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REVIEW PAPER

Odontogenic Cysts and Tumors


Dana Rioux-Forker, MD,* Allyson C. Deziel, MD,*
Larry S. Williams, DDS,† and Arshad R. Muzaffar, MD*

Abstract: Odontogenic cysts and tumors are mandibular and maxillary lesions that
DIAGNOSIS AND TREATMENT
occur across all patient demographics across age, sex, race, and social economic Preoperative clinical assessment and imaging are vital for devel-
status, as altered remnants of dental development. They may be incidental findings oping a provisional diagnosis of any jaw lesion. Initial imagining typically
from routine imaging in any office or found through workup for craniofacial sur- includes an x-ray series or panorex. Often, plain radiographs are insuf-
gery or injury. Many of these patients present with asymptomatic lesions, whereas ficient, and more advanced imaging including cone beam computed to-
others may be symptomatic. In this article, we review the literature on the most mography (CT), CT, magnetic resonance imaging, or ultrasound is needed
common odontogenic tumors and cysts and discuss their presentation, their de- to further characterize the lesion, surrounding tissue, or even vascularity.
fining traits, and how to approach diagnosis and definitive management. Chapelle et al6 and Stoelinga7 reviewed jaw tumors and cysts and
Key Words: odontogenic cyst, odontogenic tumor, periapical cyst, radicular cyst, developed recommendations based on provisional diagnoses and treat-
dentigerous cyst, eruption cyst, paradental cyst, botryoid cyst, gingival cyst, ment outcomes (Fig. 1). They recommended avoiding preoperative bi-
glandular odontogenic, ameloblastoma, keratocytic odontogenic tumor, opsy in unicystic lesions, unless a solid tumor is expected. Enucleation
gorlin cyst, myxoma, odontoma with histologic assessment was the recommended initial treatment of
unicystic lesions, unless the lesion was in the mandibular third molar
(Ann Plast Surg 2019;82: 469–477) region with extension onto the ramus. This extension tends to be more
consistent with odontogenic keratocysts and ameloblastomas. In the
T he World Health Organization classifies neoplasms of the mandible
as odontogenic cysts, nonodontogenic cysts, and pseudocysts.1,2 A
true cyst is defined as an epithelial lined cavity, whereas pseudocysts
past, they have recommended adding mucosal excision with applica-
tion of Carnoy's solution or liquid nitrogen, which will overtreat some
odontogenic or pseudocysts but may spare morbidity in odontogenic tu-
lack an epithelial lining.3 Odontogenic and nonodontogenic cysts are mor patients. Unfortunately, obtaining Carnoy's solution has become
differentiated by the origin of their epithelial lining. Odontogenic cyst difficult because chloroform has been classified as a carcinogen and
epithelium originates from the tooth forming organ. These include the there is often soft tissue damage that can be seen with liquid nitrogen.
reduced enamel epithelium, rests of Malassez, and rests of Serres.2,3 A suggested alternative method involves decompressing the lesion with
Odontogenic cysts can be further classified into developmental cysts a drain followed by surgical enucleation, after which long-term follow-
(gingival, dentigerous, odontogenic keratocysts, lateral periodontal, cal- up is critical because recurrent cases have been reported. Multiloculated
cified odontogenic, and glandular odontogenic cysts) or inflammatory and multicystic tumors were recommended to be treated more aggres-
cysts (radicular, paradental, residual cysts).1 sively. They recommended performing an incisional biopsy to differen-
The true prevalence and distribution of odontogenic cysts and tu- tiate ameloblastomas from odontogenic keratocytes in multilocular or
mors is not known. Odontogenic cysts are thought to comprise the vast multicystic lesions with expansion. Those without expansion were di-
majority of jaw cysts, accounting for approximately 90%.4 Johnson agnosed by aspiration.6,7 Final pathologic diagnosis then directs
et al5 did a systematic literature review and found that odontogenic cysts any further lesion specific treatment. Long-term follow-up and repeat
were 2.25 times more common than odontogenic tumors. They reported imaging are necessary given the high recurrence rates and possible
that the most common cyst found was the radicular cyst, which comprised malignant conversion.
54.6% of the odontogenic cysts. The next most common types found were
the dentigerous cysts comprising 20.6% and the keratocystic odontogenic
tumor (KCOT) comprising 11.7% of the reported odontogenic cysts.
Other cysts including calcifying odontogenic cysts, lateral periodontal Odontogenic Cysts
cysts, eruption cysts, botryoid odontogenic cysts, and residual cysts Periapical (Radicular) Cyst
were reported infrequently enough to limit further analysis. Combined,
these were thought to comprise less than 10% of odontogenic cysts.5 These are the most common odontogenic cysts (52%–70% of all
Johnson et al5 also reported on the incidence of odontogenic tu- jaw cystic lesions) (Fig. 2) and are commonly found in the maxilla in about
mors in the literature. They found that ameloblastomas were the most 60% of cases (Fig. 3).3,8 They are usually asymptomatic unless they be-
prevalent at 37.9%, followed by KCOT at 36.6%, odontogenic myxomas come infected. The epithelium of periapical cysts develops from the rests
at 3.8%, adenomatoid odontogenic tumor at 3.2%, and ameloblastic fi- of Malassez, from an inflammatory process associated with a nonvital
bromas at 1.1%. Finally, odontoma classification remains disputed, with tooth (caries, trauma, periodontal space extension), and a precursor
some including them as odontogenic tumors with a frequency of up periapical dental granuloma.1,9,10 These cysts typically present with a
to 45.8%.5 unilocular radiolucency at the apical portion of a tooth root. However,
they can develop lateral to a tooth system (lateral radicular cyst). They
typically have well-defined borders and are often under 1 cm in diame-
ter.11 The sensitivity and specificity of radiographs in detecting periapical
Received March 7, 2018, and accepted for publication, after revision October 5, 2018.
From the *Division of Plastic Surgery, University of Missouri; and †Department of Oral
cysts are low (33.3% and 61.5%, respectively).12
Surgery, Truman VA, Columbia, MO. Treatment for these lesions is simple enucleation given their be-
Conflicts of interest and sources of funding: none declared. nign pathology. These may resolve with endodontics (root canal therapy);
Reprints: Dana Rioux-Forker, MD, Division of Plastic Surgery, University of Missouri, however, if a periapical cyst is incompletely removed; it may develop
One Hospital Dr, Columbia, MO 65212. E-mail: Riouxforkerd@Missouri.edu.
Copyright © 2019 Wolters Kluwer Health, Inc. All rights reserved.
into progressively expanding residual cysts and result in significant bone
ISSN: 0148-7043/19/8204–0469 destruction. Some have hypothesized that periapical cysts may sponta-
DOI: 10.1097/SAP.0000000000001738 neously resolve once apical periodontitis is treated.1

