J. Maxillofac. Oral Surg.

https://doi.org/10.1007/s12663-023-01929-0

ORIGINAL ARTICLE

Comparison of Five Different Treatment Approaches

of Mandibular Keratocystic Odontogenic Keratocyst
(OKC): A Retrospective Recurrence Analysis of Clinical
and Radiographic Parameters
Henriette L. Moellmann1 · Aida Parviz1 ·
Marcia Goldmann‑Kirn2 · Madiha Rana3 ·
Majeed Rana1

Received: 17 July 2022 / Accepted: 28 April 2023

© The Author(s) 2023

Abstract The odontogenic keratocyst (OKC) is a benign Recurrences were most frequently diagnosed at the age of
but locally aggressive growing lesion that infiltrates the bone 31–50 (43.9%). Despite numerous studies, there is still no
and surrounding tissue. It is characterized by high rates of unanimous opinion on an effective therapy for OKC. How-
recurrence along with rapid growth. Different forms of ever, precise resection of OKC can be facilitated by preop-
partly successful treatment therapies are reported. The ret- erative 3D-imaging and virtual planning.
rospective study at hand examined 114 patients with OKC
treated over a period of 20 years. Data extracted includes Keywords Keratocystic odontogenic tumor/odontogenic
gender, age, location, previous treatment for the lesion, sur- keratocyst · Odontogenic tumor · Multicystic intraosseous
gery, outcome, recurrence rate and follow-up. 63.1% of the tumor · Virtual surgical planning (VSP) · Computer-
patients underwent cystectomy, 22.5% by cystectomy and assisted surgery (CAS)
carnoy solution, 7.2% by cystectomy, and curettage, 4.5%
by cystostomy and 2.7% by partial resection. In this study,
no significant differences could be observed regarding the Introduction
surgical method. Most recurrences occurred with 91.9% in
the mandible with an average size of 5.5 ­cm2 and increased The World Health Organization (WHO) classifies the odon-
in women. Within a mean follow-up time of 3.6 years the togenic keratocyst (OKC) as a developmental (disembrio-
recurrence rate was 36.9%, on average after 36 months. genetic) cyst in 1992. In 2005 the WHO categorized the
lesion as an ondontogenic tumor (keratocystic odontogenic
* Henriette L. Moellmann tumor, KCOT) [1]. The novel term expresses its neoplastic
[email protected] nature as “a benign uni- or multicystic, intraosseous tumor
Aida Parviz of odontogenic origin, with a characteristic lining of par-
[email protected] akeratinized stratified squamous epithelium and potential for
Marcia Goldmann‑Kirn aggressive, infiltrative behavior”. The WHO pusblished the
[email protected] 4th edition of the ‘Classification of Head and Neck Tumors’
Madiha Rana in January 2017, in which the KCOT is again classified as an
[email protected] OKC [2]. The KCOT or OKC is a benign uni- or multicystic,
Majeed Rana intraosseous tumor. It is of odontogenic origin and shows a
[email protected] characteristic aggressive, infiltrative behaviour [1, 3]. The
1
University Hospital Duesseldorf, 40225 Duesseldorf, OKC is considered an odontogenic cyst first reported by
Germany Philipsen in 1956 and attracted interest because of its patho-
2
Department for Craniomaxillofacial Surgery, Hannover logical features as well as its high recurrence rate [4]. KCOT
Medical School, Hannover, Germany or OKC occur over a wide age range [5] and it is more fre-
3
Department of Psychology, University of Applied Sciences, quent amongst men than women [6]. Numerous studies
22143 Hamburg, Germany

