Journal of Cranio-Maxillo-Facial Surgery xxx (2013) 1e4
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Case report
Ameloblastoma with varied sites of metastasis: Report of two cases and literature review Yi Lin, Jian-feng He*, Zhi-yong Li, Jian-hua Liu Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital, College of Medicine, Zhejiang University, #79 Qingchun Road, Hangzhou 310003, Zhejiang, PR China
a r t i c l e i n f o
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 confirmed. 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 confirm metastatic ameloblastoma. Ó 2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Ameloblastoma Metastases Histopathology
1. Introduction
2. Case reports
Ameloblastoma is a benign tumour of odontogenic epithelial origin, which accounts for approximately 1% of all mandibular tumours and cysts (Adebayo et al., 2011). It is a locally invasive tumour with the tendency to recur. Although histologically benign and clinically slow growing, ameloblastoma may metastasize to regional lymph nodes and distant sites (Eckardt et al., 2009; Dissanayake et al., 2011). In the WHO classification system of 2005, a clear distinction between ameloblastoma, malignant ameloblastoma, and ameloblastic carcinoma was made. Malignant ameloblastoma differs from ameloblastoma because of the presence of distant metastasis; although both share the same benign histology. Ameloblastic carcinoma has combined histologic features of ameloblastoma, with cytologic atypia and with or without metastasis (Sciubba et al., 2005). Successful treatment for malignant ameloblastoma remains elusive. We report two cases of maxillary ameloblastoma with varied sites of metastasis.
2.1. Case 1
* Corresponding author. Tel.: þ86 571 87236893; fax: þ86 571 87236395. E-mail address: [email protected] (J.-f. He).
A 40-year-old male consulted his stomatologist because of two bony hard lumps in the left maxillary alveolar bone in October 1996. An extended resection of left maxilla was performed, with a histologic diagnosis of ameloblastoma. Subsequently, he experienced multiple frequent recurrences during the 10-year follow-up period (February 1999eMarch 2009). Pathological features of recurrent lesions were consistent with ameloblastoma, with tumour nests comprising stellate cells and peripheral palisading. In August 2009, computer tomography (CT) confirmed multiple spherical lesions in both lungs without calcification. The largest lesion (3.1 cm) was located in the right hilum (Fig. 1). CT-guided percutaneous transthoracic lung biopsy was performed. Microscopic evaluation showed primarily ameloblastoma (Fig. 2). In November 2011, multiple metastatic lymph nodes in the left neck and superior mediastinum with second rib destruction were found on CT (Fig. 3). Although the patient refused lymph node biopsy at that point, the imaging characteristics and clinical presentation were consistent with malignant ameloblastoma with metastases to multiple lymph nodes.
1010-5182/$ e see front matter Ó 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
Please cite this article in press as: Lin Y, et al., Ameloblastoma with varied sites of metastasis: Report of two cases and literature review, Journal of Cranio-Maxillo-Facial Surgery (2013), http://dx.doi.org/10.1016/j.jcms.2013.10.010
2
Y. Lin et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2013) 1e4
Fig. 1. Computed tomography shows metastatic nodules in both lungs.
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) confirmed 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
Fig. 4. Computed tomography shows metastatic nodules in both lungs.
Please cite this article in press as: Lin Y, et al., Ameloblastoma with varied sites of metastasis: Report of two cases and literature review, Journal of Cranio-Maxillo-Facial Surgery (2013), http://dx.doi.org/10.1016/j.jcms.2013.10.010
Y. Lin et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2013) 1e4
Fig. 5. Lung specimen shows metastatic ameloblastoma (H&E, 400).