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Rioux-Forker et al Annals of Plastic Surgery • Volume 82, Number 4, April 2019

FIGURE 1. Odontogenic cyst and tumor initial management flow chart. Reprinted with permission from Elsevier from Chapelle KA et al.
Copyright Elsevier. All permission requests for this image should be made to the copyright holder.

Dentigerous (Follicular) Cyst tooth follicle and prevents tooth eruption.13 These cysts are associated with
These are the second most common developmental odontogenic the crown of an unerupted tooth, developing tooth, or an odontoma. The
cysts, which make up about 20% of all mandibular cysts (Fig. 4).1 third molars and maxillary canines are most frequently involved. Dentigerous
Dentigerous cysts are noninflammatory and form when fluid accumu- cysts are commonly diagnosed in teenage years and early adulthood and are
lates between a tooth crown and the enamel epithelium, which dilates the typically asymptomatic, unless they cause local swelling or become infected.

FIGURE 2. Odontogenic cyst and tumor chart.

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Annals of Plastic Surgery • Volume 82, Number 4, April 2019 Odontogenic Cysts and Tumors

FIGURE 5. Eruption cyst. Reprinted with permission from


Wolters Kluwer from Nagaveni NB et al.17 Copyright Wolters
Kluwer. All permission requests for this image should be made
to the copyright holder.

