The Saudi Dental Journal (2011) 23, 61–65

King Saud University

The Saudi Dental Journal

www.ksu.edu.sa
www.sciencedirect.com

REVIEW ARTICLE

Surgical treatment of keratocystic odontogenic tumour:

A review article
Walid Ahmed Abdullah

Maxillofacial Surgery Dept., College of Dentistry, King Saud University, Riyadh, Saudi Arabia
Oral and Maxillofacial Surgery Dept., College of Dentistry, Mansoura University, Egypt

Received 25 October 2009; accepted 23 December 2009

Available online 28 January 2011

KEYWORDS Abstract KCOT is one of the most aggressive odontogenic cysts with a high recurrence rate, this
Keratocystic odontogenic was explained histopathologically as it typically shows a thin, friable wall, which is often difficult to
tumour; enucleate from the bone in one piece, and have small satellite cysts within the fibrous wall. Multiple
Marsupialization; surgical approaches were introduced including decompression, marsupilization, enucleation with or
Surgical treatment without adjunct (Carnoy’s solution, enucleation) and resection. Depending on other studies KCOT
can be conservatively treated with enucleation and application of Carnoy’s solution or cryotherapy.
This can be used specially in the large lesions that when treated with resection, the continuity of the
jaw will be interrupted. This technique shows comparable results to other more aggressive tech-
niques.
ª 2011 King Saud University. Production and hosting by Elsevier B.V. All rights reserved.

Contents

1. Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 62
2. Decompression and marsupialization . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 62
3. Enucleation with and without adjuncts . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 63
4. Enucleation and treatment of the bony defect with Carnoy solution . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 63
5. Enucleation and liquid nitrogen cryotherapy . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 63

E-mail address: Waa007@gmail.com

1013-9052 ª 2011 King Saud University. Production and hosting by

Elsevier B.V. All rights reserved.

Peer review under responsibility of King Saud University.

