Management of odontogenic cysts by endonasal endoscopic

techniques: A systematic review and case series

Michael J. Marino, M.D., Amber Luong, M.D., Ph.D., William C. Yao, M.D., and
Martin J. Citardi, M.D.

ABSTRACT
Background: Odontogenic cysts and tumors of the maxilla may be amendable to management by endonasal endoscopic techniques, which may reduce the
morbidity associated with open procedures and avoid difficult reconstruction.

P Y
Objective: To perform a systematic review that evaluates the feasibility and outcomes of endoscopic techniques in the management of different odontogenic
cysts. A case series of our experience with these minimally invasive techniques was assembled for insight into the technical aspects of these procedures.
Methods: The Preferred Reporting Items for Systematic Reviews and Meta-Analyses was used to identify English-language studies that reported the use
of endoscopic techniques in the management of odontogenic cysts. Several medical literature data bases were searched for all occurrences in the title or abstract

O
of the terms “odontogenic” and “endoscopic” between January 1, 1950, and October 1, 2016. Publications were evaluated for the technique used,
histopathology, complications, recurrences, and the follow-up period. A case series of patients who presented to a tertiary rhinology clinic and who underwent
treatment of odontogenic cysts by an endoscopic technique was included.
Results: A systematic review identified 16 case reports or series that described the use of endoscopic techniques for the treatment of odontogenic cysts,

C
including 45 total patients. Histopathologies encountered were radicular (n ⫽ 16) and dentigerous cysts (n ⫽ 10), and keratocystic odontogenic tumor (n ⫽
12). There were no reported recurrences or major complications for a mean follow-up of 29 months. A case series of patients in our institution identified seven
patients without recurrence for a mean follow-up of 10 months.
Conclusion: Endonasal endoscopic treatment of various odontogenic cysts are described in the literature and are associated with effective treatment of these
lesions for an average follow-up period of ⬎2 years. These techniques have the potential to reduce morbidity associated with the resection of these lesions,

T
although comparative studies would better define specific indications.
(Am J Rhinol Allergy 32, 40 –45, 2018; doi: 10.2500/ajra.2018.32.4492)

T raditionally, odontogenic cysts have been treated by enucleation,

curettage, and marsupialization.1–3 Radicular cysts account for
nusitis, and extended recovery time.12,15 Open approaches can also
result in a more-complex reconstruction than an endonasal tech-

O
half of all odontogenic cysts and are followed in frequency by denti-
gerous cysts and keratocystic odontogenic tumor (KCOT).4–6 The opti-
mal management strategy for odontogenic cysts is controversial, partic-
ularly with regard to the extent of resection for KCOT.3 There is a
nique.15 Differentiation of odontogenic cyst histopathology before
surgery can be difficult, in which case, endoscopic management offers
a potentially diagnostic and therapeutic approach.16 Treatment of all
of the three most-common types of odontogenic cysts (radicular

N
predilection for recurrence in cases of KCOT, and this may be related to
the pathophysiology of these lesions and to the treatment technique.3,7–9
The recognition that these lesions represent a benign neoplasm of odon-
togenic origin resulted in the change of terminology by the World Health
Organization from odontogenic keratocyst to KCOT,10 which also dis-
tinguishes KCOT from radicular cysts, which seem to have an inflam-
cysts,18,19,24,25 dentigerous cysts,18,19,25–28 and KCOT15,17,19,20,22,23,29) that
used an endonasal endoscopic technique has been described.
The purpose of this systematic review was to identify the several
recent reports of endonasal endoscopic management of odontogenic
cysts and to determine if these techniques represent a feasible and
effective treatment strategy for these lesions. Analysis of the recurrence

O
matory origin, and from dentigerous cysts, which are of developmental
origin.6 Therefore, more-extensive resection has been advocated for cases
of KCOT with the inclusion of peripheral ostectomy.3
Numerous reports over the past 10 years describe endonasal endo-
scopic techniques in the management of odontogenic cysts of the
and complication rates and of the length of follow-up would allow for
evaluation of the usefulness of these techniques in regard to both effec-
tiveness and morbidity. Studies of both endonasal and open approaches
would be particularly useful in comparing relative effectiveness and
were included in the systematic review search strategy. Finally, a case

D
maxilla.11–26 The presumed benefit of an endoscopic approach is
avoidance of morbidity associated with open, transoral approaches,
including alterations in dentition, oroantral fistula, chronic rhinosi-
series of endonasal endoscopic treatment of odontogenic cysts at our
institution was included for pooled analysis with previously published
reports. The case series also offered insight into technical considerations
of this management strategy in patients with odontogenic cysts.