Eruption Cysts
FIGURE 3. Periapical cyst. Eruption cysts are a form of dentigerous cyst associated with
erupting teeth (Fig. 5). These cysts form at the time of deciduous or per-
Radiographically, patients have a unilocular radiolucency associ- manent tooth eruption and can even be seen in newborns. They develop
ated with an unerupted tooth. These cysts typically have well-demarcated from the crown of a tooth when it is within the soft tissue.2 Eruption cysts
borders and display an origin from the cemento-enamel junction.14 Iden- overlie the tooth, presenting with a swelling on the alveolar ridge, often hav-
tifying the crown of a tooth projecting into the cystic cavity is pathogno- ing a bluish hue.13 The erupting tooth typically expands through and ruptures
monic15; however, definitive diagnosis still requires histologic analysis. the cyst. Rarely is unroofing required because of delayed eruption.17
The affected tooth may be displaced large distances or even be re-
sorbed.13 These cysts have shown malignant degeneration, and both squa- Paradental Cysts
mous cell carcinoma and mucoepidermoid carcinomas arising from
Many consider paradental cysts to be a variant of dentigerous
dentigerous cysts have been reported. Simple enucleation is curative.
cysts (Fig. 6). Food-induced periodontitis, impaction, and subsequent
Tooth sparing marsupialization has been used, but with higher
occlusion of a pericoronal tooth pocket are thought to result in cystic
recurrence rates.16
formation leading to the inflammatory cyst. They originate from the
cemento-enamel junction, often on the buccal side of the tooth, rarely
on the mesial surface, and never on the lingual. Mandibular molars

FIGURE 6. Paradental cyst. Reprinted with permission from Elsevier


from Lacalta MG et al.42 Copyright Elsevier. All permission
FIGURE 4. Dentigerous cyst. requests for this image should be made to the copyright holder.

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Rioux-Forker et al Annals of Plastic Surgery • Volume 82, Number 4, April 2019

FIGURE 7. Lateral periodontal cyst. Reprinted with permission from Wolters Kluwer from Meseli SE et al.43 Copyright Wolters Kluwer. All
permission requests for this image should be made to the copyright holder.

are often involved. Tooth extraction is typically performed for any third alveolar bone of erupted vital teeth and treated with enucleation with
molar paradental cyst, as opposed to enucleation in other locations.18 bone curettage.19
Gingival Cyst of the Adult
Lateral Periodontal Cyst This is the soft tissue equivalent of the lateral periodontal cyst
These lesions develop on the lateral surface of a vital tooth from (Fig. 9).2 This lesion is derived from dental lamina remnants but found
dental lamina and comprise less than 3% of odontogenic jaw cysts (Fig. 7). in the gingiva. These may be blue in color and are commonly found in
They can displace tooth roots and appear as a swelling in the interdental the fifth and sixth decades.1 Radiographically, superficial cortical cup-
gingival papilla.1 They retain neoplastic potential but are typically found ping may occur, along with bone erosion.13 Treatment with local exci-
incidentally in the interproximal area between tooth roots.2 Lateral peri- sion and histopathologic analysis is recommended.
odontal cysts are typically diagnosed in the fifth and sixth decades and Gingival Cyst of the Newborn
more often in males.
These are small lesions, being only a few millimeters in diameter,
Radiologically, these are unilocular and well corticated. Histology
present on the alveolar ridge, and seen only in the first few months of
differentiates these cysts from KCOT, odontogenic cysts of undetermined
life. They are isolated to the soft tissue only, with no bony component.
origin, and lateralized periapical cysts.
Histologic analysis is unnecessary. These lesions nearly always rupture
Simple enucleation is indicated for unilocular lesions. If
spontaneously before 3 months of age and rarely require intervention.1
multilocular, the lesion is likely a botryoid odontogenic cyst, and light
Enucleation or deroofing may be indicated when a lesion significantly
curettage is indicated.2
disrupts feeding.20

Botryoid Odontogenic Cyst Keratinizing Odontogenic Cyst (Orthokeratinized


Some describe this as a variant of the lateral periodontal cyst, but Odontogenic Cyst)
multiloculated and occasionally multicystic (Fig. 8). Others differentiate Originally, these lesions were not differentiated from KCOTs
it from lateral periodontal cysts because of its larger size, minor histo- (Fig. 10). However, they have a significantly different prognosis, with
logic differences, and increased recurrence risk. They are found in

FIGURE 8. Botryoid odontogenic cyst. Reprinted with FIGURE 9. Gingival cyst of the adult. Reprinted with permission
permission from Wolters Kluwer from Arora P et al.44 Copyright from Hindawi from Brod JMM et al.45 Copyright Juliana Mancano
Wolters Kluwer. All permission requests for this image should be Melhado Brod. All permission requests for this image should be
made to the copyright holder. made to the copyright holder.

