J Stomatol Oral Maxillofac Surg 122 (2021) 192–198

Available online at

ScienceDirect
www.sciencedirect.com

Review

Prognostic evaluation of metastasizing ameloblastoma: A systematic

review of reported cases in literature
R. Hosalkar a, T.S. Saluja b,*, N. Swain a, S.K. Singh b,**
a
Department of Oral Pathology, MGM Dental College and Hospital, MGMIHS, Navi Mumbai, Maharashtra, India
b
Stem Cell/Cell Culture Lab, Center For Advance Research, King George’s Medical University, Chowk, Lucknow, Uttar Pradesh, India

A R T I C L E I N F O A B S T R A C T

Article history: Ameloblastoma is a benign odontogenic tumor which undergoes malignant transformation to
Received 29 June 2020 ameloblastic carcinoma. However, rarely it metastasizes without undergoing cytological malignant
Accepted 6 July 2020 changes, an entity referred to as Metastasizing Ameloblastoma (MA). Through this study, we aimed to
Available online 10 July 2020
review cases of MA reported since 2000 to explore the impact of clinico-demographic variables on its
prognosis. Based on PRISMA guidelines, a review of relevant literature from PubMed/Medline, Science
Keywords: Direct and Cochrane database was performed from January 2000 to March 2019. A total of 65 cases were
Metastasizing ameloblastoma
considered for further evaluation as per predefined inclusion and exclusion criteria. Results showed that
Metastasis
Recurrence
lungs followed by lymph nodes were the most common sites for benign metastatic deposits. Multiple
Survival recurrences and inadequate surgical removal increase the probability of distant metastatic spread.
Despite having benign cytological features, tumor recurrence and metastasis were associated with an
unfavorable clinical outcome in MA.
C 2020 Elsevier Masson SAS. All rights reserved.

1. Introduction histological and malignant clinical presentation is an oxymoron.

Ameloblastoma is the second most frequently encountered
benign odontogenic tumor. It exhibits slow growth, local invasion
and high rate of recurrences. Despite frequent local recurrences,
mortality is an extremely rare occurrence in ameloblastoma.
However, invasion to adjacent vital structures such as cranium in
cases of maxillary ameloblastoma, and loco-distant metastasis
generally results in significant morbimortality [1–4]. Ameloblas-
toma has been infrequently (< 1%) reported in literature to
demonstrate metastasis. Intriguingly, the metastatic deposits are
microscopically identical to the primary tumour [5]. This entity is
known as metastasizing ameloblastoma (MA). This unusual
behaviour was first reported almost nine decades ago by Simmons
(1928), followed by Vorzimer and Perla (1932) [6,7]. Often, a long-
time interval has been reported between primary tumor and
development of metastasis. The most common site of metastasis is
lungs and cervical lymph nodes [8,9].
The nature of MA has been a matter of debate owing to its
biological behaviour when compared to conventional ameloblas-
toma. The actual definition of MA made by combining benign
* Corresponding author.
** Corresponding author.
E-mail addresses: salujatajindersingh@gmail.com (T.S. Saluja),
satyendraks@kgmcindia.edu (S.K. Singh).
This amalgamation of contradictory clinical and histological
features highlights the exception to an otherwise invariable fact
that metastasis means cancer. Such tumors with complex biologic
behaviours are of interest for pathologists as well as surgeons. As
this entity is not much studied owing to rare occurrence, its clinical
course and prognosis is poorly defined. Through this study, we
aimed to analyse the clinico-demographic details of MA cases
reported in previous 19 years and address the clinical conditions
which increase the likelihood of metastasis in ameloblastoma. We
also evaluated the impact of clinical parameters on survival.
2. Materials & methods
2.1. Search strategy
In accordance with the guidelines of Preferred Reporting Items
for Systematic Review and Meta-Analysis (PRISMA), an electronic
search of PubMed/Medline, Science Direct and Cochrane database
was performed [10]. Boolean search with keywords ‘‘metastasis
and ameloblastoma’’ or ‘‘malignant ameloblastoma’’ or ‘‘metasta-
sizing ameloblastoma’’ were used. The retrieved record was
merged in a reference management software and duplicates were
removed. Thereafter, on the basis of a specified inclusion and
exclusion criteria, analysis of full text was carried out indepen-
dently by two authors (R.H and T.S.S) to rule out any selection bias.

