey. Int. J. Oral
Maxillofac. Surg. 2015; 44: 15651568. # 2015 International Association of Oral and
Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Abstract. The most appropriate management for the lesion now known as the
keratocystic odontogenic tumour (previously known as the odontogenic keratocyst)
remains controversial. This article reviews the different management protocols
adopted by one surgical unit over the last 30 years and the results obtained from the
different treatment modalities. A current treatment protocol consisting of initial
decompression followed by aggressive curettage and peripheral ostectomy with
methylene blue staining appears to be successful, but our longest follow-up is only 6
years.

The lesion now called the keratocystic

odontogenic tumour (KCOT) was first
described by Philipsen in 19561 under
the name odontogenic keratocyst. This
original article was in Danish but had a
summary in English. It was, however, not
widely recognized as a separate entity
until the articles by Browne in 1970 and
1971,2,3 which clearly delineated the clinical and histological features of the lesion,
coupled with its high recurrence rate with
simple enucleation. Prior to this time, it is
felt that most of these lesions were identified as primordial cysts, which is a term no
longer used.
Following the confirmation of the accuracy of histological diagnosis and of the
high recurrence rate of these lesions with
simple treatment, there was a division of
philosophies on their management. These
varied from the feeling of my old chief,
Gordon Hardman from North Wales, who
essentially stated that we always knew
there were some cysts that recurred, even

0901-5027/01201565 + 04

though we did not have a special name for

them, and all we did was curette them out
every 510 years. Why shouldnt we just
continue to do that? all the way to the
opinion of people like Paul Bramley4 who
recommended much more aggressive
treatment, consisting of either en-bloc
or segmental resection, depending on the
size. With these two diametrically opposing views, an effort was made to identify a
middle course that might result in an
acceptable success rate in the long term,
with an acceptably low morbidity. Paul
Stoelinga and others are of the opinion
that these lesions can arise from downgrowth from the oral epithelium and that
therefore the overlying oral epithelium
should be excised along with the lesion.5
Since these lesions normally occur in
the posterior mandible, this can involve
incisions over on the lingual side of the
crest of the ridge, so care has to be taken
to identify and protect the lingual nerve,
if appropriate.

Key words: keratocystic odontogenic tumour;

odontogenic keratocyst; treatment protocols.
Accepted for publication 10 March 2015
Available online 21 May 2015

In an effort to identify this middle

course, treatment has consisted of techniques ranging from enucleation plus cryosurgery a technique learned from one of
my mentors, Paul Bradley,6,7 and utilized
extensively over the last 30 years8,9 to
enucleation plus Carnoys solution,10,11
enucleation with peripheral ostectomy,12
and more recently marsupialization and
decompression,1316 with various combinations.
We first published an article on the use
of enucleation coupled with liquid nitrogen in 19938 and at that time reported a
recurrence rate of around 11.5%, which
was less than the recurrence rates of
2060% that were being reported for simple enucleation. With the extended followup of these patients for a longer period
(up to 25 years now), it does appear that
the recurrence rate has remained around
10%. The problem with the technique,
however, has been access to the equipment, which is being used less and less

# 2015 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

1566

Pogrel

in other medical specialties, so hospitals

often do not have access to it. Also, if one
is not meticulous in protecting the soft
tissues, there will be necrosis of the soft
tissues leading to wound breakdown and
a prolonged recovery, and at least partial
loss of any bone graft that has been used.
At that time we were recommending bone
grafting of any lesion over 4 cm in size,17
but ultimately reduced this to 3 cm in size
in order to accelerate the healing of smaller lesions.
Around 1995, following the final retirement of my mentor, Gordon Hardman, in
North Wales, I spent some time sorting
out his slide collection which consisted
of many thousands of 35-mm Kodachrome slides. In this I found a number
of cases of cysts that he had obviously
marsupialized and decompressed in the
early 1950s or possibly even earlier. From
examining these slides, it did appear to
be a practical technique, which had
been largely forgotten following the widespread use of enucleation and primary
closure, coupled with the liberal use of
antibiotics.
It was therefore decided to apply this as
a definitive technique for the management
of odontogenic keratocysts, and we

Fig. 1. A drainage tube made from a paediatric feeding tube and wired around the first molar.
The tube is going into a large cyst of the left mandibular angle area, and the tube ends opposite
the first bicuspid tooth making it easier for the patient to irrigate.

published our first results in 2004. These

seemed to show very satisfactory outcomes for a relatively small number of
patients,14 with a recurrence rate around
5%. However, during the follow-up of
these patients, we started to see more
recurrences, and in 2007 I published a
partial retraction stating a recurrence rate
of around 12% over a relatively short
period of time.16 Since that time, the
number of patients that we have treated

with this technique has risen to 73, and the

recurrence rate at the present time has
risen to 27% (20 recurrences). The longer
we follow these patients, the more recurrences we seem to see, and one can only
speculate what the final recurrence rate
may be when these patients have been
followed up for 20 years or longer.
Once we realized that the recurrence
rate was going to be as high as it had
turned out to be, we looked around for an

Fig. 2. Radiograph showing a large keratocystic odontogenic tumour extending from the lower left first molar over to the lower right first molar
region (A). This was drained from two separate sites (the drainage tubes can be seen on the radiograph), resulting in almost complete resolution of
the lesion (B).

