MAXILLOFACIAL SURGERY

LARGE ODONTOGENIC KERATOCYST OF THE MANDIBLE: A COMBINED INTRA/EXTRA

Case Rreports
ORAL APPROACH FOLLOWED BY ENUCLEATION
Maximilien Vercruysse1a*, Patricia D’Haeseleire2b, Sidney Kunz2b, Bart Lutin3c, Constantinus Politis1,4d

1
Department of Oral and Maxillofacial Surgery, University Hospitals Leuven, Leuven, Belgium
2
Departments of Oral and Maxillofacial Surgery, AZ Groeninge, Kortrijk, Belgium
3
Department of Radiology, AZ Groeninge, Kortrijk, Belgium
4
Department of Oral and Maxillofacial Surgery, University Hospitals Leuven, Leuven, Belgium; OMFS IMPATH Research Group, Department of Imaging &
Pathology, Faculty of Medicine, University Leuven, Leuven, Belgium

a
MD, Trainee
b
MD, DDS
c
MD
d
MD, DDS, MHA, MM, PhD, Professor, Head
OPEN ACCESS This is an Open Access
ABSTRACT DOI: https://doi.org/10.25241/stomaeduj.2019.6(2).art.6 article under the CC BY-NC 4.0 license.

Aim: An odontogenic keratocyst (OKC) of the mandible is a benign intraosseus Peer-Reviewed Article
lesion of odontogenic origin characterized by a high recurrence rate. In this case Citation: Vercruysse M, D’Haeseleire P, Kunz S,
report, we highlight the challenging diagnosis and propose a potential treatment Lutin B, Politis C. Large odontogenic keratocyst
of the mandible: A combined intra/extra oral
for an extensive OKC with lingual expansion. approach followed by enucleation. Stoma Edu J.
Summary: A 26-year-old male with an OKC in the ramus of the right mandible near 2019;6(2):129-137

the second and third molars was treated by a combined intra/extra- oral approach. Received: June 03, 2019
Revised: June 13, 2019
A reconstruction plate was adapted and fixed by extra-oral submandibular access, Accepted: June 20, 2019
followed by intra/extra-orally executed enucleation. Published: June 21, 2019

Key learning points: The combined intra/extra oral approach seems a reasonable *Corresponding author:
Maximilien Vercruysse, Jakob van Maerlantstraat 7,
technique for the treatment of similar extensive OKC’s in order to avoid pathological 8500 Kortrijk, Belgium, , Telephone: +32 (0)
fractures as well as guaranteeing total removal of the lesion. 16.341780, Fax: +32 (0) 16 3 32437,
e-mail: [email protected]
Keywords: Odontogenic keratocyst; Mandible; WHO classification; Treatment;
Copyright: © 2019 the Editorial Council for the
Intra/Extra-oral approach. Stomatology Edu Journal.

1. Introduction their histological characteristics as they present with

Odontogenic keratocysts (OKC’s) of the mandible a cystic architecture but the epithelial lining has
have been of interest since first presented by a proliferative potential. Histologically the OKC is
Philipsen in 1956 [1,17]. OKC’s are considered to lined by a regular, keratinized, stratified, squamous
be benign intraosseous lesions of odontogenic epithelium which is 5-10 layers thick and has no rete
origin. Evidence suggests that OKC’s are to be found ridges. The parakeratin surface typically appears
posterior to the third molars if offshoots of dental corrugated, the basal layer is well defined and
lamina remnants are involved. OKC’s occupying the frequently palisaded with hyperchromatic nuclei
ascending ramus, often present with epithelial island and focal areas showing reversed nuclear polarity.
in the mucosa overlying the cyst [1,2,36]. The lesions [3,7,8,41]. OKC’s do not cause metastases or lymph
are characterized by a tendency to grow within node invasion and do not form tumoral masses. The
the medullar space of the mandible/maxilla and WHO reclassification could lead to confusion and
have a relatively high recurrence rate. The potential decreased alertness concerning this lesion. [2,3]
aggressiveness of OKC’s is reflected by their potential When consulting the literature, one must be aware
to extend into soft tissues and bone, although rare. of the different appellations. OKC has a broad age
In contrast to ameloblastomas, OKC’s cause bone predominance, with a peak incidence in the 2nd-
destruction but do not invade through an intact 3th decades. In addition, the incidence is higher in
periosteum [1,33]. This was questioned by Stoelinga, male patients, with a male to female ratio of 3:1 [4].
who cited a few rare instances of keratocysts with Presentation occurs twice as often in the mandible
soft tissue penetrance [2,38,43,44,45]. (70-75%) as in the maxilla. The posterior body
OKC’s exhibit characteristics of both cysts and and ascending ramus of the mandible are typical
benign tumors and were reclassified as odontogenic locations. As a consequence, all cysts in the posterior
tumors by the World Health Organization (WHO) in mandibular area have to be treated as if they were
their 2005 Classification of Head and Neck Tumors. OKC’s of unicystic ameloblastomas. However, OKC’s
In the 2017 classification, the WHO re- categorized can also occur in the dentate area of the mandible
the keratocystic odontogenic tumor into the “cyst” or maxilla, mimicking ordinary odontogenic cysts.
category, and the term “odontogenic keratocyst” Signs and symptoms can be subtle, but the typical
has been used since. This ambiguity is reflected by presentation is pain, local swelling, infection and