https://doi.org/10.1016/j.jormas.2020.07.001
2468-7855/ C 2020 Elsevier Masson SAS. All rights reserved.
 R. Hosalkar et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 192–198 193

A total of 50 articles were included which reported 65 cases of MA
(Fig. 1).
Inclusion criteria:
 case reports with relevant clinico-demographic, therapeutic and
follow-up details of MA;
 relevant articles from references of published cases or reviews;
 case reports published between January 2000 and March 2019;
 case reports published in English language.
Exclusion criteria:
 reports with insufficient case details;
 published articles in languages other than English;
 letters to the editors or reviews.
2.2. Data extraction
Pertinent clinico-demographic details (gender, age at onset,
site, treatment modality, local or distant metastasis, recurrence
and follow-up) were evaluated. Age was categorised as children
and young adults ( 30 years), middle age (> 30 to  60 years) and
old age (> 60 years). Site of involvement were categorised as
maxilla, mandible and tumor involving maxilla/mandible with
other sites. These other sites included floor of mouth, maxillary
sinus, submandibular and parotid gland region, and orbital area.
Therapeutic management for MA included surgery, surgery with
chemotherapy or radiotherapy and chemotherapy or radiotherapy
only. Tumor recurrence was identified as present or absent while
metastasis as local, or distant with or without local spread. Both
recurrence and metastasis were considered after the initial
treatment provided. Follow up of cases was categorised into alive
with disease, free of disease, dead and lost to follow up. The data
obtained by T.S.S & R.H was further scrutinized by N.S & S.K.S to
maintain objectivity and avoid bias with qualitative data analysis.
2.3. Statistical analysis
All analysis mentioned in the current study was carried out
using SPSS 16.0 (SPSS for Windows, Version 16.0. Chicago, SPSS
Inc.). The baseline features for categorical variables were compared
using Chi2 test. Differences in variables relevant to survival such as
gender, age, tumor site, therapeutics, recurrence and metastasis,
were assessed using univariate and multivariate Cox Regression
analysis with a fixed reference for each category. Comparison was
done using 2-log likelihood ratio test. Kaplan–Meier analysis was
used to assess the effect of different variables on survival.
Censoring was carried out wherever survival data was unavailable.
Log-rank, Breslow and Tarone–Ware tests were used to compare
survival functions. In entire analysis, two-tailed tests were used
and P-value  0.05 was considered statistically significant.

3. Results

3.1. Demographic and baseline characteristics of MA

A total of 65 cases of MA retrieved from 50 articles as per

inclusion and exclusion criteria were included (Fig. 1). MA was
reported almost 1.85 times more frequently in males (64.6%)
as compared to females (35.4%). The mean age at diagnosis was
45 years. The middle age group (47.7%) showed high predilection
followed by children and young adults (27.7%), and old age

Table 1
Demographic characteristics of metastasizing ameloblastoma cases.

Variable Number Percentage

Gender
Male 42 64.6
Female 23 35.4
Age (y)
Below 31 18 27.7
31 to 60 31 47.7
Above 60 16 24.6
Site
Mandible 40 61.5
Maxilla 19 29.2
Mandible or maxilla involving other regions 6 9.2
Treatment
Surgery 31 47.7
Surgery with chemotherapy and/or radiotherapy 9 13.8
Chemotherapy or radiotherapy 5 7.7
Palliative or missing 20 30.8
Recurrence
Absent 49 75.4
Present 16 24.6
Metastasis
Local 21 32.3
Distant with or without local 44 67.7
Follow-up
AWD 19 29.2
FOD 13 20
Dead 12 18.4
LTF 21 32.3
Fig. 1. Search strategy for retrieving metastasizing ameloblastoma cases.
194 R. Hosalkar et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 192–198

