1. The study analyzed 147 cases of central mucoepidermoid carcinoma (CMC) reported in 36 publications to understand clinical aspects, histology, treatment and prognostic factors. 2. It found that CMC was more common in females than males, most often in the mandible, and usually low grade histologically. The main treatment was surgical resection. 3. Significant prognostic factors associated with worse survival in multivariate analysis included male sex, high histological grade, conservative rather than radical treatment, and lymph node metastasis.

1. Review
Central mucoepidermoid carcinoma: An up-to-date analysis of 147
cases and review of prognostic factors
Lucas Lacerda de Souza a, *
, Flavia Sirotheau Correa Pontes a
,
Helder Antonio Rebelo Pontes a
, Nicolau Conte Neto a
,
Waldner Ricardo Souza de Carvalho b
, Douglas Magno Guimar~aes a
a
Oral Surgery and Pathology Department, Jo~ao de Barros Barreto University Hospital/Federal University of Para, Belem, Para, Brazil
b
Oral Diagnosis Department, Semiology and Oral Pathology Areas, Piracicaba Dental School, University of Campinas, Piracicaba, S~ao Paulo, Brazil
a r t i c l e i n f o
Article history:
Paper received 13 July 2017
Accepted 24 October 2017
Available online 31 October 2017
Keywords:
Central mucoepidermoid carcinoma
Bone lesion
Jaw bones
Prognostic
a b s t r a c t
Objective: To integrate the available data published on central mucoepidermoid carcinoma (CMC) into a
comprehensive analysis of its clinical aspects, histology, treatment, and prognostic factors.
Methods: An electronic search was undertaken in July 2017. Eligibility criteria for publications included
having clinical, histological, treatment, and time of follow-up data to confirm the diagnosis.
Results:
36 publications (147 CMCs) were included.
The lesion showed a slightly higher prevalence among women than men.
There was a high prevalence in the fifth to seventh decades of life.
The most common symptom was swelling and the most commonly observed area was the
mandible.
Histologically, low-grade tumors were the most common.
The treatment of choice was tumor resection.
In addition, gender, histological grade, conservative treatment, and lymph node metastasis were
significant independent prognostic factors.
Conclusion: Male patients with histological grade III CMC of the jaw bone, who used conservative
treatment as the main treatment, and presented with lymph node metastasis, were more likely to have a
worse prognosis.
© 2017 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights
reserved.
1. Introduction
Central mucoepidermoid carcinoma (CMC) was first described
by Lepp (1939). The author presented a case of a 66-year-old
woman who presented with CMC of the mandible. Almost 25 years
later, Bhaskar (1963)reported two cases, discussing the criteria for
their origin, histological composition, and possible explanations for
tumor pathogenesis. In 1991, Seifert and Sobin recommended the
term ‘mucoepidermoid carcinoma’ after a systematic review of its
histology and degree of differentiation for the World Health
Organization (Seifert and Sobin, 1991). In addition, Waldron and
Mustoe suggested the inclusion of CMC in primary intraosseous
carcinoma of the jaw as type 4 (Waldron and Mustoe, 1989).
In 2008, the Armed Forces Institute of Pathology published
the inclusion of CMC in the intraosseous lesions category, and
its differential diagnosis under clinical/histological/radiological
characteristics (Ellis and Auclair, 2008).
CMC is considered a rare lesion, so there is limited information
in the literature regarding its clinical aspects, histology, treatment,
and prognostic factors. The epidemiological study of such rare
lesions is of great importance in allowing pathologists and sur-
geons to make informed decisions and refine treatment plans in
order to optimize clinical outcomes (Chrcanovic and Gomez, 2016).
* Corresponding author. Jo~ao de Barros Barreto University Hospital, Department
of Surgery and Oral Pathology, Mundurucus Street, Nº 4487 66073-000, Belem, Para,
Brazil.
E-mail address: lucaslac@hotmail.com (L.L. de Souza).
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery
journal homepage: www.jcmfs.com
https://doi.org/10.1016/j.jcms.2017.10.020
1010-5182/© 2017 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 162e167

2. The aim of this study was to integrate the available data published
in the literature on CMC into an up-to-date, comprehensive
analysis of its clinical, histological, treatment, and prognostic
factors.