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Case Rreports

Figure 1. Panoramic radiograph depicting a large cystic mass in the right mandible.

cellulitis [1,4,6,12,39]. OKC’s are often asymptomatic, change [5,12,13,34]. The conservative therapies
probably because they grow in the anteroposterior consist of enucleation, but it is generally agreed upon
direction into the intramedullary space, with little that additional measures for enucleation are crucial
cortical expansion. When these lesions reach a to minimize recurrence. The three main techniques
large size or perforate the cortical bone, they will are peripheral ostectomy, chemical curettage with
often become symptomatic [9,21,40]. Of all the application of Carnoy’s solution, and cryotherapy. In
cases, 1:3 will be related to an unerupted tooth; the literature, there is immense variability in the use
the relationship between OKC’s and impacted third of additional techniques, and studies have shown a
molars is 10-15%. In addition, growing OKC’scan similar efficacy between peripheral ostectomy and
dislocate away associated teeth [23,24]. OKC’s Carnoy’s solution. Superior outcomes of cryotherapy
represent approximately 10% of all cysts of the have not been described [6,12,13,21,26]. Lesions
jaw. They are frequently discovered incidentally exceeding 3 cm are not fully suitable for enucleation.
by radiographic examination. On radiography, the In these cases, decompression by marsupialization
OKC presents itself as a well-defined radiolucent can reduce the lesion size. The literature has
area. They are commonly unilocular, more rarely not shown an increased risk of recurrence after
multilocular. Often they are not distinguishable from decompression [25-27]. It seems advisable to treat
regular odontogenic cysts [6,9]. Specific clinical and each cyst in the mandibular third molar region with
radiographic characteristics that point to a certain possible extension into the ascending ramus by
diagnosis pre-operatively are lacking. enucleation, with excision of the overlying, attached
The Gorlin Goltz syndrome has to be considered mucosa if possible. Subsequently, treatment with
if a patient presents with multiple OKC’s. This is an Carnoy’s solution or liquid nitrogen has to be
autosomal dominant multisystem disease that leads considered. The targeted treatment of the OKC
to multiple OKC’s, as well as several nevoid basal cell seems achievable, as multiple mutations have been
carcinomas, palmar or plantar pits, calcification of elucidated. Mutations in the PTCH gene or the gene
falx cerebri, and skeletal abnormalities. The Gorlin encoding smoothened protein that enhances sonic
Goltz syndrome is associated with mutations in the hedgehog signaling (SMO) can be drug targets.
PTCH gene situated on 9q22.3-q31, with described The antimetabolite 5-fluorouracil may affect the
mutation rates of 80-90% [1,7,8,16,19,20]. sonic hedgehog pathway and has shown lower
The established treatment modalities for OKC’s can postoperative morbidity in studies [25,32].
be divided into radical and conservative treatment The best treatment for an OKC of the mandible is still a
options. The radical treatment consists of en bloc matter of debate, as diagnosis is not straightforward.
resection with negative margins of the segment With this case report, we demonstrate the ambiguity
and has been associated with a recurrence rate of in diagnosis and existing treatment modalities
approximately 0%. Knowing the benign nature of and propose treatment for a large OKC in the right
this lesion and the morbidity of en bloc resection, mandible with lingual expansion.
this technique has to be reserved for wide, extensive
lesions. A retrospective study showed that the main 2. Case presentation
reasons for radical treatment are invasion of the A 26-year-old Caucasian male was referred to the
pterygoid muscles and the presence of malignant Department of Oral and Maxillofacial Surgery

