CLINICAL CONTROVERSIES IN ORAL AND MAXILLOFACIAL SURGERY: PART ONE
J Oral Maxillofac Surg
63:1662-1666, 2005
The Treatment of Odontogenic
Keratocysts by Excision of the Overlying,
Attached Mucosa, Enucleation, and
Treatment of the Bony Defect With
Carnoy Solution
Paul J.W. Stoelinga, DDS, MD, PhD*
Odontogenic keratocysts (OKCs) are known for their
propensity to recur. The rate of recurrence is proba-
bly largely depending on the mode of treatment used.
Unfortunately, there are only a few reliable studies
available from which meaningful conclusions can be
drawn with regard to the best possible treatment
strategy.1 The main drawback of most studies is the
retrospective nature of the study, the variable fol-
low-up time, the lack of information about the per-
centage of patients available for follow-up, and the
lack of details about the actual surgery. Many papers
are written by pathologists who obviously are de-
pending on the written and often incomplete notes of
surgeons, hence the link with the clinical parameters
is often based on less than optimal information. It is
the aim of this article to present a rational treatment
protocol based on clinical and histologic studies.
Etiology and Pathogenesis
To understand the clinical presentation of OKCs,
and their propensity to recur, it is necessary to have
some knowledge of the etiology and pathogenesis of
OKCs. The etiology is probably closely related to the
development of the dental lamina and in particular
remnants of it after this organ has served its purpose.
OKCs that occur in the dentate areas of both the
maxilla and mandible probably derive from those rem-
nants. However, one must realize that epithelial is-
lands that are derived from the dental lamina are
*Professor and Chairman, Department of Oral and Maxillofacial
Surgery, University of Nijmegen, The Netherlands.
Address correspondence and reprint requests to Dr Stoelinga:
P.O. Box 9101, 6500 HB Nijmegen, The Netherlands; e-mail:
p.stoelinga@mkc.umcn.nl
© 2005 American Association of Oral and Maxillofacial Surgeons
0278-2391/05/6311-0015$30.00/0
doi:10.1016/j.joms.2005.08.007
1662
mainly found in the gingiva and probably in the peri-
odontal ligament. This explains the often seen lateral
periodontal or lateral follicular presentation of these
cysts. It is not known why keratocysts develop from
such epithelial residues. It is also not clear why a
keratocyst develops from one such epithelial island,
while others remain dormant. The clinical implication
may be, however, that if one removes such an OKC
some of these epithelial residues may be left behind
which may later give rise to a new keratocyst.
The frequently seen presentation of keratocysts in
the ascending ramus of the mandible is less easily
explained by this theory. Stoelinga and Peters2 first
pointed toward the course of the dental lamina form-
ing the third molar and the unlikeliness of possible
remnants or offshoots of this part of the dental lamina
to be located in the mucosa posterior to the last
molar. Yet, there is plenty of evidence that the ma-
jority of epithelial islands, as found in the wall of
OKCs, are in fact located in the mucosa that is over-
lying the OKC and attached to it. This is the reason
why it is thought that offshoots of the basal layer of
the epithelium of the oral mucosa may also be in-
volved in the etiology of OKCs.3,4
Whatever the causative epithelial source may be,
epithelial islands and/or microcysts are found in ap-
proximately 50% of the cases in the overlying, at-
tached mucosa.5 It is, therefore, of paramount impor-
tance to locate the area where the cyst is attached to
the mucosa and to excise that part of the mucosa,
preferably in conjunction with enucleation of the
cyst. In failing to do so, in approximately 50% of cases
one will leave behind possible sources of recurrent
OKCs, or better to say newly formed cysts.