2. Materials and methods
This study followed the PRISMA Statement guidelines (Moher
et al., 2009).
2.1. Search strategies
An electronic search without time restrictions was undertaken
in June 2017 in the following databases: Pubmed, National Library of
Medicine, the National Institute of Health, and Science Direct. The
following terms were used separately in the search strategies:
‘central mucoepidermoid carcinoma’ and ‘intraosseous mucoepi-
dermoid carcinoma’.
The reference list of identified studies and relevant reviews on
the subject were also scanned for possible additional studies.
Publications with lesions identified by other authors as being CMC,
even without the term ‘central/intraosseous mucoepidermoid car-
cinoma’ in the title of the article, were also re-evaluated by an
author of our study.
2.2. Inclusion and exclusion criteria
Eligibility criteria included publications reporting cases of CMC.
The studies needed to have enough clinical/histological informa-
tion to confirm the diagnosis. Allon et al. (2006). described the
microscopic parameters of salivary gland tumors, based on bicel-
lular theory, as follows:
Duct luminal cells and/or acinar cells plus myoepithelial cells
Duct luminal cells or acinar cells
Myoepithelial cells only
Positive mucin staining
Present intraosseous location/evolvement
All cases that did not include theseparameters were not
included in our analysis.
Histopathology descriptions for mucoepidermoid carcinoma
consider three grades of differentiation, depending on the intra-
cystic component, neural invasion, necrosis, mitotic activity, and
pleomorphism (Auclair et al., 1992). Histological grading was based
on AFIP (Ellis and Auclair, 2008), published in the World Health
Organization Classification of Tumors.
In order to understand and clearly identify the microscopic
parameters described in the text, only articles published in the
English language were included. Controlled clinical trials, cohort
studies, cross-sectional studies, case-control studies, case series,
and case reports were included.
Exclusion criteria were immunohistochemical studies, histo-
morphometric studies, radiological studies, genetic expression
studies, histopathological studies, cytological studies, cell prolifer-
ation/apoptosis studies, and in vitro studies, unless any of these had
reported any cases with enough clinical, histological grading, and
radiological information. In addition, papers that did not report
maxilla and mandible as the first location, histological grade,
treatment, and survival time were excluded.
2.3. Study selection
The titles and abstracts of all reports identified through the
electronic searches were read independently by four authors (LLS,
NCN, WRSC, and DMG). For studies appearing to meet the inclusion
criteria, or for which there were insufficient data in the title and
abstract to make a clear decision, the full report was obtained. Any
disagreement was solved by discussion among the authors. The
clinical and radiological aspects, as well as the histological de-
scriptions of the lesions reported by the publications, were thor-
oughly assessed by two authors of our sent study (FSCP and HARP),
who are experts in oral pathology, in other to confirm the diagnosis.
2.4. Data extraction
Three authors independently extracted data using specially
designed data extraction forms. Any disagreement was solved by
discussion. For each of the identified studies included, the following
data were then extracted, when available: author and year of
publication, number of patients, patient's sex (male or female), age
(40 or 40), evolution time, first symptoms, tumor location
(mandible or maxilla), lymph node metastasis, histological grade
(low, low-to-intermediate, intermediate, and high; the grading was
established by three doctors specialized in oral pathology e HARP,
FSCP, and DMG), treatment (radical surgery [tumor and margin
excision without lymph node dissection], conservative sur-
gery, radical surgery þ neck dissection, radical surgery þ
radiotherapy þ neck dissection, radical surgery þ radiotherapy þ
chemotherapy), and follow-up (died of the disease, no evidence of
disease, alive with disease, recurrence, and metastasis). Contact
was made with authors for possible missing data.
2.5. Analysis
The mean and percentages were presented as descriptive sta-
tistics. Overall survival rates were estimated by KaplaneMeier
analysis and compared using a log-rank test. A p-value 0.05 was
considered statistically significant. The factors that were signifi-
cantly associated with CMC prognosis in univariate analysis were
introduced stepwise into a Cox proportional hazard model to
identify the independent predictors of survival. Data were analyzed
using the Statistical Package for Social Sciences software for Win-
dows, version 23.0 (SPSS Inc, Chicago, IL, USA).