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 LARGE ODONTOGENIC KERATOCYST OF THE MANDIBLE: A COMBINED INTRA/EXTRA ORAL APPROACH
FOLLOWED BY ENUCLEATION

Case Rreports
a
b

Figure 2. Cone beam computed tomography

(a) of the right mandible showing a unilocular cystic mass (42 mm x 37 mm)
(b) and 3D volume rendering . The green line indicates the inferior alveolar nerve channel

complaining of a pain in the right ramus of more suggestive of a benign odontogenic cyst rather
the mandible, without swelling of this region. than an OKC because of the lack of high intensity on
Radiographically, there was a unilocular radiolucent T1-weighted imaging before contrast administration,
lesion with defined borders located in the right correlating with ortho/parakeratin or hemorrhage
mandibular ramus, measuring 42 mm associated in keratocysts. An ultrasound-guided fine needle
with the retained third molar (Fig. 1). He had no biopsy was performed, which was suggestive of
relevant medical history. an inflammatory follicular or radicular cyst, rather
Clinical examination did not reveal abnormalities. than a keratocyst or ameloblastoma. The surgeons
No regional lymph nodes were palpable and intra- preferred to do a fine needle biopsy instead of an
oral examination revealed no swelling. Cone beam incisional biopsy as it is less invasive and incisional
computed tomography (Fig. 2) showed an expansive, biopsies have the potential for sampling error [42].
well defined, unilocular cystic lesion longitudinally A provisional diagnosis of radicular/inflammatory
in the right mandible near the second molar and odontogenic cyst was determined. Two weeks later,
closely adhering to the third unerupted molar. surgery was performed under general anesthesia.
The lesion had a radiopaque border but caused First, a submandibular neck incision (10 cm) was
thinning of the lingual cortex of the mandible with made, followed by dissection and local excision
destruction of the cortex at the medial and caudal of the enlarged submandibular lymph nodes. The
edge of the mandible. Peri-apical resorption was periosteum was incised over the mandible inferior
apparent lateral and posterior to the apex of the edge and the mental foramen located. A Martin 2.3
second mandibular molar with destruction of the plate was adapted and fixated with 7-9 mm screws
two roots. Neither calcification nor a periosteal (Fig. 4). The extra oral approach was preferred due to
reaction wasere identifiable. the size of the lesion, the necessity of wide exposure
On MRI (Fig. 3), were the lesion had homogenous high and the fear for a pathological fracture.
signal intensity on T2-weighted imaging, low signal Subsequently, teeths 47 and 48 were extracted after
intensity on T1-weighted imaging, and homogenous preparation of a buccolingual mucoperiosteal flap.
enhancement of the cyst wall after administration of Via combined intra/extra-oral access, the cystic mass
intravenous gadolinium. These characteristics were was exposed. A local posterior gingival resection

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FOLLOWED BY ENUCLEATION

Case Rreports

a b c
Figure 3. Magnetic resonance imaging.
(a) T2-weighted imaging showing homogenous high signal intensity.
(b) T1-weighted imaging before and
(c) after administration of gadolinium, showing low signal intensity and homogenous enhancement of the cyst wall.

a b

Figure 4. Peri-operative images.

(a) Removed cystic mass and (b) plate osteosynthesis of the right mandible.