doi:10.1016/j.sdentj.2011.01.002

Production and hosting by Elsevier

62
6. Block resection, with or without preservation of the continuity of the jaw . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 64
7. Summary . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 64
References . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 64
1. Introduction
Odontogenic keratocyst OKC is a developmental cyst that was
first described by Philipsen (1956). OKC is now referred to by
the World Health Organization (WHO) as a keratocystic
odontogenic tumour KCOT, and WHO defined it as ‘‘a benign
uni- or multi-cystic, intraosseous tumour of odontogenic ori-
gin, with a characteristic lining of parakeratinized stratified
squamous epithelium and potential for aggressive, infiltrative
behaviour (Barnes et al., 2005).
The KCOT is one of the most aggressive odontogenic cysts.
It can become quite large because of its ability for significant
expansion, extension into adjacent tissues and rapid growth
(Morgan et al., 2005). Different studies showed the incidence
of KCOT to be 3–11% of the odontogenic cysts (Chuong
et al., 1982; Payne, 1972). Generally, KCOT are solitary le-
sions unless they are associated with nevoid basal cell carci-
noma syndrome (Payne, 1972; Chirapathomsakul and
Sastravaha, 2006).
KCOT arises from cell rests of the dental lamina (Neville
et al., 1995). Histopathologically, KCOT typically shows a
thin, friable wall, which is often difficult to enucleate from
the bone in one piece, and have small satellite cysts within
the fibrous wall. Therefore odontogenic keratocysts often tend
to recur after treatment (Brannon, 1977; Gang et al., 2006).
Radiographically KCOT demonstrates a well-defined unilocu-
lar or multilocular radiolucency with smooth and often corti-
cated margins. In 25–40% of cases, there is an unerupted
tooth involved in the lesion. KCOT tend to grow in the anter-
oposterior direction within the medullary cavity of the bone
without causing obvious bone expansion causing its delayed
observation by the patients (Brannon, 1977; Gang et al.,
2006; Neville et al., 2002).
The treatment of the KCOT remains controversial. Treat-
ments are generally classified as conservative or aggressive.
Conservative treatment generally includes simple enucleation,
with or without curettage, or marsupialization. Aggressive
treatment generally includes peripheral ostectomy, chemical
curettage with Carnoy’s solution, cryotherapy, or electrocau-
tery and resection (Morgan et al., 2005; Meiselman, 1994;
Williams and Connor, 1994; Bataineh and Al Qudah, 1998;
Blanas et al., 2000).
The choice of treatment should be based on multiple fac-
tors; patient age, size and location of the cyst, soft tissue
involvement, history of previous treatment and a histological
variant of the lesion. The goal is to choose the treatment
modality that carries the lowest risk of recurrence and the least
morbidity (Rogerson, 1991; Williams, 1991).
2. Decompression and marsupialization
Decompression of a cyst involves any technique that relieves
the pressure within the cyst as this pressure is the way by which
the cyst grows by expansion. Decompression can be performed
W.A. Abdullah
by making a small opening in the cyst and keeping it open with
a drain (Pogrel, 2005; Eyre and Zakrzewska, 1985; Brondum
and Jensen, 1991).
Marsupialization, on the other hand, involves converting
the cyst into a pouch so the cyst is decompressed, but this is
a more definitive treatment than decompression as it exposes
the cyst lining to the oral environment. Mandibular cysts are
normally marsupialized into the oral cavity, while maxillary
cysts can also be marsupialized into the maxillary sinus or na-
sal cavity, as well as the oral cavity (Pogrel,2005, 2003; Seward
and Seward, 1969).
Decompression and marsupialization of cysts is probably
the earliest recommended treatment and was first suggested
by Partsch in the late 19th century. In many parts of the world,
marsupialization is still described as a Partsch I procedure (the
Partsch II procedure is enucleation and primary closure)
(Partsch, 1892, 1910).
Although decompression or marsupialization was not rec-
ommended as treatment for the KCOT by some authors, be-
cause it was thought that the pathologic tissue would be left
in situ (Pogrel and Jordan, 2004), decompression or marsupi-
alization has been recommended in a number of studies as a
technique that allows partial decrease in size in the KCOT
so that vital structures like teeth or the inferior alveolar nerve
can be preserved, then the KCOT was certainly enucleated
(Pogrel and Jordan, 2004; Partridge and Towers, 1987; Marker
et al., 1996).
Those authors who are against the use of marsupialization
or decompression for the treatment of KCOT depend on, that
this technique does not remove completely the whole cystic
covering, which would lead to a continuation of epithelial pro-
liferation and facilitate the recurrence (Bataineh and Al
Qudah, 1998; Maurette et al., 2006). Brondum and Jensen
(1991) reported a recurrence rate of 25% in 32 (OKCT) pa-