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Annals of Plastic Surgery • Volume 82, Number 4, April 2019 Odontogenic Cysts and Tumors

or sclerotic borders, root resorption, or tooth displacement.22 This lesion


can be confused with other odontogenic cysts or mucoepidermoid carcino-
mas. Clear or red-brown viscous fluid on aspiration may help lead to a clin-
ical diagnosis.23 Surgical treatment should include enucleation with bone
curettage and follow-up, as high recurrence rates up to 25% are reported.1

Odontogenic Tumors
Ameloblastomas
Ameloblastomas are the most common jaw neoplasm and are of-
ten asymptomatic, arising from the enamel organ (Fig. 12).13 Generally,
they are the most aggressive benign neoplasm. They are typically slow
growing but can reach large and destructive sizes.1 Preoperative CT or
magnetic resonance imaging may help to direct biopsy, guide resection,
clarify soft tissue involvement, and guide surgical margins. Surgical re-
section is typically the treatment of choice, with wide margins from 1 to
1.5 cm to 2 cm for more aggressive solid subtypes. Curettage and enu-
cleation result in high recurrence rates, 55% to 100% for solid, 18% to
25% for unicystic, and 8% for peripheral. Ameloblastomas have malig-
nant potential and may metastasize. Metastasis occurs in under 1% of
ameloblastomas, which are then termed malignant ameloblastoma.1 The
histologic features of metastatic ameloblastomas are identical to benign
FIGURE 10. Keratinizing odontogenic cyst. Reprinted with
ameloblastomas. Subsequently, diagnosis in a distant or regional site is
permission from Wolters Kluwer from Kulkarni M et al.46
required to label an ameloblastoma as malignant. Metastatic sites include
Copyright Journal of Oral and Maxillofacial Pathology. All
lungs (70%–85%), bone, liver, and brain. Malignant ameloblastomas
permission requests for this image should be made to the
can also be found regionally, as up to 35% of malignant ameloblastomas
copyright holder.
have metastasized to the neck. Metastases are preferentially treated with sur-
little to no neoplastic potential and low recurrence rates. Keratinizing gical excision or radiotherapy in nonoperable tumors. Chemotherapy and ra-
odontogenic cysts have variable presentations, with no unifying clinical diotherapy are yet to be validated as treatment options.24 Ameloblastic
trait. They may present on erupted or unerupted dentition and in vari- carcinoma conversely is any ameloblastoma with anaplastic cytopathologic
able locations often adjacent to the crown.2 They may become very large features without apparent metastasis. Anaplastic features may include in-
and are often found in the third to fifth decades of life. Radiologically, creased mitotic activity, nuclear/cytoplasmic ratio, and hyperchromatism.2
they are uniloculated, well-corticated lesions. Surgical enucleation, histo- Unicystic Ameloblastoma
logic assessment, and follow-up are sufficient treatment for these lesions.21
These lesions are usually asymptomatic and found incidentally,
unless they become secondarily inflamed. They comprise 15% of all
Glandular Odontogenic (Sialoodontogenic) Cyst
This lesion is poorly understood (Fig. 11). It typically presents
asymptomatically but can be very large and destructive. Radiographi-
cally, this lesion may be unilocular or multilocular, and often anteriorly
positioned. The margins are generally well defined with a sclerotic rim.
Aggressive growth and expansion may lead one to consider this rare cyst.2
Aggressive features include multilocularity, cortical expansion, scalloped

FIGURE 11. Glandular odontogenic cyst. Reprinted with


permission from Wolters Kluwer from Shah AA et al.47 Copyright
Wolters Kluwer. All permission requests for this image should be
made to the copyright holder. FIGURE 12. Ameloblastoma.

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Rioux-Forker et al Annals of Plastic Surgery • Volume 82, Number 4, April 2019

overlying mucosa are typically sent with the specimen. Soft tissue mar-
gins should be assessed with preoperative advanced imaging and
intraoperative frozen sections. The goal is to include 2 biologic barriers
in your resection.