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have concluded that the mandible is perpetually infiltrated for the descriptive evaluation and for the examination of
compared to the maxilla, particularly the posterior body and special peculiarities of the data. T-test was used to check
ascending ramus [6]. Clinical symptoms include pain, swell- whether two independent variables (e.g., age/recurrence)
ing and hypaesthesia. However, there are patients without also had two mean values of different magnitudes in the
any clinical symptoms that are diagnosed accidentally or in population and not only in the case group. This allowed the
late stages, e.g., developing pathological fractures [6]. There comparison an interpretation of the significance between
are reports on OKC penetrating the surrounding soft tissues the occurrence of a relapse and a particular variable in the
[7], base of skull [8], orbit and infratemporal fossa [9]. In population. To record correlations between two variables
radiological images, OKC appears as a unilocular or multi- (e.g., recurrence/gender) in our collective, we used a cross
locular radiolucency with an irregular contour [10]. Histo- table and tested with the Chi-square test whether these cor-
logic features include a thin layer of epithelium, a basal cell relations can be transferred to the whole. We further con-
layer consisting of palisading cuboidal or columnar cells, ducted the Kaplan–Meier analysis to assess time to recur-
and a luminal surface. Furthermore, the OKC shows satel- rence regarding the different surgical methods, gender, age,
lite cells in connective tissue [11]. Being one of the major and significance.
diagnosis criteria, OKC recurrently occurs in association
with basal cell nevus syndrome/Gorlin Goltz syndrome. This
is an autosomal dominant syndrome characterized by basal Results
cell carcinomas, intracranial calcifications (especially of falx
cerebri), and distinct facial abnormalities [12]. Three out of 114 patients (2.6%) suffered from NBCCS. In
Surgical therapy of OKC is still controversial [13]. On the investigated collective, men were affected twice as often
the one hand, there are conservative therapies including (66.7%; 74/111) as women (33.3%; n = 37/111). At initial
an enucleation (with or without curettage), a marsupializa- diagnosis, men were on average 44 and women 41 years
tion, or decompression [14]. On the other hand, there are old. The tumor most frequently occurs in the age group of
ostectomy (curettage with Carnoy’s solution, cryotherapy or 31–50-year-olds (37.8%; n = 42/111). 91.9% of the OKC
electrocautery) or the resection. The choice of the treatment were found in the mandible commonly in the angle between
depends on the size of the cyst, the location and patients’ age the jaw and the mandibular branch (64.0%). Only 8.1% were
[15]. When choosing the appropriate therapy, reduction of located in the maxilla. On average the cysts had a size of 5.5
the recurrence rate and the minimization of morbidity must ­cm2 (measured mesio-distal, cranio-caudal).
be considered [14]. The following methods were used in the investigated col-
Despite numerous studies, there is still no consistent lective of patients (see Table 1):
opinion on the effective therapeutic treatment of KCOT. In most cases (n = 62, 55.9%), the defect after surgery
The aim of the present retrospective study was to record needed no augmentation. In 44.1%, an augmentation was
and analyse the clinical behaviour of KCOT as well as its necessary (see Table 2).
occurrence of recurrences to be able to make treatment rec- Postoperative monitoring of the patient population was
ommendations and to place these in the overall context of based on the clinical examination and radiological find-
the scientific literature. ings over a period of 2 months to 17 years (202 months).
The mean value of the follow-up times was 41.9 months
(3.5 years), the median value 24 months (2.0 years). 70
Material and Methods
Table 1  Overview surgical methods
This retrospective study is based on 114 patients with OKC
who were operated from 1990 to 2010 and was approved by Surgical method Number of patients (n) Percent-
the local ethics committee. Medical records were analyzed age (in
%)
by using a self-developed data registration form. The follow-
ing parameters were queried: age, age at initial diagnosis, CO 25 4.5
gender, medical history, symptoms, location, Relationship CE 70 63.1
to surrounding tissues (teeth, n. alveolaris inferior), radio- CE + CS 5 22.4
logical expansion, previous treatment for the lesion, surgery, CO + CU 8 7.2
time until first recurrence rate and follow-up. The collected PRES 3 2.7
data were recorded with Excel and processed using the CRES 0 0
SPSS 21.0 (IBM Corp. Released 2012. IBM SPSS Statis-
CO Cystostomy; CE Cystectomy; CE + CS Cystectomy and Carnoy
tics for Windows, Version 21.0. Armonk, NY: IBM Corp) solution; CO + CU Cystectomy and curettage; PRES Partial resection
statistics program. The explorative data analysis was used of the jaw; CRES Continuity resection

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J. Maxillofac. Oral Surg.

Table 2  Overview method of augmentation

Method of augmentation Total
No treatment Col- Osteo-synthesis with Osteo-synthesis with Osteo-synthesis with Osteo-synthe-
lagen- bone via bone-scraper cancellous bone from iliac mono-, bi-cortical sis + collagen-
vlies crest bone vlies

CO 15 0 0 10 0 0 25
CE 36 1 1 26 4 2 70
CE + CS 5 0 0 0 0 0 5
CO + CU 4 1 0 1 2 0 8
PRES 2 0 0 1 0 0 3
CRES 0 0 0 0 0 0 0
Total 62 2 1 38 6 2 111