individuals (24.6%). It was also noted that ameloblastoma
occurring in mandible metastasized more frequently (61.5%) as
opposed to maxilla (29.2) and maxilla/mandible with involvement
of other regions (9.2%). Surgical management (47.7%) was the
mainstay of treating MA while adjuvant treatment was given only
to a small percentage (13.8%) of cases. Almost one-fourth cases
(24.6%) presented with recurrence. Local metastasis was reported
in less than one-third cases (32.3%) as against distant metastasis
with/without local spread (67.7%). Nearly half of MA cases (47.6%)
were dead or alive with disease, while only one-fifth (20%) of cases
were completely free of disease. However, insufficient scientific
documentation of few MA cases prevented us from ascertaining
the status of almost one-third (32.3%) cases which were considered
lost to follow-up (Tables 1 and 2a).
The fact that benign tumor cells of ameloblastoma can
metastasize makes it a clinically distinct tumor entity. We
therefore seek to study the association of clinico-demographic
variables with metastasis. Distant with/without local metastasis
group had a significantly high association with age, mode of
treatment, and prognosis while a trend of association was evident
with recurrence. No significant difference was found with gender,
and site of tumor.

Table 2a
Baseline characteristics of metastasizing ameloblastoma cases.

Age Group P-value

Variable < 31 y 31–60 y > 60 y

Cases (n) % Cases (n) % Cases (n) %

Gender 0.032*
Male 8 44.40 20 64.50 14 87.50
Female 10 55.60 11 35.50 2 12.50
Site 0.046*
Mandible 15 83.30 18 58.10 7 43.80
Maxilla 1 5.60 10 32.30 8 50.00
Mandible or maxilla involving other regions 2 11.10 3 9.70 1 6.20
Treatment 0.832
Surgery 9 50.00 15 48.40 7 43.80
Surgery with chemotherapy and/or radiotherapy 4 22.20 4 12.90 1 6.20
Chemotherapy or radiotherapy 1 5.60 2 6.50 2 12.50
Palliative or missing 4 22.20 10 32.30 6 37.50
Recurrence 0.132
Absent 12 66.70 22 71.00 15 93.80
Present 6 33.30 9 29.00 1 6.20
Metastasis 0.016*
Local 7 38.90 5 16.10 9 56.20
Distant with or without local 11 61.10 26 83.90 7 43.80
Status < 0.0005*
AWD 6 33.33 13 41.95 0 0.00
FOD 8 44.44 4 12.90 1 6.20
Dead 2 11.11 6 19.35 4 25.00
LTF 2 11.11 8 25.80 11 68.80

* P-value  0.05.

Table 2b
Association of metastasis with other variables.

Variable Metastasis P-value

Local Distant with or without local

Cases (n) % Cases (n) %

Age (y) 0.016*

 30 7 38.90 11 61.10
31–60 5 16.10 26 83.90
> 60 9 56.20 7 43.80
Gender 0.427
Male 15 35.70 27 64.30
Female 6 26.10 17 73.90
Site 0.476
Mandible 12 30.00 28 70.00
Maxilla 8 42.10 11 57.90
Mandible or maxilla involving other regions 1 16.70 5 83.30
Treatment 0.014*
Surgery 16 51.60 15 48.40
Surgery with chemotherapy and/or radiotherapy 1 11.10 8 88.90
Chemotherapy or radiotherapy 0 0.00 5 100.00
Palliative or missing 4 20.00 16 80.00
Recurrence 0.051
Absent 19 38.80 30 61.20
Present 2 12.50 14 87.50
Status < 0.0005*
AWD 1 5.30 18 94.70
FOD 9 69.20 4 30.80
Dead 1 8.30 11 91.70
LTF 10 47.60 11 52.40
 R. Hosalkar et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 192–198 195

Distant metastasis group showed higher percentage of cases

Distant with or

0.759–48.887

0.183–63.914
without local
which succumbed to MA or were living with the disease.

Metastasis
Importantly, the distribution of cases which were lost to follow
up were almost similar in local and distant metastasis group, thus

3.415
6.093

0.411
0.089
increasing the likelihood of an unbiased statistical comparison
between groups (Table 2b).