Peripheral (Extraosseous) Ameloblastoma


These soft tissue lesions may arise from the dental lamina or from
basal cells of the overlying mucosa. By definition, these do not infiltrate
adjacent bone. Surgical excision with histologically confirmed margins
is the treatment of choice.2 Some advocate for 1 cm intraoperative mar-
gins with negative frozen sections to obtain complete excision. Often a
marginal mandibulectomy is performed with resection of a cuff of un-
involved alveolar bone.24

Ameloblastic Fibroma
FIGURE 13. Ameloblastic fibroma. Reprinted with permission These are benign neoplasms with proliferating mesenchyme and
from Wolters Kluwer from Verma N.48 Copyright Wolters ameloblastic cells (Fig. 13). They are differentiated from fibroodontomas
Kluwer. All permission requests for this image should be made to or odontomas by their lack of enamel, detin, or cementum.2 They are slow
the copyright holder. growing and asymptomatic unless they become secondarily inflamed.
Once large enough, they may cause an expansion of the overlying buc-
ameloblastomas and are often found in the second or third decades of cal cortex. Ameloblastic fibromas are commonly diagnosed before the
life.1 These ameloblastomas are differentiated by their unilocular, age of 20 or during middle age. Radiologically and clinically indistinct,
unicystic nature, and lack of connective tissue invasion, giving them a they appear as a well-defined radiolucency with a corticated (or sclerotic)
better prognosis. border and may be unilocular or multilocular.1 Histopathology is required
Radiologically, they appear similar to dentigerous cysts (radiolu- to differentiate these lesions from ameloblastomas. Fine needle aspiration
cent, unilocular, with well-corticated borders) but are not confined to or biopsy may help identify this lesion preoperatively. Surgical enucle-
unerupted teeth or the crown region. Up to 80% of these tumors further ation often with curettage is the treatment of choice, with low recurrence
mimic dentigerous cysts by enclosing a tooth crown.13 Performing a rates and a favorable prognosis.25 Follow-up remains important because
biopsy with curettage is advocated for diagnosis given the similarities there are cases of malignant transformation into ameloblastic fibrosar-
shared with dentigerous cysts. Aspiration is typically nondiagnostic. If comas.26 There remains no indication for more extensive surgical treat-
the pathology returns positive for ameloblastoma, then any surgically ment unless there is recurrence or malignant change.
violated mucosa or gingiva would require resection. For unilocular
and unicystic ameloblastomas with no soft tissue involvement and mea- Ameloblastic Fibroodontomas
sure less than 2 cm in diameter, the ideal surgical treatment continues to These are rare odontogenic tumors that are similar to ameloblastic
be debated. Simple enucleation with or without curettage is performed fibromas but display radiographic or histopathologic mineralization
by many but at the expense of an increased recurrence rate of 10% at and contain enamel and dentin (Fig. 14).1 They are benign and are
10 years.13 Others have added cryotherapy, Carnoy's solution, drilling,
or electrocautery to help decrease recurrence rates. Some advocate an
even more aggressive approach at the expense of additional morbidity.
This includes en bloc resection with margins 1 to 1.5 cm beyond the ra-
diographic margin and negative frozen sections of any soft tissue involve-
ment. Immediate vascularized bone reconstruction typically follows
this approach.24

Intraosseous Ameloblastoma
These comprise the majority of ameloblastomas, making up
about 80%.2 Prognosis is often related to tumor location. The more su-
perficially located or cortically confined lesions often have better out-
comes. These lesions can readily invade soft tissue and medullary bone;
however, cortical bone and periosteum often act as barriers. Radiologi-
cally, these lesions have a classic soap bubble or honeycomb appear-
ance; however, smaller lesions may be unilocular and indistinguishable
from other lesions of the jaw.2 The lesion itself may displace teeth or
cause root resorption.24
Treatment of intraosseous ameloblastomas with enucleation with
or without curettage has shown a high recurrence rate. Subsequently,
surgical resection with negative radiographic margins and negative his-
tologic margins is the treatment of choice. Even with en bloc resection
beyond the radiologic margins, recurrence rates of 10% to 15% have
been reported. To ensure complete removal, many advocate surgical
margins of 1.5 to 2 cm past the radiographic margin as well as negative
frozen margins intraoperatively.24 Others recommend an excision 1 cm
past the radiologic margin.2 Soft tissue, except alveolar mucosa, is pre-
served unless there is periosteal involvement. Teeth, gingiva, and FIGURE 14. Ameloblastic fibroodontoma.