3. Results
3.1. Literature search
The study selection process is summarized in Fig.1. The database
search strategy resulted in 2,738 papers. 459 articles were cited in
more than one database (duplicates). Four authors (LLS, NCN,
WRSC, and DMG) independently screened the abstracts for those
articles related to the focus question. Of the resulting 2,279 studies,
1,899 were excluded for not being related to the topic. The full-text
reports of the remaining 330 articles were evaluated and led to the
exclusion of 294 articles because they did not have mandible and
maxilla as the primary site, histological grading, or data on treat-
ment or survival time. Thus, 36 publications were included in the
review.
3.2. Description of the studies and analyses
Thirty-six publications, reporting 147 CMCs, were included in
our study. Table 1 presents demographic information, clinical fea-
tures, and survival analysis for CMCs. The lesion was slightly more
prevalent in women than in men, with a male:female ratio of 1:1.07.
The mean age of the patients was 46.51 years (range 11e79 years);
men were older (mean age 47.18 years; range 11e78 years) than
women (mean age 45.89 years; range 8e79 years). Fig. 2 shows the
L.L. de Souza et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 162e167 163

3. distribution of the lesions according to age and gender, with the
highest prevalence in the fifth to seventh decades of life. The most
commonly reported symptoms were swelling, pain, and numbness.
The lesions were more prevalent in the mandible than in the
maxilla. Seventeen patients presented regional (lymph node)
metastasis on clinical exam.
The histopathological tumor type most commonly observed was
low-grade (54.4%), followed by intermediate (22.2%), low-to-
intermediate (12.9%), and high (10.2%).
The most common treatments were radical surgery alone (63;
42.9%), conservative surgery (8; 8.2%), radical surgery and neck
dissection (20; 13.6%), radical surgery, radiotherapy and neck
dissection (11; 7.5%), radical surgery, radiotherapy and chemo-
therapy (3; 2.0%), and others (42; 25.8%). Among the 38 patients
who underwent neck dissection, 17 cases (44.73%) had regional
lymph nodes metastasis.
The mean follow-up was 49.71 months (range 1e384 months),
and revealed that 126 (85.7%) patients were alive with no evidence
of disease, 16 (10.9%) patients had died of the disease, three (2.0%)
had recurrence but were alive, while two (1.36%) had died of
another cause. Among the patients who had died of the disease,
seven were classified as high grade, seven as intermediate grade
and two as low grade. The treatments employed in the cases of
patients who died were total tumor resection (10 cases) and con-
servative treatment (six cases).
Sixteen patients (10.88%) had a local recurrence at a mean in-
terval of 51.93 months (range 1e240 months). Distant metastasis
affected three patients (2.0%), with reports of metastasis in the
lungs (1), clavicle (1), and knee (1). Among these, one case had CMC
in the mandible and two in the maxilla; two of these cases were
classified as low-grade CMC, and one was classified as high grade.
Concerning the individuals who presented with recurrence, 11
cases were classified as low grade, three as intermediate grade, and
one as high grade.
Univariate survival analysis of clinical variables and
KaplaneMeier analysis revealed that an age older than 40 years
(p ¼ 0.006), male gender (p ¼ 0.014), high-grade tumor
(p 0.0001), conservative treatment (p ¼ 0.002), recurrence
(p ¼ 0.003), and metastasis (p ¼ 0.003) were statistically significant.
Multivariate survival analysis revealed that gender, histological
grade, conservative treatment, and lymph node metastasis were
independent significant prognostic factors. The logistic regression
showed that the odds ratio of death among males was 4.7 times
more (p ¼ 0.047) than among females (Fig. 3A). The odds of death
for histological grade-III tumors were 2.7 times more than for
grade-I tumors (p ¼ 0.006) (Fig. 3b). The odds of death among those
Fig. 1. Study screening process.
Table 1
Analysis of demographic, clinical, and survival aspects of central
mucoepidermoid carcinoma cases (n ¼ 147) described in the
literature.