From the Department of Otorhinolaryngology—Head and Neck Surgery, John P. and

Kathrine G. McGovern Medical School, The University of Texas Health Science Center
at Houston, Houston, Texas
Poster presentation, the American Rhinologic Society Spring Meeting, April 26 –30,
2017, San Diego, CA
No external funding sources reported
M.J. Citardi serves as a consultant for Acclarent (Irvine, CA), Biosense Webster (Haifa,
Israel), Factory CRO (Bilthoven, The Netherlands), Hemostatis, L.L.C. (St. Paul, MN)
Medical Metrics (Houston, TX), Medtronic ENT (Jacksonville, FL), and Optinose (Yard-
ley, PA). The remaining authors have no conflicts of interest pertaining to this article
Address correspondence to Martin J. Citardi, M.D., Department of Otorhinolaryngol-
ogy—Head and Neck Surgery, John P. and Kathrine G. McGovern Medical School, The
University of Texas Health Science Center at Houston, 6431 Fannin St., MSB 5.036,
Houston, TX 77030
E-mail address: martin.j.citardi@uth.tmc.edu
Copyright © 2018, OceanSide Publications, Inc., U.S.A.
METHODS
Literature Search and Systematic Review
A systematic review was performed according to Preferred Report-
ing in Systematic Reviews and Meta-analyses guidelines,27 on the
outcomes of endonasal endoscopic management of odontogenic cysts.
The systematic review protocol was specified in advance and regis-
tered in the PROSPERO (University of York, York, UK) database with
the registration number CRD42016049553.29 Publications were evalu-
ated for the surgical technique used and the lesion histopathology.
Outcome measures of interest were reported complications, recur-
rences, and the follow-up period. Case reports, case series, and ret-
rospective and prospective cohorts that report original data on odon-
togenic cysts treated by an endonasal endoscopic technique were