tients treated with decompression of the lesion. On the other
hand, other studies have shown that marsupialization of
KCOT can be followed by total resolution of the lesion with-
out any further surgery (Eyre and Zakrzewska, 1985; Pogrel
and Jordan, 2004; Hopper, 1982).
The marsupialization technique was described by Pogrel
(2005) as a window at least 1 cm in diameter is made into a
cyst, and an attempt is made to suture the cyst lining to the
oral mucosa. In the maxilla, the cyst is then often packed open
with the packing protruding through the opening. The packing
consists of iodoform gauze impregnated with bacitracin oint-
ment. When it is removed in the maxilla, the cavity is usually
self retaining and the patient needs to irrigate twice a day to
prevent food accumulation or closure of the fistula. In the
mandible, there is a greater tendency for spontaneous closure
of the fistula and reformation of the cyst, particularly in the
posterior mandible. In these cases, we have found that the
use of a nasopharyngeal anaesthesia tube suitably cut down
makes an excellent stent to keep the cyst open. Again, the cav-
ity is irrigated twice daily (Pogrel, 2005).
Surgical treatment of keratocystic odontogenic tumour: A review article
Studies have shown that when the OKCT is open to the
oral cavity by marsupialization, a number of changes occur
in the cyst lining. Histologically, the lining of OKCT is only
5 or 6 cells thick and tears easily on attempted enucleation;
which is one of the causes of the high recurrence rate. With
decompression or marsupialization, the lining appears to be-
come thicker and easier to enucleate, and histologically it does
appear to change and resemble normal oral mucosa, both with
routine histology and with immunohistochemistry (Pogrel and
Jordan, 2004; August et al., 2000).
Pogrel (2005) concluded that, decompression and/or marsu-
pialization has at least as high a success rate as the other more
aggressive treatments with lower morbidity and preservation
of important vital structures.
3. Enucleation with and without adjuncts
To enucleate is ‘‘to remove whole or clean, as a tumour from
its envelope.’’ Curettage is defined as ‘‘the removal of growths
or other material from the wall of a cavity (Giuliani et al.,
2006). Enucleation with and without various adjuncts has been
utilized for many years. Although enucleation/curettage has
the advantage over marsupialization of providing a complete
specimen for histopathologic analysis, it shows recurrence
rates as high as 62.5%, which is no longer an acceptable treat-
ment modality. This high incidence of recurrence is explained
by the thin, friable wall of the OKCT, which is often difficult
to enucleate from the bone in one piece, and the small satellite
cysts within fibrous wall.8,9 Many clinicians consider enucle-
ation and curettage as the minimal requirement in the treat-
ment of KCOT (Giuliani et al., 2006; Pindborg and Hansen,
1963).
Regarding curettage, clinicians have advocated mechanical
techniques (hand, rotary) alone or in combination with a
chemical solution (Carnoy’s) (Stoelinga, 2003) or cryosurgical
agents (liquid nitrogen) (Jensen et al., 1988; Schmidt and Po-
grel, 2001).
4. Enucleation and treatment of the bony defect with Carnoy
solution
As a result of the difficulty of enucleating the thin, friable
wall of the KCOT as one piece, and due to the small satel-
lite cysts, therefore, treatment should aim to eliminate the
possible vital cells left behind in the defect. For this reason
a mild, not deeply penetrating, cauterizing agent is used such
as Carnoy’s solution {consists 3 ml of chloroform, 6 ml of
absolute ethanol, 1 ml of glacial acetic acid and 1 g of ferric
chloride} (Morgan et al., 2005). This should be enough to
do cauterization of the remaining cells. In case the cyst
has penetrated through the lingual or buccal cortex, authors
described the use electrocauterization to avoid a recurrence
in the soft tissues (Stoelinga, 2003, 2005; Júnior et al.,
2007).
Other studies showed that, although the defect was treated
with Carnoy’s solution. Microcysts and epithelial islands were
always seen in the overlying attached mucosa. And so recur-
rence took place. So, the authors of these studies recom-
mended the complete excision of the overlying mucosa to
decrease the recurrence (Stoelinga, 2005, 2001). Morgan et
al. (2005) also reported in their study that the treatment with
63
Carnoy’s solution did not show a significant association with
recurrence. Yet, Voorsmit et al. (1981) reported a decreased
recurrence rate following treatment with enucleation and Car-
noy’s solution (2.5%) compared with enucleation alone
(13.5%).
According to (Blanas et al., 2000) enculation of KCOT fol-
lowed with application of Carnoy’s solution appears to be the
least invasive procedure with the lowest recurrence rate. And
they reported that adding Carnoy’s solution to the cyst cavity
for 3 min after enucleation results in a recurrence rate compa-
rable to that of resection without unnecessarily aggressive