Variables
Age, n (%)a
40 51 (34.7%)
40 96 (65.3%)
Gender, n (%)a,b
Men 71 (48.3%)
Women 76 (51.7%)
Primary location, n (%)
Maxilla 54 (36.7%)
Mandible 93 (63.3%)
Lymph node metastasis, n (%)a,b
Yes 17 (11.6%)
No 130 (88.4%)
Histological grade, n (%)a,b
Low 80 (54.4%)
Low-to-intermediate 19 (12.9%)
Intermediate 33 (22.4%)
High 15 (10.2%)
Treatment, n (%)a,b
RS 63 (42.9%)
CS 8 (8.2%)
RS þ ND 20 (13.6%)
RS þ RT þ ND 11 (7.5%)
RS þ RT þ CT 3 (2%)
Other 42 (25.9%)
Recurrence, n (%)a
Yes 16 (10.9%)
No 131 (89.1%)
Metastasis, n (%)
Yes 3 (2.0%)
No 144 (98.0%)
UAeunivariate analysis, MAemultivariate analysis, RSeradical
surgery, CSeconservative surgery, NDeneck dissection, RTera-
diotherapy, CTechemotherapy.
a
Significant result under univariate analysis.
b
Significant result under multivariate analysis. p-value: 0.05.
Fig. 2. Distribution of CMC according to age and gender.
L.L. de Souza et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 162e167164

4. receiving conservative treatment were 3.4 times more than for
those receiving radical surgery (p ¼ 0.001) (Fig. 3c). Finally, the
odds of death among patients who presented with lymph node
metastasis were 4.1 times more than for patients with no lymph
node metastasis (p ¼ 0.004) (Fig. 3d).
4. Discussion
Among the central malignant salivary gland tumors, mucoepi-
dermoid carcinoma is the most common (Abu-Karaky et al., 2012;
Bell et al., 2016; Maremonti et al., 2001; Waldron and Koh, 1990;
Rathore et al., 2014; Li et al., 2008; do Prado et al., 2007). To
establish a central origin of CMC, several criteria are accepted,
including: absence of any primary lesions in the salivary glands or
other tissues that can mimic the histological architecture of salivary
lesions; absence of any odontogenic tumors; radiographic evidence
of bone osteolysis; integrity of the cortical plates; positive mucin
staining; and microscopic confirmation of the diagnosis. Several
theories have been introduced to explain the origin of CMC, such as
(a) ectopic salivary gland tissue that becomes entrapped in the jaws
during development, (b) neoplastic transformation of the epithelial
lining of an odontogenic cyst, or (c) maxillary sinus or submucosal
mucous gland with intraosseous extension (Rathore et al., 2014; Baj
et al., 2002).
In our study, we observed that peak age of occurrence of CMC
ranged from 50 to 70 years of age, which is in agreement with other
previous findings (Li et al., 2008; Browand and Waldron, 1975; Dain
et al., 2015; Zhou et al., 2012). We found a mean age of 46.51 years,
with a higher age among men than women. Women were slightly
more affected than men (ratio male:female, 1:1.07). We also
observed that the most prevalent symptom was swelling, which
corroborates the information previously published in the literature
(Li et al., 2008; Jiang et al., 2007; Martínez-Madrigal et al., 2000). Our
literature review demonstrated that 63.75% of tumors affected the
mandible and 36.25% affected the maxilla (Abu-Karaky et al., 2012).
The mucoepidermoid carcinoma grading for prognostic pur-
poses is based on a histological characteristic of the lesion and in-
dicates the degree of cystic development and squamous cell
differentiation (Kanmani and Daniel, 2014; He et al., 2012). There
are several grading systems for mucoepidermoid carcinoma, the
most popular being that of the Armed Forces Institute of Pathology
(AFIP) (Ellis and Auclair, 2008). These grading systems assess a
similar set of parameters and are points based, assigning point
values to cytomorphological and architectural features, including
perineural and angiolymphatic invasion. Regarding histological
grade, 80 patients (54.4%) were classified as low grade, 19 patients
(12.9%) as low-to-intermediate grade, 33 (22.4%) as intermediate
grade, and 15 (10.2%) as high grade. This is consistent with previous
case series that show a higher incidence of low-grade tumors in
CMC cases compared with high-grade tumors (Zhou et al., 2012; He
et al., 2012; Pires et al., 2003).