40 January–February 2018, Vol. 32, No. 1

Table 1 Diagnostic Codes for Odontogenic Cysts
ICD-9 Code ICD-10 Code
522.8 K04.8
526.0, 526.2 K09, K09.0
213.0 D16.4
520 K00
520.2 K00.2
520.5 K00.5
520.9 K00.9
ICD-9 ⫽ The International Classification of Diseases, Ninth Revision; ICD-10 ⫽ The International Classification of Diseases, Tenth Revision; KCOT ⫽
keratocystic odontogenic tumor.
Figure 1. Schematic diagram of the search strategy and article selection
process. *One article for full-text review was excluded because it was a case
O
of ameloblastoma.
included. Only English language articles were eligible for inclusion.
N
Studies that reported on combination open and endoscopic proce-
dures were excluded, although studies with a comparison open in-
tervention group were considered for inclusion. Only human studies
were included in the systematic review.
A systematic search of the MEDLINE (PubMed [National Center for
Biotechnology Information, Bethesda, MD] and OVID [Ovid Technolo-
O
gies, New York, NY] portals), EMBASE (Elsevier, Amsterdam, The Neth-
erlands), and Cochrane Review (Cochrane Collaboration, London, UK)
databases was performed for all English language articles published
between January 1, 1950, and October 1, 2016. Search criteria included all
occurrences in the title or abstract of the terms “odontogenic” and
D
“endoscopic.” Duplicate citations were removed, and all abstracts were
reviewed for the stated eligibility criteria. A full-text review was per-
formed for all abstracts that met the eligibility criteria. Additional studies
not included in the search strategy were identified by a manual screen-
ing of the reference lists of those studies included in the full-text reviews.
Authors were contacted for any missing or incomplete data of interest in
the articles that underwent full-text review. Data were extracted from the
source articles by using a standardized template. The surgical technique,
histopathology, complications, recurrences, and follow-up period were
recorded when available. The risk of bias was assessed at the study level
by the length of follow-up, basis for treatment allocation, and blinding of
investigators. The level of evidence28 was determined to provide an
overall estimate of the strength of the study design.
Case Series
A retrospective review of patients who presented to a tertiary
otorhinolaryngology clinic with a diagnosis of an odontogenic cyst
was performed. Patients were identified by searching for the relevant
International Classification of Diseases,30 (ICD), Ninth Revision (ICD-9)
American Journal of Rhinology & Allergy
Clinical Diagnosis
Radicular cyst
Developmental odontogenic cyst (including dentigerous cyst)
Benign neoplasm of bones of skull and face (including KCOT)
Disorders of tooth development and eruption
Abnormalities of size and form of teeth
Y
Hereditary disturbances of tooth structure, not elsewhere classified
Disorder of tooth development, unspecified
P
and the ICD, Tenth Revision (ICD-10) codes (Table 1) from March 1,
2008, to October 24, 2016. Those patients treated by using an endo-
O
nasal endoscopic technique were included in a case series. Medical
records were evaluated for the surgical technique, histopathology,
complications, recurrences, and follow-up period. The study was
approved by the University of Texas Health Science Center at Hous-
ton Committee for the Protection of Human Subjects.
RESULTS
Systematic Review
C
T
A total of 138 articles were identified through the defined search
strategy of the MEDLINE, EMBASE, and Cochrane Review data bases.
Of these, 54 articles were identified as duplicates among the data bases
and were removed. Review of the article titles and abstracts after dupli-
cate removal identified 15 articles for full-text review. An additional two
studies were identified for inclusion from the reference lists of articles
that underwent full-text review. From these articles, 16 studies were
determined to meet the stated search criteria (Fig. 1).
Studies included in the systematic review are summarized in Table
2.11–26 In the 16 included studies, a total of 45 patients were treated for
different odontogenic cysts by using a variety of endonasal endo-
scopic techniques. No recurrences were reported, and the weighted
mean follow-up was 29.2 months. The mean follow-up of cases of
KCOT was 21.6 months. There was a single patient who was thought
to have a new occurrence of KCOT secondary to basal nevus (Gorlin)
syndrome, and this was not counted as a recurrent lesion.25 No major
complications were reported, with persistent inferior meatus acces-
sory ostia in a single patient treated by the endoscopic medial max-
illary sinus wall transposition technique described by Maxfield et al.13
Image guidance was used in two studies,11,13 whereas its use was not
explicitly stated in the remaining studies.
The level of evidence was generally low with all of the included
studies that represented case reports or series. All the patients were
allocated to endoscopic management at the direction of the treating
physician, and the same physicians evaluated for recurrences and
complications. Systematic review did not reveal any comparative
studies between open and endoscopic techniques in the treatment of
odontogenic cysts, which precluded assessment of the relative effec-
tiveness of these approaches. Assessment of study design and the
level of evidence is presented in Table 3.
Case Series
A retrospective review of the patients with qualifying ICD-9 or
ICD-10 diagnostic codes (Table 1) identified seven patients (six men
and one woman, with a mean age of 40.4 years) who were treated by
using an endonasal endoscopic technique (Table 4). An inframeatal
window was used in one case due to a relatively large inferior-
superior-anterior dimension of the maxillary sinus; this extra access
point ensured complete removal of the tumor. In this case, an ectopic
molar was also addressed via the inframeatal window when complete
41
Table 2 Summary data of the included studies
Study No. Diagnoses Intervention Mean Follow- Recurrences Complications
Subjects Up, mo
Subramaniam et 1 Odontogenic Revision endoscopic resection 3 None N.R.
al.,11 2017 myxoma of pterygoid plates,
maxillary antrostomy, total
ethmoidectomy,

Y
sphenoidotomy
Barry et al.,12 2016 4 KCOT Endoscopic medial 12.5 None N.R.
maxillectomy, application
of Carnoy solution

P
Maxfield et al.,13 5 “Ondontogenic Transnasal endoscopic medial 13.8 None Inferior meatus
2016 cyst” maxillary sinus wall accessory
transposition, maxillary ostia (1/5
antrostomy patients)
Nomura et al.,14 1 KCOT Endoscopic medial 12 None N.R.