CO Cystostomy; CE Cystectomy; CE + CS Cystectomy and Carnoy solution; CO + CU Cystectomy and curettage; PRES Partial resection of the
jaw; CRES Continuity resection

patients (63.1%) remained without recurrence within the recurrences, followed by the age group of 51–70-year-olds
study period of 20 years. 41 patients had relapses during (29.2%, see Fig. 2).
this period. The recurrence rate was 36.9%. According to the Kaplan–Meier analysis, recurrences
In 27 (65.8%) of the 41 patients with recurrence, recur- occurred after 60 months in 50% of the 31–50-year-old
rence occurred in the first four postoperative years with patients, and after 84 months (51–70-year-old). A signifi-
a decreasing tendency. From the fifth postoperative year cance between the time of recurrence after the first oper-
onwards, 14 patients (34.1%) were still suffering from recur- ation and a certain age group could not be demonstrated
rence. On average, recurrences occurred after approximately (p = 0.869). Within the total number of 41 recurrences, 18
36 months (mean = 46.2 months, see Fig. 1). women (43.9%) and 23 men (56.1%) suffered from recur-
Regarding localization, most recurrences occurred unilat- rence. On a gender-specific basis, 48.6% of women (18/37)
erally in the fourth quadrant (n = 23) or in the third quadrant and 31.1% of men (23/74) received a recurrence within
(n = 16). Only two recurrences were found in the upper jaw. the study period. Using the Chi-square test, no statistically
The recurrence in the upper jaw differed in time from the significant relationship could be established between the
recurrence in the lower jaw. In the maxilla the median recur- occurrence of a recurrence and the gender (p = 0.071). The
rence occurred after 18 months (1.5 years), in the mandible median relapse-free, postoperative time, after which 50% of
after 36 months (3 years) postoperatively, so that the differ- the women received a recurrence, was 36 months (3 years)
ence was 18 months (1.5 years). within the study period. In comparison, 50% of men received
The average age at recurrence was 44 years. The group of a recurrence after 84 months (7 years). These values show
31–50-year-olds with 43.9% was most frequently affected by a time difference of 48 months (4 years) between the recur-
rence of the tumor and the gender. The Kaplan–Meier

Fig. 1  Frequency of occurrence

of the 41 recurrences by month
(in percent)

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 J. Maxillofac. Oral Surg.

Fig. 2  Frequency distribu-

tion of recurrences within age
groups (in percent)

analysis showed a p-value of 0.053, which does not statisti- number of n = 111 patients, the highest recurrence rate
cally support the differences (see Figs. 3 and 4). after cystectomy was observed (18%), followed by cys-
Regarding the individual surgical methods, the fol- tectomy with carnoy solution (12.6%) and cystectomy
lowing differences in recurrence rates were found: 28.6% with curettage (4.5%). Partial resection and cystostomy
after cystectomy, 56% after cystectomy with carnoy solu- showed the lowest recurrence rate with 0.9%. The chi-
tion and 62.5% after cystectomy with curettage. The square test showed a p-value of 0.064 and thus did not pro-
cystostomy showed 20% and the partial lower jaw resec- vide a valid statement as to whether one surgical method
tion showed a recurrence rate of 33.3%. Within the total

Fig. 3  Recurrence-free time in

months

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J. Maxillofac. Oral Surg.

Fig. 4  Observation time until

first recurrence (in months)

Table 3  Recurrence frequency Surgical method Total

depending on the surgical
method CE + CS CE CO CE with Partial resec-
curettage tion of the
jaw

Recurrence No Number without recurrence 11 50 4 3 2 70

% within no recurrence 15.7 71.4 5.7 4.3 2.9 100.0
% within surgical method 44.0 71.4 80.0 37.5 66.7 63.1
% of total number 9.9 45.0 3.6 2.7 1.8 63.1
Yes Number of recurrences 14 20 1 5 1 41
% within no recurrence 34.1 48.8 2.4 12.2 2.4 100.0
% within surgical method 56.0 28.6 20.0 62.5 33.3 36.9
% of total number 12.6 18.0 0.9 4.5 0.9 36.9
Total Total Number 25 70 5 8 3 111
% within no recurrence 22.5 63.1 4.5 7.2 2.7 100.0
% within surgical method 100.0 100.0 100.0 100,0 100.0 100.0
% of total number 22.5 63.1 4.5 7.2 2.7 100.0

Bold values indicate the total number of recurrences

is significantly better against the recurrence patient than neoplasia of a tumorous process after 2005. The WHO’s
another (see Table 3). classification, which in 2005 did justice to the new pathoge-
netic findings about the KCOT as a tumorous event, did not
lead to any discernible change in treatment. It has also not
Discussion been possible to reduce the enormous recurrence incidence
of 2–62%, which is confirmed by the result of the present
Only patients with confirmed histopathological findings and evaluation with a mean recurrence incidence of 36.9% [16].
the diagnoses KCOT and OKC were included in the evalu- Therefore, a better understanding of aetiopathogenesis and
ation. All 114 patients underwent primary surgery between factors influencing relapse behavior as well as prognosti-
1990 and 2010. Within this timespan, “keratocyst” were cally relevant factors to develop effective, low relapse thera-
treated as a dysontogenetic cyst starting in 1991 and as a pies for patients seems crucial. With a median of 42 years

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 J. Maxillofac. Oral Surg.