was performed by intra-oral access. The inferior Rigorous follow-up was organized with a panoramic
alveolar nerve was released of extensive adhesions radiograph, cephalometric X-ray, and cone beam
over 4 cm without causing a continuity defect. computed tomography after 6 months, showing no
Local bone trepanation was performed to facilitate recurrence. The cone beam computed tomography
enucleation while safeguarding bony continuity showed a favorable ossifying pattern (Fig. 7).
of the lower border. After flushing, achieving Patient history and clinical examination indicated a
hemostasis, and applying a tetracycline suspension favorable healing process (Fig. 8).
in the intramandibular cavity, intra-oral suturing was
performed. The submandibular incision was closed 3. Discussion
after placing a drain. A postoperative panoramic The OKC is an expansive, solitary, mostly unilocular
radiograph showed adequate positioning of the (approximately 80% of cases) jaw lesion thought
reconstruction plate at the lower border of the right to arise from remnants of the dental lamina. The
mandible (Fig. 5). active epithelial lining and high proliferation rate
One week after the procedure, the pathology analysis reflect a potentially aggressive growth pattern.
was complete. The microscopic characteristics There is a high recurrence rate, between 25 and 60%,
of the H&E stained section showed Malpighian linked to the dental lamina origin and its epithelial
epithelium with marked peripheral palisading of islands. Epithelial islands, or micro cysts, can be
the stratum basale. Parakeratosis and orthokeratosis found in the overlying mucosa in almost 50% of
were present with characteristic corrugations of cases. Research on recurrent OKC’s has shown that
the superficial layer. Some epithelial neutrophilic epithelial islands or micro cysts are present in almost
granulocytes were present and part of the cystic wall 100% of recurrent cases [36,37]. The high recurrence
was replaced by inflammatory granulation tissue rate is attributed to the parakeratotic character of
(Fig. 6). Taken together, these findings confirmed the OKC’s[8-10]. Most cases of recurrence present within
diagnosis of OKC. 5 years of treatment, but recurrence after more than

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 LARGE ODONTOGENIC KERATOCYST OF THE MANDIBLE: A COMBINED INTRA/EXTRA ORAL APPROACH
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Case Rreports
Figure 5. Postoperative panoramic radiograph after removal of the cystic mass and plate osteosynthesis at the lower border of the the right mandible.

is rare with OKC’s, in contrast to ameloblastomas,

with which root resorption is seen frequently. In our
case, the radiolucent lesion caused thinning of the
lingual cortex of the mandible and root resorption of
the second molar. This highlights that a diagnosis of
OKC cannot be made using imaging techniques only.
When evaluating a cystic lesion of the mandible,
MRI is a complementary technique to cone beam
CT and can be applicable in cases of soft tissue
involvement. MRI is superior in illuminating the soft
tissue involvement, and a number of studies argue
that MRI is crucial in discriminating ameloblastomas
from OKC’s [28,29]. As such, cone beam CT and
MRI, in select cases, are crucial in the diagnosis of
Figure 6. Biopsy of the cystic mandibular mass. Note the wall of the OKC’s. Some lesions can be indistinguishable from
keratocyst with notable palisading and slightly wavy surface.
other osteolytic jaw lesions on imaging; therefore,
histopathology is always necessary for a definitive
10 years has been described. Higher recurrence diagnosis. A prospective study of 82 OKC’s reported
rates have been reported with multilocular lesions that 40% were not suspected before surgery [1,22,24].
and in patients with the Gorlin Goltz syndrome [8- The surgeons chose not to execute a preoperative
10,14,18,25]. Our patient consulted with aggravating incisional biopsy as it can cause inflammation and
pain in the right ramus of the mandible and interfere in the histopathological analysis of the pos-
dysgeusia. The dysgeusia was probably caused surgical specimen.
by a fistula between the lesion and the oral cavity, The differential diagnosis consisted of OKC,
through which keratin could enter the mouth. ameloblastoma, dentigerous cyst, or radicular cyst.
Panoramic radiography is helpful in the preliminary Clinically, these lesions can be indistinguishable, but
assessment, as an OKC will present as a defined on imaging they each have typical characteristics. An
radiolucent lesion, mostly unilocular, with smooth OKC can be a unilocular or multilocular lesion with
and corticated margins [1,3]. Panoramic radiography few septa and minimal buccolingual expansion. An
will show the possible relationship with (impacted) ameloblastoma typically presents as a multilocular
teeth. Cone beam CT should be the next step, as it lesion with root resorption and high tendency for
is considered superior to panoramic radiography buccolingual expansion. A dentigerous cyst will
because of the possibility to detect bone cortical present as a unilocular cyst around the crown of
perforations in the region where the overlying oral an impacted tooth with possible buccolingual
mucosa is adhered to the OKC. It is a valuable tool in expansion and without septa. Lastly, a radicular/
detecting all dimensions of the lesion with a better inflammatory cyst will present as a unilocular lesion
identification of the anatomical structures. It has a connected to the apex of a non-vital tooth [1,24].
high spatial resolution but poor contrast resolution, Fine needle aspiration biopsy can be considered
which is not suitable for soft tissue discrimination a safe technique, offering a possibly valuable
[1]. Cortical expansion in OKC’s will occur more often contribution to pre-operative diagnosis. However, it
lingually than buccally. Furthermore, root resorption can be misleading due to inflammation in OKC’s.