found the most frequent site of metastasis to be the lungs (72.7%), especially the patients undergoing multiple operations (80%). Because of this high incidence of lung metastasis, the risk of intraoperative implantation via endotracheal tube aspiration Table 1 Patient demographics: 22 cases of malignant ameloblastoma. Author/year reported
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
1
29
Mandible
Lungs
1
19
Maxilla
Iliac
Multiple
15
Mandible
Lungs, kidney
1
3
Mandible
Lungs
Multiple
18
Mandible
Multiple
13
1
Mandible
Cervical lymph nodes Cervical lymph nodes Cervical lymph nodes Lungs
1
Primary diagnosis Primary diagnosis 7
Mandible
Lungs
Multiple
27
Mandible
Lungs, skull
1
4
Mandible
1
23
Mandible
Cervical lymph nodes Lungs
Multiple
5
Maxilla
Lungs
Multiple
6
Mandible
0
Mandible
Cervical lymph nodes Lungs
1
Primary diagnosis 29
Maxilla
Lungs
1
7
Maxilla Maxilla
Multiple Multiple
8 13
Present case 2
Maxilla
Lungs Lungs, cervical lymph nodes Lungs
1
15
3
cannot be ignored. It is impossible to determine whether surgery for multiple recurrences increase the risk for pulmonary metastasis. Metastases to other regions such as the skull, iliac nodes, kidney, and liver are even rarer. The most clinically significant cases are the two with cervical lymph node enlargement prior to surgery, with a pathologic diagnosis of ameloblastoma; these cases affirm the ability of ameloblastoma to spread via the lymphatics. Although the longest reported survival after pulmonary metastasis is 37 years (Hasim et al., 2007), the prognosis for metastatic ameloblastoma is still poor. In 1981, Laughlin reviewed 43 patients with documented cases of metastatic ameloblastoma: the disease-free interval from first diagnosis to the appearance of metastasis was 9 years; the median survival time after metastases was 2 years (Laughlin, 1989). In 1993, Sheppard (Sheppard et al., 1993) reported the time from initial diagnosis to pulmonary metastases to range from 0.3 to 31 years. The mean diseasefree interval for pulmonary metastasis is 14.37 years and for cervical lymph node metastasis is 12.96 years. This indicates that cervical metastasis may precede lung metastasis (Duffey et al., 1995). In our study, the mean disease-free interval from diagnosis of tumour to distant metastasis is 13 years. Thus, development of metastases is relatively slow, and long term follow-up is recommended for patients with ameloblastoma (Adebayo et al., 2011). Since aggressive surgery is the only effective treatment for primary ameloblastoma, this therapy is often applied to metastatic disease (Sheppard et al., 1993). If preoperative tissue diagnosis is desired, transbronchial biopsy can be performed. Neck dissection is recommended for patients with lymph node metastases. Radiotherapy and chemotherapy are reserved for the palliative setting and for inoperable tumours (Laughlin, 1989). Platinum-based regimens are considered to be first line treatment as far as chemotherapy is concerned (Amzerin et al., 2011). Campbell reported a patient who received cyclophosphamide (50 mg t.i.d.), with accompanying decrease in size of the left lung lesion (Campbell et al., 2003). Nonetheless, the effect of these chemotherapy regimens is of limited value in the treatment of malignant ameloblastoma. 4. Conclusion
Mandible Mandible
1
From these findings, 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 confirm metastatic ameloblastoma. The treatment of ameloblastoma metastasis requires further research and practice. Conflict 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).