Clinical Presentation
One of the problems when discussing keratocysts is
that there are in fact 2 clinical types. The first type
is the commonly, relatively small, unilocular cyst in
PAUL J.W. STOELINGA
the dentate area that often presents as a lateral peri-
odontal or lateral follicular cyst. The clinician has no
way of knowing in advance whether the lesions are to
be considered an OKC. Excision of the attached mu-
cosa is, therefore, usually not performed and most
often the cysts are just enucleated. A prospective
study including 82 OKCs showed that in approxi-
mately 40% of the cases a preoperative diagnosis was
not made because of the specific presentation of the
cyst.5 Recurrences will, therefore, be unavoidable but
will present themselves in an area that is easily acces-
sible. The second type is the frequently large, often
multilobular or multilocular cyst in the posterior max-
illa or angle and ascending ramus of the mandible. In
these locations a high degree of alertness should be
present and aspiration biopsies may be performed to
ascertain the diagnosis.5,6 The differential diagnosis in
all these cases should include unicystic ameloblas-
toma. Treatment of these 2 lesions should not be all
that different.6,7 An algorithm on how to approach
cystic lesions of the jaws has been presented else-
where.6
Analysis of Nature of Recurrences
Studies on recurrent OKCs have shown that in
almost 100% of the cases epithelial islands and or
microcysts are present in the overlying, attached mu-
cosa.5,8,9 No microcysts were found in the surround-
ing bone when block resections were carried out on
5 recurrent OKCs.8 Therefore, this author is very
critical of proponents of radical resections including
continuity resections of the mandible or maxillecto-
mies. To the best of my knowledge no studies or even
case reports are available to prove that microcysts do
occur in the bone surrounding the OKC. For this
reason there is very little reason to perform these
resections or to freeze the bone over the full thick-
ness of the mandible.
If one wants to eliminate possible recurrences after
treatment of an OKC it is of crucial importance that
one realizes where these recurrences originate from.
Considering the histopathology of the OKC with the
active basal layer of its epithelial lining, there is no
doubt that recurrences may arise if parts of the lining
are left behind. All efforts should, therefore, be aimed
at proper enucleation and elimination of possible
remnants of the cyst wall in case the cyst ruptures and
has to be removed piecemeal.
There is also the possibility that microcysts are
present in the connective tissue of the cyst wall and
that these are left behind after enucleation. The like-
lihood of this phenomenon is rather remote because
a prospective study has shown that these microcysts
are only sporadically found elsewhere in the cyst
wall.5 The majority of islands and microcysts are to be
1663
found in the mucosa to which the cyst is attached.
Some keratocysts show active budding of the basal
layer of the epithelial lining that may reach to the
periphery of the connective tissue wall and, there-
fore, may also be the source of a true recurrent
cyst.10-12
The third reason for a recurrent OKC is the devel-
opment of a new keratocyst from an epithelial island
or microcyst left behind in the mucosa. It is the
author’s opinion that this probably is by far the most
likely reason because most clinicians are keen to re-
move OKCs with great accuracy because they are
aware of the tendency toward high recurrence. Even
in a case where one has excised the overlying at-
tached mucosa, this may still be the reason for a
recurrence because it is not macroscopically visible
whether one has included the whole area where
these clusters of islands occur. In fact, the author has
described a few recurrences showing epithelial is-
lands in the overlying, attached mucosa, despite the
fact that the mucosa was excised at the first opera-
tion.5,9
Rational Surgical Approach
to Keratocysts
Because it is not the aim of this article to explain
the possible diagnostic procedures or to discuss the
differential diagnosis, the reader is referred to the
appropriate literature.13,14 When considering removal
of a keratocyst, however, it is important to keep in
mind the 3 possible reasons why an OKC could recur
(explained above). Therefore, treatment should aim
at elimination of possible vital cells left behind in the
defect. Whether they are from the original lining or
derived from microcysts in the wall, they are bound
to be located rather superficially in the defect. For this
reason a mild, not deeply penetrating, cauterizing
agent such as Carnoy’s solution should be enough to
do the job. In case the cyst was multilocular, care
should be taken to eliminate the bony septae as to
assure proper treatment of the whole bony cavity
with Carnoy’s solution. In case the cyst has pene-
trated through the lingual or buccal cortex, one may
use electrocauterization to avoid a recurrence in the
soft tissues. Identification and retraction of the lingual
nerve is recommended if the area is close to this
structure.