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Annals of Plastic Surgery • Volume 82, Number 4, April 2019 Odontogenic Cysts and Tumors

macrocephaly, frontal bossing, palate abnormalities, palmar pits, bifid


ribs, polydactyly, and syndactyly among other findings.29
Optimal treatment is currently debated. The goal of surgical
treatment is removal of the cyst and lining, including satellite cysts
and dental lamina rests. Enucleation alone has high recurrence rates of
about 62.5%. Enucleation with thorough curettage decreases recurrence
rates down to 10%.2 Many have included cryotherapy, electrocautery, or
chemical cautery for decreased recurrence rates.30 Some advocate a more
aggressive approach with enucleation, excision of overlying mucosa, pe-
ripheral osteotomy, and chemical cautery. Pogrel31 has suggested a
treatment protocol that was developed due to the difficulty of obtaining
Carnoy's solution and the soft tissue damage that can be seen with liquid
nitrogen. His method involves decompressing the lesion with a drain
until it is 2 to 3 cm in size, after which he performs surgical enucleation
with peripheral ostectomy.31 Marginal mandibulectomy or segmental
FIGURE 15. Keratocystic odontogenic tumors (4) in a patient resection may be indicated with aggressive tumor invasion or penetra-
with Gorlin syndrome. tion.32 Long-term follow-up is crucial because recurrence can be seen
decades after initial treatment.
typically diagnosed in patients before the age of 20 years. Like
ameloblastic fibromas, these lesions can be locally expansive and de-
structive. Surgical enucleation is the treatment of choice, with curettage Calcifying Cystic Odontogenic Tumor (Gorlin Cyst)
for multilocular lesions. In many cases, adjacent dentition can be pre- There is debate on whether to classify calcifying cystic odontogenic
served.27 There are isolated cases of malignant transformation requiring tumors as neoplasms or cysts (Fig. 16). Thought to arise from dental
more aggressive resection, but recurrence and malignant transformation lamina rests, this lesion may occur intraosseously or peripherally in the
are atypical and highlight the importance of long-term follow-up.28 gingiva.2 It may be unilocular or multilocular and is radiolucent (with
occasional opacities).33 These are benign in nature but are thought to
have rare neoplastic potential. Cases have been reported of recurrent
KCOT (Odontogenic Keratocyst, Parakeratizing calcifying cystic odontogenic tumors and progression into Ghost cell
Odonotogenic Keratocyst) odontogenic carcinoma.34 Enucleation with curettage is the advocated
These lesions make up 10% to 12% of odontogenic cysts treatment for these lesions with extended clinical follow-up.35
(Fig. 15).1 Debate exists on whether to classify these lesions as cysts
or neoplasms, but the general trend is toward neoplastic classification. Odontogenic Myxoma
Often asymptomatic, KCOTs can cause symptoms of swelling, pain, This benign neoplasm arises from the dental papilla or dental sac
trismus, sensory deficits, or infection. They have a peak incidence in (Fig. 17). They are often locally aggressive tumors but without malig-
the second and third decades of life. Radiologically, these lesions can nant or metastatic potential. Clinical examination often shows a smooth,
be unilocular or multilocular with well-defined borders. They can be bony, hard expansion of the alveolus. Odontogenic myxomas are often
found at any site of odontogenesis and may extend significant distances identified in the second or third decades of life. As with most odontogenic
and aggressively invade tissue planes.2 neoplasms, these may be unilocular or multilocular and are radiolucent.
Gorlin syndrome (basal cell nevus syndrome) is commonly asso- Cortical penetration can occur, and a periosteal reaction can result giving
ciated with KCOT and should be considered in any patient with multiple a honeycomb or tennis racket appearance. Grossly, the contents of the
KCOTs. Gorlin syndrome typically results from an autosomal domi- myxoma are gelatinous, which makes surgical enucleation difficult.
nant PTCH gene mutation. Patient history may include numerous basal For noninvading smaller lesions (<3 cm), enucleation and curettage of
cell carcinomas, KCOTs, cleft lip, hypertelorism, ovarian fibromas, medullary bone going 0.5 to 1 cm past radiographic margins (+/− marginal
resection) with negative histologic margins may be performed.2

FIGURE 16. Calcifying cystic odontogenic tumor. Reprinted with FIGURE 17. Odontogenic myxoma. Reprinted with permission
permission from Springer Nature from Masuda K et al.49 from College of American Pathologists from Li TJ et al.50 Copyright
Copyright Masuda et al. 2005. All permission requests for this College of American Pathologists. All permission requests for this
image should be made to the copyright holder. image should be made to the copyright holder.