Please cite this article in press as: Lin Y, et al., Ameloblastoma with varied sites of metastasis: Report of two cases and literature review, Journal of Cranio-Maxillo-Facial Surgery (2013), http://dx.doi.org/10.1016/j.jcms.2013.10.010
4
Y. Lin et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2013) 1e4
References Adebayo ET, Fomete B, Adekeye EO: Delayed soft tissue recurrence after treatment of ameloblastoma in a black African: case report and review of the literature. J Craniomaxillofac Surg 39: 615e618, 2011 Amzerin M, Fadoukhair Z, Belbaraka R, Iraqui M, Boutayeb S, M’rabti H, et al: Metastatic ameloblastoma responding to combination chemotherapy: case report and review of the literature. J Med Case Rep 5: 491, 2011 Berger AJ, Son J, Desai NK: Malignant ameloblastoma: concurrent presentation of primary and distant disease and review of the literature. J Oral Maxillofac Surg 70: 2316e2326, 2012 Campbell D, Jeffrey RR, Wallis F, Hulks G, Kerr KM: Metastatic pulmonary ameloblastoma. An unusual case. Br J Oral Maxillofac Surg 41: 194e196, 2003 Cardoso A, Lazow SK, Solomon MP, Berger JR, Rock A: Metastatic ameloblastoma to the cervical lymph nodes: a case report and review of literature. J Oral Maxillofac Surg 67: 1163e1166, 2009 Ciment LM, Ciment AJ: Malignant ameloblastoma metastatic to the lungs 29 years after primary resection: a case report. Chest 121: 1359e1361, 2002 Devenney-Cakir B, Dunfee B, Subramaniam R, Sundararajan D, Mehra P, Spiegel J, et al: Ameloblastic carcinoma of the mandible with metastasis to the skull and lung: advanced imaging appearance including computed tomography, magnetic resonance imaging and positron emission tomography computed tomography. Dentomaxillofac Radiol 39: 449e453, 2010 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 Endod 111: 734e741, 2011 Duffey DC, Bailet JW, Newman A: Ameloblastoma of the mandible with cervical lymph node metastasis. Am J Otolaryngol 16: 66e73, 1995 Eckardt AM, Kokemüller H, Flemming P, Schultze A: Recurrence ameloblastoma following osseous reconstruction-a review of twenty years. J Craniomaxillofac Surg 37: 36e41, 2009 Eliasson AH, Moser 3rd RJ, Tenholder MF: Diagnosis and treatment of metastatic ameloblastoma. South Med J 82: 1165e1168, 1989 Gilijamse M, Leemans CR, Winters HA, Schulten EA, van der Waal I: Metastasizing ameloblastoma. Int J Oral Maxillofac Surg 36: 462e464, 2007 Golubovi c M, Petrovi c M, Jelovac DB, Nenezi c DU, Antunovi c M: Malignant ameloblastoma metastasis to the neckeradiological and pathohistological dilemma. Vojnosanit Pregl 69: 444e448, 2012
Hasim FW, Poon CC, Smith AC: Prolonged survival with confirmed metastatic pulmonary ameloblastoma. Int J Oral Maxillofac Surg 36: 953e955, 2007 Hayakawa K, Hayashi E, Aoyagi T, Hata M, Kuramoto C, Tonogi M, et al: Metastatic malignant ameloblastoma of the kidneys. Int J Urol 11: 424e426, 2004 Henderson JM, Sonnet JR, Schlesinger C, Ord RA: Pulmonary metastasis of ameloblastoma: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 88: 170e176, 1999 Houston G, Davenport W, Keaton W, Harris S: Malignant (metastatic) ameloblastoma: report of a case. J Oral Maxillofac Surg 51: 1152e1157, 1993 Lai H, Wang J: Benign metastasizing ameloblastoma or malignant ameloblastoma. J Craniofac Surg 22: 995e997, 2011 Laughlin EH: Metastasizing ameloblastoma. Cancer 64: 776e780, 1989 Lin Z, Chen F, Wang T, Hu Q, Sun G: The variability and complexity of ameloblastoma: carcinoma ex ameloblastoma or primary ameloblastic carcinoma. J Craniomaxillofac Surg 41: 190e193, 2013 Luo DY, Feng CJ, Guo JB: Pulmonary metastases from an ameloblastoma: case report and review of the literature. J Craniomaxillofac Surg 40: e470ee474, 2012 an R, Sungur A: Pulmonary metastasectomy in the treatOnerci M, Yilmaz T, Dog ment of recurrent ameloblastoma of the maxilla and mandible: a case report. Eur Arch Otorhinolaryngol 258: 25e27, 2001 Papaioannou M, Manika K, Tsaoussis B, Cheva A, Sichletidis L, Kioumis J: Ameloblastoma of the mandible with pulmonary metastases 45 years after initial diagnosis. Respirology 14: 1208e1211, 2009 Reid-Nicholson M, Teague D, White B, Ramalingam P, Abdelsayed R: Fine