1.355–699.345
1.143–11.393
Recurrence

Present 3.2. Univariate and multivariate analysis

30.778
0.029*

0.032*
3.609
Cox regression analysis of risk factors was carried to ascertain
the effect of prognostic variables on survival. In univariate analysis,
recurrence was the only factor which significantly affected the

0.088–30.965
0.246–5.219
Palliative or

overall survival. No significant difference was observed in other

variables -age, gender, site of tumor, treatment modality and
missing

1.134

1.648
0.872

0.739
metastasis.
Potential prognostic factors were included in multivariate
analysis. The risk of death was significantly increased in middle
Chemotherapy or

and old age individuals as compared to children/young age group.

0.377–242.583
0.847–27.973
radiotherapy

No significant gender-based risk was observed. Considerable

increase in hazard ratio was seen in cases treated with chemo/
4.867

9.566

0.171
0.076

radiotherapy alone while surgical management resulted in a

favorable outcome. Death risk was significantly increased in cases
with recurrence (Table 3).
and/or radiotherapy

3.3. Kaplan–Meier analysis

chemotherapy
Surgery with

0.382–7.699

0.044–2.008
Treatment

The Kaplan–Meier survival curves are shown in Fig. 2. In our

1.715

0.482

0.296

0.213

study, the Kaplan–Meier survival curves indicated that survival

was significantly poor in MA cases which recurred post-
therapeutic management (117.67 months, 95%CI 57.95–177.38;
maxilla involving

P = 0.018). Distant metastasis showed a trend for poor survival

0.012–12.104
other regions

(147.7 months, 95%CI 108.12–187.27; P = 0.054) as compared to

0.133–8.903
Mandible or

local metastasis (344.30 months, 95%CI 292.16–396.45). No

obvious difference in survival was noted based on age and gender.
1.089

0.937

0.387

0.589

MA involving maxilla only (257.45 months, 95%CI 174.41–340.49)

was found to be the associated with more favorable outcome as
0.118–9.864
0.081–1.434

compared to mandible (108.51 months, 95%CI 79.66–137.36) or

maxilla/mandible along with other regions (74.25 months, 95%CI
Maxilla

0.341

0.142

0.178

0.072

37.33–111.16). Surgical management (290.60 months, 95%CI

Site

214.69–366.51) conferred better survival rate as compared to

chemo/radiotherapy with (159 months, 95%CI 76.49–241.50) or
0.118–9.864
0.151–2.077

without (72 months, 95%CI 48.48–95.52) surgery (Table 4).

Gender

Female

0.561

0.387

1.081

0.945

4. Discussion

One of the characteristic hallmarks that distinguishes malig-

1.225–262.343
0.438–13.095

nant from benign tumor is metastasis. Metastasis generally leads

to considerable clinical complications and is the primary cause of
17.926

0.035*

tumor related morbidity as well as mortality. However, there are

2.394

0.314
> 60
Hazard ratio of death with metastasizing ameloblastoma.

several benign entities such as benign metastasizing leiomyoma,

metastasizing pleomorphic adenoma, giant cell tumor of bone that
0.655–102.847

show metastasis despite their benign histopathological nature. The

0.414–10.354

focus of current study was MA which also presents with benign

Variable

metastatic deposits. WHO (2005) categorized malignancy in

Age (y)

31–60

0.376

8.209

0.103

amleoblastoma into MA and ameloblastic carcinoma [11]. The

2.07

former does not show cytological atypia whereas the latter show
characteristic cytological features of malignancy even at the
Lower bound–upper bound

Lower bound–upper bound

distant metastatic site. However, in new classification of odonto-

genic tumors, WHO (2017) reclassified MA as a benign tumor,
though not unanimously [12]. With categorization of MA as a
benign entity, it may be construed that MA has perplexed
Hazard Ratio
Hazard ratio

Multivariate

pathologists, and the debate concerning its exact biological nature

is far from over. It is, therefore, critical to analyse the bizarre
Univariate

95.0% CI

95.0% CI
P-value

P-value

behaviour in amleoblastoma presenting as MA along with its

Table 3

prognostic implications. Thus, we carried out a systematic review

of the reported cases of MA in last 19 years.
196 R. Hosalkar et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 192–198

Fig. 2. Kaplan–Meier survival of metastasizing ameloblastoma.