We observed that treatments involving resection of the lesion
presented a better prognosis (tumor and margin excision without
lymph node dissection); this is in agreement with the study by
Abu-Karaky et al., which showed that only 4% of CMCs recurred
after radical methods (Abu-Karaky et al., 2012). Radical surgery
associated with neck dissection was reported in 38 cases; 15 of
these had metastatic lymph node involvement and nine presented
as high grade in histological analysis. We suggest that the role of
neck dissection in CMC without lymph node involvement needs to
be better established.
Regional metastasis and local recurrence were found in 11.56%
and 10.73% of cases, respectively. We also observed that the mean
follow-up was 49.71 months, with 128 patients alive with no
evidence of disease during follow-up, and 16 dying from the
disease. Although we have demonstrated that CMC presents with
a good prognosis (Jiang et al., 2007; He et al., 2012; Tornes et al.,
1987), we also emphasize that malignant salivary gland tumors
are characterized by possible late recurrence and metastasis,
so patients should be followed up over long periods after
Fig. 3. KaplaneMeier graphics evidencing statistically significant results for gender (p-value: 0.047) (A), lymph node metastasis (p-value: 0.004) (B), histological grade (p-value:
0.006) (C), and conservative treatment (p-value: 0.001) (D).
L.L. de Souza et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 162e167 165

5. treatment (Zhou et al., 2012; Pires et al., 2003; Verma et al.,
2014), given that even after 240 months some patients pre-
sented with recurrence.
Our study found that gender is a significant prognostic factor,
although most high-grade tumors are found in male patients (Bell
et al., 2016; Tornes et al.,1987). Histological grade is one of the most
important prognostic factors in CMC; patients with high-grade
tumors had a 2.7 times higher risk of death compared with pa-
tients with low-grade tumors (Bell et al., 2016; Martínez-Madrigal
et al., 2000; He et al., 2012; Tornes et al., 1987). Our study is
consistent with the studies of Zhou et al. and He et al., which
highlighted the importance of the tumor's histological grade when
prognosis is evaluated. In addition, the surgical procedure
employed was a significant prognostic factor; the odds of death
among those with conservative treatment was 3.4 times higher
than for those who underwent radical surgery (Zhou et al., 2012; He
et al., 2012). Radical surgery has been reported as the treatment of
choice in some case series (Bell et al., 2016; Li et al., 2008; Zhou
et al., 2012; Martínez-Madrigal et al., 2000; He et al., 2012).
Although just a few cases presented with lymph node metastasis,
these patients showed poor prognosis and had a 4.1 times higher
risk of death than patients with no lymph node metastasis (Bell
et al., 2016). Metastasis is considered rare (2.01%), but can occur
in patients with CMC, even after a significant time since treatment;
this is why long follow-up times are a necessity (Bell et al., 2016;
Lebsack et al., 1990; Kochaji et al., 2004; Bell et al., 2011).
5. Conclusions
CMC in the jaw bones is a neoplasia with a low propensity for
local recurrence. Male gender, high-grade tumors, conservative
treatment, and the occurrence of regional lymph node metastasis
are all associated with poor prognosis. An effective follow-up is
necessary to ensure the success of treatment, because recurrence
has been observed after long periods of follow-up.
Funding source
This research did not receive any specific grant from funding
agencies in the public, commercial, or not-for-profit sectors.
Conflict of interest
The authors declare that they have no conflict of interest. This
research received no specific grant from any funding agency in
public, commercial, or not-for-profit sectors.
Author Contributions
LLS: conception and design, acquisition of data, statistical anal-
ysis, drafting the article, revising the article critically for important
intellectual content, final approval of the version to be published.
FSCP: conception and design, revising the article critically for
important intellectual content, final approval of the version to be
published.
HARP: conception and design, interpretation of data, revising
the article critically for important intellectual content, final
approval of the version to be published.
WRSC: conception and design, statistical analysis, drafting the
article, revising the article critically for important intellectual
content, final approval of the version to be published.
NCN: conception and design, acquisition of data, statistical anal-
ysis, drafting the article, revising the article critically for important
intellectual content, final approval of the version to be published.
DMG: conception and design, acquisition of data, statistical
analysis, drafting the article, revising the article critically for
important intellectual content, final approval of the version to be
published.
Acknowledgements
No acknowledgements.
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