needle aspiration findings in malignant ameloblastoma: a case report and differential diagnosis. Diagn Cytopathol 37: 586e591, 2009 Senra GS, Pereira AC, Murilo dos Santos L, Carvalho YR, Brandão AA: Malignant ameloblastoma metastasis to the lung: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 105: e42e46, 2008 Sciubba JJ, Eversole LR, Slootweg PJ: Odontogenic/ameloblastic carcinomas. In: Barnes L, Eveson JW, Reichart P, Sidransky D (eds), World Health Organization classification of tumours, pathology and genetics of head and neck tumours. Lyon: International Agency for Research on Cancer, 287e288, 2005 Sheppard BC, Temeck BK, Taubenberger JK, Pass HI: Pulmonary metastatic disease in ameloblastoma. Chest 104: 1933e1935, 1993 Vorzimer J, Perla D: An instance of adamantinoma of the jaw with metastases to the right lung. Am J Pathol 8: 445e454, 1932 Zarbo RJ, Marunick MT, Johns R: Malignant ameloblastoma, spindle cell variant. Arch Pathol Lab Med 127: 352e355, 2003
Please cite this article in press as: Lin Y, et al., Ameloblastoma with varied sites of metastasis: Report of two cases and literature review, Journal of Cranio-Maxillo-Facial Surgery (2013), http://dx.doi.org/10.1016/j.jcms.2013.10.010
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Case report
Ameloblastoma with varied sites of metastasis: Report of two cases and literature review Yi Lin, Jian-feng He*, Zhi-yong Li, Jian-hua Liu Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital, College of Medicine, Zhejiang University, #79 Qingchun Road, Hangzhou 310003, Zhejiang, PR China
a r t i c l e i n f o
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 confirmed. 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 confirm metastatic ameloblastoma. Ó 2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Ameloblastoma Metastases Histopathology
1. Introduction
2. Case reports
Ameloblastoma is a benign tumour of odontogenic epithelial origin, which accounts for approximately 1% of all mandibular tumours and cysts (Adebayo et al., 2011). It is a locally invasive tumour with the tendency to recur. Although histologically benign and clinically slow growing, ameloblastoma may metastasize to regional lymph nodes and distant sites (Eckardt et al., 2009; Dissanayake et al., 2011). In the WHO classification system of 2005, a clear distinction between ameloblastoma, malignant ameloblastoma, and ameloblastic carcinoma was made. Malignant ameloblastoma differs from ameloblastoma because of the presence of distant metastasis; although both share the same benign histology. Ameloblastic carcinoma has combined histologic features of ameloblastoma, with cytologic atypia and with or without metastasis (Sciubba et al., 2005). Successful treatment for malignant ameloblastoma remains elusive. We report two cases of maxillary ameloblastoma with varied sites of metastasis.
2.1. Case 1
* Corresponding author. Tel.: þ86 571 87236893; fax: þ86 571 87236395. E-mail address: [email protected] (J.-f. He).
A 40-year-old male consulted his stomatologist because of two bony hard lumps in the left maxillary alveolar bone in October 1996. An extended resection of left maxilla was performed, with a histologic diagnosis of ameloblastoma. Subsequently, he experienced multiple frequent recurrences during the 10-year follow-up period (February 1999eMarch 2009). Pathological features of recurrent lesions were consistent with ameloblastoma, with tumour nests comprising stellate cells and peripheral palisading. In August 2009, computer tomography (CT) confirmed multiple spherical lesions in both lungs without calcification. The largest lesion (3.1 cm) was located in the right hilum (Fig. 1). CT-guided percutaneous transthoracic lung biopsy was performed. Microscopic evaluation showed primarily ameloblastoma (Fig. 2). In November 2011, multiple metastatic lymph nodes in the left neck and superior mediastinum with second rib destruction were found on CT (Fig. 3). Although the patient refused lymph node biopsy at that point, the imaging characteristics and clinical presentation were consistent with malignant ameloblastoma with metastases to multiple lymph nodes.