MA is an ambiguous odontogenic tumor as it exhibits
characteristics of both benign ameloblastoma and its malignant
counterpart ameloblastic carcinoma. The underlying mechanism
to answer why a benign neoplasm like amleoblastoma metastasize
is not completely known. Many authors owe its development to
incomplete removal or tumor spillage during primary or recurrent
management. Literature on benign pleomorphic adenoma show-
ing metastasis points out that most of the cases experiencing
metastasis have undergone surgical management of the tumor one
or more times. This increases the likelihood of hematogenous
spread leading to distant metastasis. Similarly, reports on benign
metastasizing cutaneous fibrous histiocytoma and leiomyoma
suggests that cases with multiple recurrences are at higher risk of
developing metastasis [13–18]. However, others did not support
this hypothesis as distant foci of benign tumor cells are generally
destroyed by the natural immune mechanisms [19].
In the current study, we found that the overall occurrence of
distant MA was more than local MA. A trend for poor clinical
outcome (P = 0.054) was seen in cases with distant metastasis. Out
of 65 cases included in this study, 31 cases were treated only
surgically for primary lesion and/or metastasis of which only 10
(15.38%) presented with metastasis at the time of primary lesion.
For the remaining 21 cases the time for metastasis ranged from
3 months to 24 years (mean – 8.23 years).
In addition, out of 40 cases who underwent surgical manage-
ment with/without adjuvant treatment, 55% (22/40 case) showed
 R. Hosalkar et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 192–198 197

Table 4
Survival time (mean) in metastasizing ameloblastoma.

Variable Estimate (months) 95.0% CI P-value

Lower bound–upper bound

Age (y) 0.56

 30 212.8 172.096–253.504
31–60 206.268 111.151–301.386
> 60 208.103 94.947–321.258
Overall 220.366 151.795–288.936
Gender 0.374
Male 210.915 131.12–290.71
Female 197.143 146.645–247.641
Overall 220.366 151.795–288.936
Site 0.285
Mandible 108.517 79.666–137.367
Maxilla 257.455 174.412–340.497
Mandible or maxilla involving other regions 74.25 37.331–111.169
Overall 220.366 151.795–288.936
Treatment 0.233
21
Surgery 290.606 4.692–366.519
Surgery with chemotherapy and/or radiotherapy 159 76.496–241.504
Chemotherapy or radiotherapy 72 48.48–95.52
Palliative or missing 144.433 108.624–180.241
Overall 220.366 151.795–288.936
Recurrence
Absent 291.454 225.728–357.18 0.018*
Present 117.671 57.957–177.386
Overall 220.366 151.795–288.936
Metastasis 0.054
Local 344.308 292.16–396.455
Distant with or without local 147.703 108.129–187.277
Overall 220.366 151.795–288.936

