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a b s t r a c t
Article history:
Paper received 23 June 2013
Accepted 8 October 2013
Objective: We report two rare cases of lung metastasis from maxillary ameloblastoma, in order to review
its risk and analyse the types of metastases that can present with this disease.
Methods: A 40-year-old male with multiple recurrences and a 46-year-old female, who had undergone
successful surgical treatment of a maxillary ameloblastoma, presented with metastatic lesions. The
primary tumour and metastases were benign in both patients. We reviewed and analysed 20 cases of the
same condition reported in recent years.
Results: Our initial treatment for the primary maxillary lesion was performed more than 10 years before
the pulmonary lesions presented. Due to the aggressive nature of this tumour, metastases in the lungs
and cervical lymph nodes (male patient) were conrmed.
Conclusion: These cases presented a diagnostic challenge due to the multiple and varied sites of recurrence, which indicate the natural behaviour of this tumour. Different routes of metastasis can occur,
including implanting, haematogenous, and lymphatic spread. CT-guided percutaneous transthoracic lung
biopsy is an important method to conrm metastatic ameloblastoma.
2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights
reserved.
Keywords:
Ameloblastoma
Metastases
Histopathology
1. Introduction
2. Case reports
2.1. Case 1
1010-5182/$ e see front matter 2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.jcms.2013.10.010
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Fig. 3. Computed tomography shows metastatic lymph nodes in the superior mediastinum with destruction of the second rib (arrow).
2.2. Case 2
A 46-year-old female underwent extended resection of the right
maxilla. 15 years later, in 2011, multiple nodules in both lungs were
found on routine chest X-ray. Thoracic CT (Fig. 4) and CT-guided
percutaneous transthoracic lung biopsy (Fig. 5) conrmed metastatic ameloblastoma. Second opinion consultation at the University of California, Los Angeles concurred with the diagnosis.
3. Discussion
Ameloblastoma is a histologically benign, but locally invasive
tumour with a high likelihood of recurrence, but rarely metastasizing. The incidence of malignancy/metastasis in relation to
ameloblastoma has been reported as 2%, but more realistically is far
Fig. 2. The lung metastasis is similar to the primary tumour, demonstrating islands of
epithelium with prominent basaloid and columnar peripheral cells and evidence of cell
streaming (H&E, 100).
less (Houston et al., 1993). Many cases of metastasis of ameloblastomas are linked to multiple operations or recurrences (Luo
et al., 2012), and furthermore, it is reported that malignant transformation was observed in patient with multiple recurrences (Lin
et al., 2013). Most reported cases of metastatic ameloblastoma
suggest haematogenous or lymphatic spread (Houston et al., 1993).
Another unusual mechanism (aspiration of tumour cells from the
primary lesion during surgery) was described by Vorzimer and
Perla in 1932, which may contribute to pulmonary, lymphatic, or
haematogenous spread (Vorzimer and Perla, 1932). This hypothesis
was supported by the presence of tumour in the bronchi and
bronchioles. Nonetheless, diffusely scattered metastatic lesions in
both lungs and in the surrounding vasculature is more likely the
result of haematogenous spread (Henderson et al., 1999).
The most common site of metastases in ameloblastoma is the
lung, followed by cervical lymph nodes, brain, and bone (Eliasson
et al., 1989). We analysed 20 cases in addition to our two cases
reported in the global literature from 1999 to 2013 (Table 1). We
Primary
site
Site of
metastasis
Number of
surgeries
First
metastasis
(years)
Henderson et al.
(1999)
Onerci et al.
(2001)
Ciment and Ciment
(2002)
Campbell et al.
(2003)
Zarbo et al.
(2003)
Hayakawa et al.
(2004)
Hasim et al.
(2007)
Gilijamse et al.
(2007)
Cardoso et al.
(2009)
Reid-Nicholson et al.
(2009)
Senra et al.
(2008)
Papaioannou et al.
(2009)
Devenney-Cakir et al.
(2010)
Dissanayake et al.
(2011)
Amzerin et al.
(2011)
Lai and Wang
(2011)
Golubovi
c et al.
(2012)
Luo et al.
(2012)
Berger et al.
(2012)
Lin et al. 2013
Present case 1
Mandible
Lungs
Multiple
33
Maxilla
Lungs
Multiple
12
Mandible
Lungs
29
Mandible
Lungs
19
Maxilla
Iliac
Multiple
15
Mandible
Lungs, kidney
Mandible
Lungs
Multiple
18
Mandible
Multiple
13
Mandible
Cervical
lymph nodes
Cervical
lymph nodes
Cervical
lymph nodes
Lungs
Primary
diagnosis
Primary
diagnosis
7
Mandible
Lungs
Multiple
27
Mandible
Lungs, skull
Mandible
23
Mandible
Cervical
lymph nodes
Lungs
Multiple
Maxilla
Lungs
Multiple
Mandible
Mandible
Cervical
lymph nodes
Lungs
Primary
diagnosis
29
Maxilla
Lungs
Maxilla
Maxilla
Multiple
Multiple
8
13
Present case 2
Maxilla
Lungs
Lungs, cervical
lymph nodes
Lungs
15
e303
Mandible
Mandible
From these ndings, it is important to note that multiple operations or recurrences are the risk factors for ameloblastoma
metastasis; meanwhile, patients without multiple recurrences of
ameloblastoma still have the potential to develop metastatic lesions. In order to detect metastases as early as possible, the postoperative review should be regular and for life, especially in patients with a history of relapse, as distant metastasis can occur
after a long time. For the diagnosis of metastases, CT-guided
percutaneous transthoracic lung biopsy and lymph node biopsy
are useful methods to conrm metastatic ameloblastoma. The
treatment of ameloblastoma metastasis requires further research
and practice.
Conict of interest
None.
Acknowledgements
This work was supported by grants from the National Natural
Science Foundation of China (No. 81001213) and the Zhejiang
Provincial Science and Technology Plan (No. 2012C33010).
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