(median women = 41 years old/median men = 44 years old), Conclusion

the age range extends from twelve to 90 years and is thus in
agreement with a large part of the literature [17]. The large Cystectomy showed the best results in terms of surgical
standard deviation of 19 years suggests that odontogenic method used and postoperative recurrence rate, but even this
keratocysts develop largely independently of age [18]. Men method could not reliably prevent recurrences. Neverthe-
were twice as often affected by OKC as women in a ratio of less, many consider cystectomy alone to be an appropriate
2:1. This is similar to the results described in the literature therapy because, in addition to being a gentle procedure, it
[19]. The OKC were found significantly more frequently can reduce the recurrence rate and prolong the recurrence-
(p < 0.001) in the lower jaw (91.9%) than in the upper jaw free period. Close follow-up is required for early detection
(8.1%). This conspicuous frequency and the predilection of OKC. The follow-up intervals showed that annual clini-
site in the posterior mandibular angle range with 68.3% are cal and radiographic control of the affected jaw section by
largely consistent with the current literature [17, 19–21]. The three-dimensional imaging is recommended in the first three
fact that the odontogenic keratocysts develop from develop- postoperative years. Basically, very long-term clinical and
ment-related residues of Mallessez’s and Serres’ epithelial radiographic controls are recommended because recurrences
remains can explain the above-mentioned predilection in can occur even 12 years after surgery. Further randomized,
the mandible, since these cell residues often remain in the multicenter studies need to be performed to provide evi-
posterior region of the mandible during odontogenesis [22]. dence-based treatment recommendations.
The vast majority of OKCs (59.5%) were found to be asymp-
tomatic random findings in routine radiographic diagnostics
[20]. The rather high proportion of asymptomatic odonto- Funding Open Access funding enabled and organized by Projekt
genic keratocysts in this study is due to a small radiological DEAL. No funds, grants, or other support was received.
average size of 5.5 c­ m2 compared to the values of up to 19
Data Availability The data presented in this study are available on
­cm2 given in the literature [23]. The connection between the request from the corresponding author. The data are not publicly avail-
size and the rather late symptoms in the lower jaw can be able due to privacy regulations.
illustrated by the anterior–posterior growth direction in the
Conflict of interest The authors declare no conflict of interest. The
cancellous bone. This allows the odontogenic keratocysts
authors have no relevant financial or non-financial interests to disclose.
to assume considerable size before causing discomfort by
perforating the compact bone [22]. The size data of other Institutional Review Board Statement Ethical approval was waived
authors of up to 19 c­ m2 may explain their higher values of by the local Ethics Committee of University Hannover in view of the
retrospective nature of the study and all the procedures being per-
symptomatically diagnosed lesions [24]. Due to the predilec-
formed were part of the routine care.
tion site in the toothless area of the posterior mandible, only
32.4% of the cases studied had a close spatial relationship to Informed Consent Statement Patient consent was waived due to the
one or more teeth. 67.6% had no connection to teeth. In the retrospective study design.
literature, these data correlated with the size of the teeth and
deviated from the values of the present study [10]. As in the Open Access This article is licensed under a Creative Commons
literature, 98.2% of the KCOT were unilocular [20]. Large Attribution 4.0 International License, which permits use, sharing, adap-
tation, distribution and reproduction in any medium or format, as long
or multiple odontogenic keratocysts require an extended
as you give appropriate credit to the original author(s) and the source,
radiological diagnostic procedure such as a computer tomo- provide a link to the Creative Commons licence, and indicate if changes
gram (CT) or a digital volume tomography (DVT) in order were made. The images or other third party material in this article are
to determine the topographical-spatial extent more precisely included in the article’s Creative Commons licence, unless indicated
otherwise in a credit line to the material. If material is not included in
and to be able to plan therapeutic measures more specifically
the article’s Creative Commons licence and your intended use is not
[22]. In 45.9% of patients (n = 51), an augmentation proce- permitted by statutory regulation or exceeds the permitted use, you will
dure was required to restore functional stability of the jaw. need to obtain permission directly from the copyright holder. To view a
Most frequently (n = 38) autogenous bone was removed from copy of this licence, visit http://​creat​iveco​mmons.​org/​licen​ses/​by/4.​0/.
the iliac crest of the anterior superior iliac spine and used to
fill the defect. This means an additional intervention for the
extraction of bone material. However, clinical studies show References
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