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Rioux-Forker et al Annals of Plastic Surgery • Volume 82, Number 4, April 2019

FIGURE 18. Adenomatoid odontogenic tumors. Reprinted with permission from Wolters Kluwer from Belgaumi U et al.51 Copyright
Annals of Medical and Health Sciences Research. All permission requests for this image should be made to the copyright holder.

However, the recurrence rate for this is thought to be as high as 25%.1 and recurrence are not typical, and associated dentition can often be
For more invasive or larger lesions, a more aggressive approach includ- preserved.39
ing segmental, en bloc resection, or hemimandibulectomy with recon-
struction is advocated by some.36,37
Odontomas
Adenomatoid Odontogenic Tumor The classification of odontomas is debated, with some consider-
Adenomatoid odontogenic tumors represent 3% to 7% of all ing them hamartomas and others considering them odontogenic tumors
odontogenic tumors (Fig. 18).1 This tumor develops from either the enamel (Fig. 19). They are the most commonly diagnosed odontogenic tumor.1
organ or dental lamina and is histologically similar to ameloblastomas.2 They consist of masses of enamel, enamel matrix, dentin, and cemen-
It is known as the two-thirds tumor, because two thirds occur in female, tum arranged either chaotically (complex odontoma) or in an organized
two thirds occur in maxilla, two thirds are associated with impacted teeth, tooth-like fashion (compound odontoma). Grossly, compound odontomas
and two thirds occur in teenagers. Adenomatoid odontogenic tumors are may have small, malformed, identifiable teeth. Odontomas may be as-
often associated with impacted canine teeth. Radiographically, these le- sociated with other odontogenic cysts or tumors. They can also deform
sions may be mistaken for dentigerous cysts because they are often asso- adjacent teeth or alter eruption.2 Radiographically, these lesions have a
ciated with impacted teeth, blend into the tooth follicle, are radiolucent, central radiopaque region with a sack of teeth or disorganized aggregate
and are unilocular. Adenomatoid odontogenic tumors often contain fine appearance. There is a peripheral radiolucent area and a well-defined
calcifications.1 Surgical enucleation is the treatment of choice because and visible corticated border. Treatment is indicated only if the diagno-
these lesions are typically well encapsulated.38,39 Aggressive behavior sis is uncertain or the odontoma is disrupting adjacent dentition.1

FIGURE 19. Compound odontoma. Reprinted with permission from Wolters Kluwer from Sreedharan S et al.52 Copyright Wolters Kluwer.
All permission requests for this image should be made to the copyright holder.

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Annals of Plastic Surgery • Volume 82, Number 4, April 2019 Odontogenic Cysts and Tumors

Treatment is simple enucleation with preservation of adjacent anatomy 25. Kumar N, Jain S. Aspiration cytology of ameloblastic fibroma: a diagnostic chal-
and dentition.40,41 lenge. Diagn Cytopathol. 2003;29:101–104.
26. Bregni RC, Taylor AM, Garcia AM. Ameloblastic fibrosarcoma of the mandible:
report of two cases and review of the literature. J Oral Pathol Med. 2001;30:
CONCLUSIONS 316–320.
Odontogenic cysts and tumors represent a diverse group of path- 27. Pontes HA, Pontes FS, Lameira AG, et al. Report of four cases of ameloblastic
ologic lesions of the jaw. Physical examination along with imaging is the fibro-odontoma in mandible and discussion of the literature about the treatment.
best initial tool for developing a differential diagnosis. Final diagnosis is J Craniomaxillofac Surg. 2012;40:e59–e63.
made from the surgical pathology, and it dictates whether further treat- 28. De Riu G, Meloni SM, Contini M, et al. Ameloblastic fibro-odontoma. Case re-
ment is required. Long-term follow-up is usually indicated after any sur- port and review of the literature. J Craniomaxillofac Surg. 2010;38:141–144.
gical excision because of high recurrence rates of many of the lesions. 29. Bresler SC, Padwa BL, Granter SR. Nevoid basal cell carcinoma syndrome
(Gorlin syndrome). Head Neck Pathol. 2016;10:119–124.
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