O
2015 maxillectomy, fenestration
of hard palate
Jain and Goyal,15 6 Radicular cyst, Maxillary mega-antrostomy 34 None N.R.
2015 dentigerous
cyst, KCOT

C
Nakayama et al.,16 6 Radicular cyst, Endoscopic medical 17.4 None N.R.
2014 dentigerous maxillectomy
cyst, KCOT
Marcotullio et 1 KCOT Maxillary antrostomy, 96 None N.R.
al.,17 2014 inferior turbinate reduction

T
Schuster et al.,18 1 Calcifying cystic Endoscopic resection Unknown Unknown N.R.
2014 odontogenic
tumor
Mun et al.,19 2014 1 KCOT Maxillary antrostomy 60 None N.R.
Ohki20 2012 1 KCOT Maxillary antrostomy 12 None N.R.

O
Sagit et al.,21 2011 1 Radicular cyst Maxillary antrostomy 6 None N.R.
Seno et al.,22 2009 13 Radicular cyst, Endoscopic medical 42 None N.R.
dentigerous maxillectomy
cyst

N
Avitia et al.,23 1 Dentigerous cyst Maxillary antrostomy Unknown Unknown N.R.
2007
Micozkadioglu 1 Dentigerous cyst Maxillary antrostomy 12 None N.R.
and Erkan24
2007
Chiang et al.,25 1 KCOT Endoscopic excision of nasal 60 No recurrence at N.R.

O
2006 floor original site,
possible
Gorlin
syndrome
Di Pasquale and 1 Dentigerous cyst Maxillary antrostomy 24 None N.R.

D
Shermetaro26
2006
KCOT ⫽ Keratocystic odontogenic tumor; N.R. ⫽ none reported.

visualization was not possible from the enlarged maxillary antros-
tomy. Image guidance was used in seven of the eight cases.
The reported histopathology included four KCOTs, two dentiger-
ous cysts, and one radicular cyst. The mean follow-up period was 10
months. There were no major complications and two minor compli-
cations. One patient was treated for acute sinusitis at 1 week after
surgery, and another patient developed a mucocele within the treated
maxillary sinus 12 months after the original surgery. Patients were
also followed up clinically for dental compromise, with no signs of
such events in our patient population.
Pooled Analysis
None of articles identified in the systematic review included objec-
tive data for synthesis in a traditional, quantitative meta-analysis.
Nevertheless, a pooled analysis of recurrence and complication rates
was performed with subjects identified in this case series. There were
no reported recurrences in the articles reviewed or in our case series.
No major complications were reported, with the minor complicat-
ions of an accessory ostia, acute sinusitis, and mucocele each identi-
fied in one patient. The overall minor complication rate was 5.8%.
DISCUSSION
Open and transoral techniques have been the traditional approach
to the treatment of odontogenic cysts.1–3 There has been some contro-
versy regarding the extent of resection necessary to avoid recurrences,
particularly in the treatment of KCOT.3 Nevertheless, endonasal en-
doscopic management of odontogenic cysts of the maxilla, including

42 January–February 2018, Vol. 32, No. 1


Table 3 Study design and the level of evidence for studies
included in the systematic review
Study
Subramaniam et al.,11 2017
Barry et al.,12 2016
Maxfield et al.,13 2015
An adequate maxillary opening is also important for allowing
postoperative surveillance. Mucocele formation occurred in a 16-
year-old boy who did not tolerate postoperative endoscopy and
Study Level of debridement (Table 4). In a revision procedure, the antrostomy
Design Evidence* was widened and the patient had better tolerated in-office endos-
Case report 5 copy and debridement. The endonasal endoscopic approach offers
Case series 4 the advantage of postoperative surveillance with in-office endos-
Case series 4 copy. This is an important consideration given the predilection for
recurrence of these lesions and KCOTs in particular.3,31,32 Because