The keratocystic odontogenic tumour

alternative technique that would combine
the best of these techniques. In 2006,
we introduced the technique whereby
we decompressed the lesions until they
appeared radiographically to have resolved
down to about 23 cm in size, and we then
surgically enucleated the lesions and carried out a peripheral ostectomy to remove
any cyst remnants that might be in the
surrounding bone. The advantages of this
technique are outlined below.
The lesions do decompress very satisfactorily. By this time we had modified the
technique to utilize a 14 French gauge
paediatric feeding tube as the decompression tube and to wire it around the adjacent
first molar tooth, passing the wire through
the wall of the tube so as not to compress
it. We could then bring the drainage tube
forward to the bicuspid region where
patients found it easy to irrigate (Fig. 1).
This meant that patients could keep the
area clean and irrigate it more easily. If the
lesion was anywhere else except the posterior mandible, the technique was modified accordingly. For extremely large
lesions that crossed the midline, a drainage
tube could be placed on each side (Fig. 2).
Patients are followed radiographically
at 3-month intervals. The drains can be

shortened as appropriate, and the radiographic shrinkage of the lesion noted.

When the lesion is approximately 2
3 cm in size, the decision can be made
to surgically enucleate the remaining cyst.
At this stage, it was noted that the cyst
lining had become thicker and easier
to enucleate than with the original KCOT.
This has been noted before, that there is
a change in the histological nature of
the cyst lining in that it does become
thicker, more robust, and more like oral
epithelium.14,18
It was, however, felt that additional
treatment was required to remove any
small daughter cysts or remnants beyond
the visible cyst lining. By this time,
Carnoys solution had become virtually
unobtainable in California, as the California Occupational Health and Safety Administration (CalOSHA) had classified
the chloroform in it as a carcinogen, and
the handling of glacial acetic acid requires
a high volume fume cupboard. Some practitioners have been removing the chloroform from the Carnoys solution, but this
new formulation has not been tested and
has not been shown to be effective.
We could have gone back to using liquid
nitrogen cryosurgery, but the problems

1567

with this have already been mentioned.

In view of the issues with liquid nitrogen,
we elected to use a technique of peripheral
ostectomy. For this, the cyst is first enucleated, and then the bony margins are painted
with methylene blue using a Q-tip. Once the
walls are completely stained, the cavity is
irrigated and then all the remaining bluestaining bone is removed with a pineappletype bur; the residual cavity is then thoroughly irrigated. It does appear that the
methylene blue stains to a depth of about
0.5 mm in cortical bone and 11.5 mm in
cancellous bone, so if the remaining cells
are within this area, they should be removed
(Fig. 3).
In the meantime, in 2006 the World
Health Organization reclassified this
lesion from an odontogenic cyst to a benign odontogenic tumour and renamed
it the keratocystic odontogenic tumour
(KCOT).19,20 Initially the odontogenic
keratocyst existed in two forms, a parakeratinized form and an orthokeratinized
form. The term keratocystic odontogenic
tumour only refers to the parakeratinized
form of this lesion, whilst the orthokeratinized form continues to be classified as
a benign odontogenic cyst and does
not appear to have the same recurrence
potential.
To date, we have treated some 29
lesions utilizing this modified technique
of decompression, enucleation, and peripheral ostectomy. Our follow-up period
varies, with six of these patients now
followed for longer than 5 years and the
shortest only followed for 4 months. To
date, we have not seen any recurrences,
but time will tell whether this proves to be
a superior technique to any of the other
techniques described. If it does stand the
test of time, it will represent a reasonable
middle course between enucleation and
resection for the lesion now known as
the keratocystic odontogenic tumour.
Funding

None.
Competing interests

None declared.
Ethical approval

Not required.
Patient consent
Fig. 3. The technique of peripheral ostectomy. (A) The area is opened up, the overlying mucosa
is excised, and the cyst is enucleated. (B) The bony margins are painted with methylene blue on a
Q-tip, and then all the blue-stained bone is removed with a pineapple bur taking care to avoid
trauma to the inferior alveolar nerve.

All our patients sign a consent to be

photographed and for the images to be
used for academic purposes.

1568

Pogrel

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Address:
M. Anthony Pogrel
Oral and Maxillofacial Surgery
University of California San Francisco
Room C522
Box 0440
521 Parnassus Avenue
San Francisco
CA 94143-0440
USA
Tel: +1 415 476 8225;
Fax: +1 415 476 6305
E-mail: tony.pogrel@ucsf.edu