surgery.
The effects of Carnoy’s solution on the inferior alveolar
nerve were first reported by Frerich et al. (1994). The authors
did not observe axonal damage during the first three minutes
of direct application. In contrast, another important study,
Wolgen et al. (1999), noted that the alterations in neural con-
ductivity developed after 2 min of direct application, with few
signs of recovery after two weeks of follow-up. However,
Júnior et al. (2007), reported that when a proper protocol is
followed, the chemical treatment of the nerve can be accom-
plished without permanent functional damage.
5. Enucleation and liquid nitrogen cryotherapy
Theoretically, the ideal treatment for the KCOT would be enu-
cleation or curettage followed by treatment of the cavity with
an agent that would kill the epithelial remnants or satellite
cysts. In addition, the osseous framework should be left intact
to allow for osteoconduction. Liquid nitrogen has the ability
to devitalize bone in situ while leaving the inorganic frame-
work untouched, as a result of this, cryotherapy has been used
for a number of locally aggressive jaw lesions, including
KCOT, ameloblastoma and ossifying fibroma (Emmings
et al., 1966; Bradley and Fisher, 1975; Pogrel, 1993). Cell death
with cryosurgery occurs by direct damage from intracellular
and extracellular ice crystal formation plus osmotic and elec-
trolyte disturbances (Schmidt and Pogrel, 2001; Rosen and
Vered, 1979).
According to Schmidt and Pogrel (2001) the standardized
technique is as follows, the initial step in management of the
lesion is enucleation of the cyst. The surrounding tissues are
then protected with sterile wooden tongue blades and gauze,
and the cavity is sprayed with liquid nitrogen twice for
1 min, with a 5-min thaw between freezes. Bone graft can in-
serted in the defect simultaneously, and then mucosa is closed
with watertight sutures.
The advantages of liquid nitrogen over alternative methods
of devitalizing the tissue beyond the visible lesion of the margin
are that (1) the bone matrix is left in place to act as a clean
scaffold for new bone formation, (2) a bone graft can be placed
immediately to accelerate healing and minimize the risk of a
pathologic fracture, and (3) decrease of bleeding and scarring.
However, because of the difficulty in controlling the amount of
liquid nitrogen applied to the cavity, the resultant necrosis and
swelling can be unpredictable (Pogrel, 1993; Salmassy and
Pogrel, 1995). The recurrence rate following enucleation and
liquid nitrogen cryotherapy has been reported at 3–9%
(Pogrel, 2005; Schmidt, 1999).
When the liquid nitrogen cryotherapy is given around the
inferior alveolar nerve, it is affected, and patients will suffer
64
paraesthesia or anaesthesia. However, the axon sheaths are left
intact and nerve regrowth is normal such that most patients
obtain partial or complete return of sensation in 3 months
(Schmidt, 1999, 2003).
6. Block resection, with or without preservation of the continuity
of the jaw
Resection refers to either segmental resection (surgical removal
of a segment of the mandible or maxilla without maintaining
the continuity of the bone) or marginal resection (surgical re-
moval of a lesion intact, with a rim of uninvolved bone, main-
taining the continuity of the bone) (Blanas et al., 2000;
Kondelland Wiberg, 1988) which is an extreme technique, that
results in considerable morbidity, particularly because recon-
structive measures are necessary to restore jaw function and
aesthetics (Stoelinga, 2005). Stoelinga (2005), wonders whether
such aggressive therapy is warranted for a benign lesion that
can be managed reasonably well with relatively simple means.
In a systematic review done by Blanas et al. (2000), the
authors reported that resection was found to have the lowest
recurrence rate (0%) but the highest morbidity rate, while
enculation with application of Carnoy’s solution can result
in a recurrence rate comparable to that of resection without
unnecessarily aggressive surgery.
Multiple studies concluded that keratocysts might be trea-
ted with a conservative approach, the only disadvantages being
the extended therapeutic time. Extensive resection of the man-
dible with its attendant morbidity may be too radical for large
KCOT and even an overtreatment (Giuliani et al., 2006;
Marker et al., 1996; Nakamura et al., 2002).
7. Summary
KCOT is one of the most aggressive odontogenic cysts with a
high recurrence rate. Multiple surgical approaches were intro-
duced including decompression, marsupilization, enucleation
with or without adjunct (Carnoy’s solution, cryotherapy),
and resection. Depending on other studies KCOT can be con-
servatively treated with enculation and application of Carnoy’s
solution or cryotherapy. This can be used specially in the large
lesions that when treated with resection, the continuity of the
jaw will be interrupted. This technique shows comparable re-
sults to other more aggressive techniques.
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