Y
Nomura et al.,14 2015 Case report 5
Jain et al.,15 2015 Case series 4 of the relationship of these lesions to the maxillary teeth, all the
Nakayama et al.,16 2014 Cases series 4 patients were encouraged to continue routine dental care after
Marcotullio et al.,17 2014 Case report 5 endoscopic management.
Adjunctive use of the Carnoy or the modified Carnoy solution has

P
Schuster et al.,18 2014 Case report 5
Mun et al.,19 2014 Case report 5 been applied to remnant epithelia in the treatment KCOT in both
Ohki et al.,20 2012 Case report 5 open and endoscopic techniques.3,12,33 The Carnoy solution consists of
Sagit et al.,21 2011 Case report 5 100% ethanol, chloroform, glacial acetic acid, and ferric chloride.33
Seno et al.,22 2009 Case series 4 Compounding with chloroform, however, was banned in 1992 by the

Avitia et al.,23 2007
Micozkadioglu et al.,24 2007
Chiang et al.,25 2006
Di Pasquale et al.,26 2006
O
Case report 5 U.S. Food and Drug Administration due to carcinogenic effects, and,
Case report 5 therefore, modified solutions without chloroform have been used.12,33
Case report 5 These modified solutions remain flammable and corrosive to instru-
Case report 5 mentation.12 Adjunctive chemical treatment of remnant epithelia was
not used in most of the reports included in this systematic review and

*From Ref. 28.
KCOTs, has been repeatedly described over the past 10 years in a
total of 52 patients (Tables 2 and 4).11–26 There were no reported
C
is not used in our endoscopic management of these lesions. The
difficulty in obtaining and handling these solutions may preclude
their routine use, and data on the effectiveness of modified solutions
without chloroform are lacking.3
The use of image-guided surgery was reported in the minority of

recurrences when using these approaches, including 12 cases of
KCOT.12,14–17,19,20,25 No major complications were associated with
these procedures, and minor complications were limited to 3 of 52
cases.13 In total, analysis of these data indicated that an endonasal
endoscopic approach to odontogenic cysts of the maxilla is a
T
studies.11,13 Several studies noted the importance of achieving wide
exposure and direct visualization of the lesion with angled endo-
scopes to maximally resect the cyst wall.12,15,16 In our practice, com-
plete visualization of the cyst is aided by image-guided surgery and
was used in 87.5% of cases and in all cases of KCOT (Fig. 3). Com-

O
feasible and effective treatment strategy, and is without undue
morbidity. Even in cases of locally aggressive KCOT, the recur-
rence rate seemed to be comparable with open approaches, al-
though follow-up remains limited.3,31
puted tomography is almost always used in the diagnostic evaluation
of these patients, and obtaining a scan with image-guidance protocol
can then be implemented in the operating room. Image guidance is
particularly useful in defining the inferior and anterior extent of the

N
A multitude of endoscopic techniques have been used in the treatm-
ent of odontogenic cysts, including maxillary antrostomy,17,19–21,23,24,26
“mega-antrostomy,”15 modified medial maxillectomy,12,14,16,22 and
medial maxillary sinus wall transposition.13 All of these techniques
have been used effectively, although, in reviewing patients from our
institution, a wide maxillary antrostomy allows for effective decom-
lesion and, in turn, in ensuring the cyst has been completely decom-
pressed and widely marsupialized.
There are two principal limitations to the current study. First,
endonasal endoscopic management of odontogenic cysts was only
described in case reports and series (Table 3).11–26 A comparative
study between patients treated with traditional open approaches and
endonasal techniques has not been performed, and the absence of