metastasis. Further, 93.75% of recurrent cases (15/16 case) had growth is essentially slow and only locally invading unlike
history of surgical management of ameloblastoma and 87.5% metastatic deposits of cancer. Though current study has limita-
recurrences (14/16 case) presented with metastasis. This indicates tions due to small sample size owing to rarity of MA, significantly
that the possibility of incomplete removal or tumor spillage during important interpretations were made about biological behavior
treatment of ameloblastoma leading to MA cannot be negated. and survival of cases presenting with this inexplicable odontogenic
Thus ensuing meticulous management of ameloblastoma with tumor.
adequate margins may play a decisive role in preventing MA.
Considering the clinical impact of recurrence in MA, we found
5. Conclusion
that recurrence was associated with significant poor clinical
outcome (P < 0.05). Results of multivariate analysis and survival
MA is a rare clinical entity. The probability of developing MA is
curves showed that recurrence posed markedly high death risk.
difficult to predict on clinical presentation or histopathological
The survival in recurrent cases was approximately 2.5 times less as
basis, rather early or multiple recurrences following ameloblas-
compared to cases without recurrence. On other hand, though the
toma treatment is a possible prognostic indicator and warrants
impact of metastasis on survival was not found to be statistically
thorough follow-up. Two possibilities for the dichotomous nature
significant, the average survival period in cases with distant
of MA can be speculated. First, it is a corollary of a long standing
metastasis was 2.34 times less as compared to local metastasis. As
ameloblastoma in which metastasis is the consequence of
a matter of fact, distant metastasis is generally associated with
hematogenous spread of tumor cells during surgical treatment
poor outcome. Hence, it is essential here to note that with these
accompanied with or without multiple recurrences. Second, a set
observations we are not highlighting whether survival was poor in
of additional genetic alterations in ameloblastoma may presum-
local or distant metastasis group. Of essence is the finding that, if
ably drive metastatic behavior which is not accompanied with
ameloblastoma undergoes metastasis, despite having benign
obvious histopathological changes. Under such circumstances,
histology it markedly affects clinical outcome.
clonal selection of a single metastatic clone can result in loco-
Currently, there is no foolproof criteria to predict how, when
distant metastasis.
and which case of ameloblastoma will metastasize. However, few
Due to the absence of well-recorded reports and its rare
clinical factors viz. large size of primary tumor, rapid local invasion,
occurrence, authors advocate for detailed individual case studies to
protracted clinical course, inadequate primary surgical removal,
avoid discrepancies in documenting its behavior. Additionally,
and multiple recurrences dictate higher plausibility of metastasis
each case of MA should be investigated from a methodological
in ameloblastoma. Thus, MA can only be construed a posteriori after
viewpoint to appraise association between metastatic process and
the development of metastasis. From a clinical perspective, it is
benign histology for better therapeutic management.
necessary here to highlight the fact that behavioral code-1 is
assigned for uncertain and unknown behavior of neoplasms by the
International Classification of Diseases for Oncology (ICD-O) [20]. Contribution
With this review, we opine that metastasis in ameloblastoma is
usually associated with an unfavorable clinical outcome. While R.H. and T.S.S contributed in data acquisition, analysis and
conducting this study, we found that the morbimortality increased interpretation. All authors contributed in study concepts and
with benign metastatic lesions in MA, despite the fact that their interpretation. Data obtained by R.H & T.S.S was reassessed
198 R. Hosalkar et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 192–198
individually by N.S & S.K.S. Dr. Vidya Wadedkar has helped in
statistical analysis. R.H, T.S.S and N.S prepared the manuscript. All
authors reviewed and approved the manuscript.