Elimination of the epithelial islands and microcysts
located in the overlying, attached mucosa should be
assured by excising this part of the mucosa. For that
reason the area needs to be identified. In the ascend-
ing ramus of the mandible it is almost always located
at the anterior aspect of the retrotrigonal area. It can
be palpated or defined using a needle. In the posterior
maxilla one must assume that the OKC is attached to
1664
FIGURE 1. A, Unilocular cyst in the ascending ramus of the mandible,
seemingly completely located in bone. B, Specimen including overly-
ing and partial attached mucosa (arrows). There was an anterior
fenestration in the bone. C, Histology of the specimen with area of
attachment of cyst wall to the mucosa. Arrow points to the cyst wall
(Hematoxylin-eosin stain; magnification ⫻ 2.) D, Magnification of the
area of attachment shows multiple epithelial islands. (Hematoxylin-
eosin stain; magnification ⫻ 25.) (Reprinted from Stoelinga.8)
Paul J.W. Stoelinga. The Best Possible Treatment Strategy for
OKCs. J Oral Maxillofac Surg 2005.
FIGURE 2. A, Original cyst distal to the impacted third molar in the left ascending ramus. B, Computed tomography scan showing 2 separate
recurrent cysts. The anterior cyst was attached to the mucosa, but it did not reveal epithelial islands or microcysts. The posterior cyst was completely
enclosed in bone. The recurrence was most likely caused by incomplete removal of the original cyst.
Paul J.W. Stoelinga. The Best Possible Treatment Strategy for OKCs. J Oral Maxillofac Surg 2005.
THE BEST POSSIBLE TREATMENT STRATEGY FOR OKCS
the thick mucosa of the tuberosity and one should
include that part in the excisional biopsy. Preferably,
the cyst with the attached overlying mucosa should
be removed in 1 piece (Fig 1). That presents the
pathologist an opportunity to examine that area for
the presence of epithelial islands and microcysts. De-
tailed descriptions on how to perform these opera-
tions for both the maxilla and mandible are presented
elsewhere.5,15
The overriding consideration of this approach is to
do as little damage as possible and yet to reduce the
chances of recurrence as much as possible. OKCs in
the dentate area that were not preoperatively diag-
nosed as such should be followed up without drastic
second interventions until a recurrence has become
apparent. These recurrences are always amenable for
relatively simple surgery without significant side ef-
fects.
Follow-Up
The recommended follow-up for OKCs is once a
year the first 5 years postoperatively. The literature
suggests that most recurrences will present the first 5
years after primary treatment. Because recurrences or
newly developed OKCs may also present late, a fol-
low-up once every 2 years thereafter seems a reason-
able policy.5 Of course, much depends on the sus-
pected cooperation of the patient and on geographic
and socioeconomic factors. Proper information on
why it is important to keep the patient under control
is important as well. It is the opinion of this author
that drastic operations, such as continuity resection,
are not warranted because of patient-bound factors.
The described method gives rise to a very low recur-
rence rate with little or no permanent morbidity.5
Recurrences
Recurrent OKCs may present as unilocular lesions
but also often present as a multilocular lesion or even
a separate unilocular lesion (Fig 2). The latter strongly
PAUL J.W. STOELINGA
FIGURE 3. A, Original, unilocular cyst in the right angle of the
mandible. B, Multilocular recurrence (arrows), diagnosed several
years later. C & D, Histologic specimens of marginal block resection
revealing several microcysts (arrow) in the mucosa overlying the recur-
rent OKC. Note the microcysts in the submucosa above the bone in D.
There were no microcysts in the surrounding bone. C is the main
recurrent cyst.
Paul J.W. Stoelinga. The Best Possible Treatment Strategy for
OKCs. J Oral Maxillofac Surg 2005.
suggests that parts of the lining have been left behind.