O
pression and marsupialization of the lesion (Fig. 2). Enucleation and
curettage of KCOT have been associated with recurrence,32 although
this may be related to the transoral approach, which closes over
epithelial remnants. When the lesion is decompressed and widely
marsupialized into the maxillary sinus, it can continue to drain and
such a comparative trial precludes comment on the relative effective-
ness of these two approaches. Such a study has likely been hindered
by endoscopic surgery being performed by otolaryngologists and
transoral approaches being the standard among oral surgeons. Nev-

D
avoid reformation of the cyst. There has not been recurrence of KCOT
with this technique, both in our case series and reports by other
investigators.14–17,19,20 Transoral marsupialization of KCOT, however,
has been reported to result in a 27% recurrence rate at 4 years of
follow up.3
The extent of the maxillary antrostomy may be scaled to a size
sufficient for adequate access for complete marsupialization of the
odontogenic cyst. A complete endoscopic medial maxillectomy does
not need to be the default surgical approach for all maxillary
odontogenic tumors. At our institution, we approach these lesions
by performing an initial maxillary antrostomy and then by enlarg-
ing that opening to the size necessary to achieve the surgical
objective. When an inframeatal window is created for visualization
into the maxillary sinus, the risk of mucus recirculation is miti-
gated by preserving the bulk of the inferior turbinate. In addition,
visualization is performed with at least a 30° telescope and often
with 45° and 70° telescopes, which afford panoramic views via the
surgically created opening into the maxillary sinus. Instrumenta-
tion for endoscopic frontal sinus surgery is also commonly used as
well during these procedures.
ertheless, numerous reports describe effective endonasal manage-
ment of KCOT and radicular and dentigerous cysts with minimal
morbidity.
The second limitation of this study was the relatively short fol-
low-up period in patients managed endoscopically. There is concern
that recurrence of these lesions, and KCOT in particular, may mani-
fest over a period of many years.3,31 KCOT recurrence has been
reported as long as 10 years after the original surgery.32 However,
distinguishing between KCOT and other odontogenic cysts is diffi-
cult, and radicular and dentigerous cysts can likely be treated com-
pletely by less-extensive surgery.16 The endoscopic technique offers a
potentially diagnostic and therapeutic approach, while minimizing
the surgical morbidity for the patient. Furthermore, surveillance can
be performed by in-office endoscopy.
CONCLUSION
Endonasal endoscopic treatment of odontogenic cysts has been
described in multiple reports over the past 10 years and includes the
most common histopathologies of radicular and dentigerous cysts

American Journal of Rhinology & Allergy 43

Table 4 Case series patient data
Age, y Gender Pathology Treatment Recurrence Complications Follow-up, Image
mo Guidance
58 M KCOT Extended maxillary antrostomy* No None 1 Yes
19 M KCOT Endoscopic medial maxillectomy No None 5 Yes
16 M KCOT Maxillary antrostomy, inframeatal No Mucocele 16 Yes
window

Y
67 M KCOT Extended maxillary antrostomy* No None 1 Yes
35 F Dentigerous cyst Maxillary antrostomy No Acute sinusitis 2 Yes
34 M Dentigerous cyst Maxillary antrostomy No None 38 No
54 M Radicular cyst Extended maxillary antrostomy* No None 4 Yes

P
KCOT ⫽ Keratocystic odontogenic tumor.
*Extended maxillary antrostomy was considered to be the extension of the standard maxillary antrostomy posterior to the vertical process of the palatine bone,
inferior to the superior portion of the inferior turbinate bone, and anterior to the lacrimal system.

C O
T
marsupialized into the maxillary sinus.

N O
Figure 2. Endonasal endoscopic decompression and marsupialization of a keratocystic odontogenic tumor (KCOT). (A) A wide maxillary antrostomy was
performed, and the cystic lesion is visualized within the maxillary sinus; (B) the KCOT was entered and decompressed; and (C) the KCOT was widely

ture comparative studies with open approaches will better define

specific indications for the endonasal endoscopic management of
odontogenic cysts.

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Figure 3. Image-guided surgery for a keratocystic odontogenic tumor
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marsupialized into the maxillary sinus, and the position of the distal tip of
the curved suction is confirmed in the inferior and lateral extent of the lesion
by using image guidance.
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