Funding
The work was supported by Department of Health Research, New Delhi,
India [No.- R.12014/08/2017-HR].
Disclosure of interest
The authors declare that they have no competing interest.
Acknowledgements
The work was supported by Department of Health Research,
New Delhi, India [No.- R.12014/08/2017-HR].
References
[1] Zwahlen RA, Grätz KW. Maxillary ameloblastomas: a review of the literature
and of a 15-year database. J Craniomaxillofac Surg 2002;30(5):273–9. http://
dx.doi.org/10.1016/s1010-5182(02)90317-3.
[2] Etetafia MO, Arisi AA, Omoregie OF. Giant ameloblastoma mortality; a conse-
quence of ignorance, poverty and fear. Case Rep 2014;2014. http://dx.doi.org/
10.1136/bcr-2013-201251 [bcr2013201251].
[3] Milman T, Ying GS, Pan W, LiVolsi V, Ameloblastoma:. 25 year experience at a
single institution. Head Neck pathol 2016;10(4):513–20. http://dx.doi.org/
10.1007/s12105-016-0734-5.
[4] McClary AC, West RB, McClary AC, Pollack JR, Fischbein NJ, Holsinger CF, et al.
Ameloblastoma: a clinical review and trends in management. Eur Arch Otorhi-
nolaryngol 2016;273(7):1649–61. http://dx.doi.org/10.1007/s00405-015-3631-8.
[5] Palanisamy JC, Jenzer AC. Cancer, Ameloblastoma. InStatPearls,[Internet].
Treasure Island (FL): StatPearls Publishing. SAvailable from:https://
www.ncbi.nlm.nih.gov/books/NBK545165/?report=classic; 2020.
[6] Simmons CC. Adamantinoma. Ann Surg 1928;88(4):693.
[7] Vorzimer J, Perla D. An instance of adamantinoma of the jaw with metastases
to the right lung. Am J Pathol 1932;8(4):445.
[8] Dissanayake RK, Jayasooriya PR, Siriwardena DJ, Tilakaratne WM. Review of
metastasizing (malignant) ameloblastoma (METAM): pattern of metastasis
and treatment. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol
2011;111(6):734–41. http://dx.doi.org/10.1016/j.tripleo.2010.12.018.
[9] Bi R, Shen L, Zhu X, Xu X. Malignant ameloblastoma (metastatic ameloblas-
toma) in the lung: 3 cases of misdiagnosis as primary lung tumor with a unique
growth pattern. Diagn Pathol 2015;10(1):123. http://dx.doi.org/10.1186/
s13000-015-0367-0.
[10] Moher D, Liberati A, Tetzlaff J, Altman DG. Preferred reporting items for
systematic reviews and meta-analyses: the PRISMA statement. Ann Intern
Med 2009;151(4):264–9. http://dx.doi.org/10.1371/journal.pmed.1000097.
[11] Sciubba JJ, Eversole LR, Slootweg PJ. Odontogenic/ameloblastic carcinomas. In:
Barnes L, Eveson JW, Reichart P, editors. World Health Organization. Classifi-
cation of tumours pathology and genetics of head and neck tumours. Lyon:
IARC Press; 2005. p. 283–327.
[12] Speight PM, Takata T. New tumour entities in the 4th edition of the World
Health Organization Classification of Head and Neck tumours: odontogenic
and maxillofacial bone tumours. Virchows Archiv 2018;472(3):331–9. http://
dx.doi.org/10.1007/s00428-017-2182-3.
[13] Patton KT, Cheng L, Papavero V, Blum MG, Yeldandi AV, Adley BP, et al. Benign
metastasizing leiomyoma: clonality, telomere length and clinicopathologic
analysis. Modern Pathol 2006;19(1):130–40.
[14] Knight J, Ratnasingham K. Metastasising pleomorphic adenoma: systematic
review. Int J Surg 2015;19:137–45. http://dx.doi.org/10.1016/j.ijsu.2015.04.084.
[15] Nouraei SA, Ferguson MS, Clarke PM, Sandison A, Sandhu GS, Michaels L, et al.
Metastasizing pleomorphic salivary adenoma. Arch Otolaryngol Head Neck
Surg 2006;132(7):788–93. http://dx.doi.org/10.1001/archotol.132.7.788.
[16] Unni K, Dahlin D. Dahlin’s bone tumors. Philadelphia: Lippincott-Raven; 1996.
p. 263–83.
[17] Doyle LA, Fletcher CD. Metastasizing ‘‘benign’’ cutaneous fibrous histiocy-
toma: a clinicopathologic analysis of 16 cases. Am J Surg Pathol
2013;37(4):484–95. http://dx.doi.org/10.1097/PAS.0b013e31827070d4.
[18] Barnaś E, Raś R, Skre˛t-Magierło J, Wesecki M, Filipowska J, Ksia˛żek M, et al.
Natural history of leiomyomas beyond the uterus. Medicine 2019;98(25).
http://dx.doi.org/10.1097/MD.0000000000015877.
[19] Houston G, Davenport W, Keaton W, Harris S. Malignant (metastatic) amelo-
blastoma: report of a case. J Oral Maxillofac Surg 1993;51(10):1152–7. http://
dx.doi.org/10.1016/s0278-2391(10)80458-6.
[20] Fritz A, Percy C, Jack A, Shanmugaratnam K, Sobin L, Parkin DM, et al.
International classification of diseases for oncology. Geneva: 3rd edition,
1st revision, World Health Organization; 2013. p. 4–6.