There is, however, very little systematic research per-
formed on the presentation and histology of recur-
rences. This author in the late 1960s performed mar-
ginal block resections on 5 recurrent OKCs to
ascertain their biological behavior.8 In none of these
cases was there any sign of microcysts hidden in the
surrounding bone. Instead, in all cases clusters of
epithelial islands and microcysts were seen in the
overlying, attached mucosa (Fig 3). Since that time we
have never treated recurrences by block resections,
but just removed them with the overlying attached
1665
mucosa. Of the 9 patients with a recurrence included
in the prospective study, 3 were located in the angle
of the mandible or ascending ramus and primarily
treated with excision of the overlying mucosa, while
the defect was treated with Carnoy’s solution. Micro-
cysts and epithelial islands were always seen in the
overlying attached mucosa.5 The recurrences oc-
curred, despite this treatment, which probably points
to incomplete excision of the overlying mucosa, be-
cause again these epithelial islands and microcysts
were seen in the recurrent cysts.
In short, recurring OKCs deserve the same treat-
ment as proposed for the primary OKC. There is no
reason for any more drastic treatment and certainly
no continuity resections.
Alternative Treatment Options
Considering the pathophysiology of the recurrent
OKCs there are 3 alternative modes of treatment that
deserve discussion. The first is decompression and
marsupialization as proposed by Brondum and
Jensen.16 This treatment can also be followed by re-
moval of the cyst wall after this has changed by
metaplasia.17,18 This treatment certainly has some-
thing to offer in case of large cysts particularly in old
or medically compromised patients. When the over-
lying, attached mucosa is removed it also eliminates
the source for newly formed cysts. There definitely is
a need for long-term, preferably prospective studies
to validate this treatment option. These studies should
include histology of the tissue removed and also bi-
opsies of the cyst wall that is left behind.
This author recently saw a dramatic recurrence as
the result of such treatment (Fig 4). This certainly is
not a completely safe method because one has no way
of knowing how the active basal layer of the original
cyst will behave.
The second option is the same as the described
protocol but instead of the use of Carnoy’s solution,
liquid nitrogen is used to freeze the defect.19,20 It
remains to be seen whether this treatment provides
the same good results as reported with Carnoy’s so-
lution. The method is also much more complicated. It
is also questionable whether it is needed to freeze
deeply in the bone for reasons explained above. As
the proponents of this method admit there might be
indications in selected patients.20
The third option is block resection, with or without
preservation of the continuity of the jaw.21,22 This is
a drastic method, which creates considerable morbid-
ity, particularly because reconstructive measures are
necessary to restore jaw function and esthetics.
Resection can only provide adequate results when it
includes the overlying attached oral mucosa. That en-
tails, in most cases, contamination from intraoral to
1666
extraoral. If the mucosa is not excised one can expect
recurrences even in the bone grafts used to bridge the
defect, as has been reported in several cases.5
One has to wonder whether such aggressive ther-
apy is warranted for a benign lesion that can be
managed reasonably well with relatively simple
means. This author has never found it necessary to
use such treatment and finds it hard to justify carrying
out continuity resections and complicated reconstruc-
tions in many patients to save a very few recurrences.
The latter remains to be seen, however, because for
this mode of treatment long-term follow-up studies
are also lacking.
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THE BEST POSSIBLE TREATMENT STRATEGY FOR OKCS
FIGURE 4. A, Computed tomography scan
showing OKC in the left maxillary sinus, al-
most completely obliterating the sinus. B, A
somewhat lower portion of the section show-
ing destruction of the lateral sinus wall. C &
D, Recurrent OKC posteriorly and anteriorly
(arrows). This OKC was primarily marsupial-
ized 4 years previously. A year later the
mucosa in the defect was removed, after
which it healed by secondary intention. Un-
fortunately, there was no histology of the sec-
ond intervention available.
Paul J.W. Stoelinga. The Best Possible
Treatment Strategy for OKCs. J Oral Max-
illofac Surg 2005.
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