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Case Rreports

b c
Figure 7. Follow-up 6 months after surgery.
(a) Panoramic radiograph, (
b) cephalometric X- ray, and
(c) cone beam computed tomography.

In addition, a biopsy of an unrepresentative area could not be neglected. The expanded resorption
of the lesion can be misleading. The result can be of the lingual cortex with broad fenestration could
indicative, but a negative result can never rule out induce a pathological fracture after enucleation
a possible diagnosis of OKC until investigation of with bone trepanation for removal of teeths 47
the final resection specimen. Baykul et al. showed and 48. Therefore, a combined intra/extra-oral
a correlation of 89.95% between cytological and technique was applied. By adapting and fixating a
histopathological diagnosis for cystic lesions in the plate before performing the enucleation, we tried
maxillofacial region [22,29,30,31]. to avoid a pathological fracture. With this degree of
With this in mind, diagnosis and subsequent extended osteolysis, we felt fixation of a plate was
treatment of OKC’s poses a challenge. The objective necessary. What if mandibular continuity resection
is to reduce the recurrence risk as much as possible, is necessary peri-operatively? Then, the mandibular
while minimizing morbidity. This delicate balance bony contour would be guaranteed by using the
has led to heavy international debate, and no reconstruction plate. As no clear arguments for OKC
consensus on treatment has been reached. were present pre-operatively and peri-operatively,
Conventional treatment modes were described the surgeons decided not to use Carnoy’s solution
in this article, however, for this specific case an because of the location of the inferior alveolar nerve
adapted treatment was performed. Because of and the potential neurotoxicity, saving soft tissue as
the size of the lesion and its lingual expansion, much as possible.
there was uncertainty about mandibular stability The described technique seems suitable for lesions
after enucleation. The location of the lesion with of this size when pathological fractures are likely.
immediate connection to the deep neck soft tissues It provides an elegant way to provide mandibular

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 LARGE ODONTOGENIC KERATOCYST OF THE MANDIBLE: A COMBINED INTRA/EXTRA ORAL APPROACH
FOLLOWED BY ENUCLEATION

Case Rreports
Figure 8. Clinical pictures 6 months after surgery

continuity while executing enucleation. We are Ethical approval

aware that the short follow-up is a limitation of this Not applicable. The present study is not a research
case report. study.

4. Conclusion Consent
With this case report, we tried to point out the Written informed consent was obtained from the
difficult diagnosis of OKC and, by extension, all patient for the publication of this case report and the
radiolucent lesions of the mandible. Although accompanying images.
the lesion does not always present with its typical
features, the possibility of OKC must be taken into Author Contributions
account when setting up a treatment protocol. MV: leading author of the manuscript. PD: critically
Given the presented case, the combined intra/ revising the manuscript. SK: critically revising the
extra oral approach is a reasonable technique for manuscript. BL: giving more insight in radiologic
the treatment of similar OKC’s in order to avoid aspect of the case report. CP: critically revising the
pathological fractures as well as guaranteeing total manuscript.
removal of the lesion.
Acknowledgments
Conflicts of interest The authors would like to thank all those who were
None involved in the construction of this article.

Funding
This research did not receive any specific grant from
funding agencies in the public, commercial, or not-
for-profit sectors.

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Maximilien VERCRUYSSE
MD, Trainee
Department of Oral and Maxillofacial Surgery
University Hospitals Leuven
Leuven, Belgium

CV
Maximilien Vercruysse is a trainee at the Department of Oral and Maxillofacial Surgery, University Hospitals Leuven, Leuven,
Belgium.

Questions
1. What percentage of the cases present the odontogenic keratocyst in the mandible
(approximately)?
qa. 10 %;
qb. 20 %;
qc. 40 %;
qd. 70 %.

2. Which syndrome is strongly associated with a higher rate of OKC’s?

qa. Pierre robin syndrome;
qb. Gorlin Goltz syndrome;
qc. Neurofibromatosis;
qd. Treacher Collins Syndrome.

3. Recurrence rate after 5 year follow up of a surgically treated non-syndromal keratocyst

is approximately?
qa. 0-5%;
qb. 0-20%;
qc. 20-40%;
qd. 60-80%.

4. Male to female ratio of OKC’s can be estimated at?

qa. 3:1;
qb. 1:1;
qc. 1:2;
qd. 1:3.

Stomatology Edu Journal 137