1010-5182/$ e see front matter Ó 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
Please cite this article in press as: Lin Y, et al., Ameloblastoma with varied sites of metastasis: Report of two cases and literature review, Journal of Cranio-Maxillo-Facial Surgery (2013), http://dx.doi.org/10.1016/j.jcms.2013.10.010
2
Y. Lin et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2013) 1e4
Fig. 1. Computed tomography shows metastatic nodules in both lungs.
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) confirmed 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
Fig. 4. Computed tomography shows metastatic nodules in both lungs.
Please cite this article in press as: Lin Y, et al., Ameloblastoma with varied sites of metastasis: Report of two cases and literature review, Journal of Cranio-Maxillo-Facial Surgery (2013), http://dx.doi.org/10.1016/j.jcms.2013.10.010
Y. Lin et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2013) 1e4
Fig. 5. Lung specimen shows metastatic ameloblastoma (H&E, 400).
found the most frequent site of metastasis to be the lungs (72.7%), especially the patients undergoing multiple operations (80%). Because of this high incidence of lung metastasis, the risk of intraoperative implantation via endotracheal tube aspiration Table 1 Patient demographics: 22 cases of malignant ameloblastoma. Author/year reported
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
1
29
Mandible
Lungs
1
19
Maxilla
Iliac
Multiple
15
Mandible
Lungs, kidney
1
3
Mandible
Lungs
Multiple
18
Mandible
Multiple
13
1
Mandible
Cervical lymph nodes Cervical lymph nodes Cervical lymph nodes Lungs
1
Primary diagnosis Primary diagnosis 7
Mandible
Lungs
Multiple
27
Mandible
Lungs, skull
1
4
Mandible
1
23
Mandible
Cervical lymph nodes Lungs
Multiple
5
Maxilla
Lungs
Multiple
6
Mandible
0
Mandible
Cervical lymph nodes Lungs
1
Primary diagnosis 29
Maxilla
Lungs
1
7
Maxilla Maxilla
Multiple Multiple
8 13
Present case 2
Maxilla
Lungs Lungs, cervical lymph nodes Lungs
1
15
3
cannot be ignored. It is impossible to determine whether surgery for multiple recurrences increase the risk for pulmonary metastasis. Metastases to other regions such as the skull, iliac nodes, kidney, and liver are even rarer. The most clinically significant cases are the two with cervical lymph node enlargement prior to surgery, with a pathologic diagnosis of ameloblastoma; these cases affirm the ability of ameloblastoma to spread via the lymphatics. Although the longest reported survival after pulmonary metastasis is 37 years (Hasim et al., 2007), the prognosis for metastatic ameloblastoma is still poor. In 1981, Laughlin reviewed 43 patients with documented cases of metastatic ameloblastoma: the disease-free interval from first diagnosis to the appearance of metastasis was 9 years; the median survival time after metastases was 2 years (Laughlin, 1989). In 1993, Sheppard (Sheppard et al., 1993) reported the time from initial diagnosis to pulmonary metastases to range from 0.3 to 31 years. The mean diseasefree interval for pulmonary metastasis is 14.37 years and for cervical lymph node metastasis is 12.96 years. This indicates that cervical metastasis may precede lung metastasis (Duffey et al., 1995). In our study, the mean disease-free interval from diagnosis of tumour to distant metastasis is 13 years. Thus, development of metastases is relatively slow, and long term follow-up is recommended for patients with ameloblastoma (Adebayo et al., 2011). Since aggressive surgery is the only effective treatment for primary ameloblastoma, this therapy is often applied to metastatic disease (Sheppard et al., 1993). If preoperative tissue diagnosis is desired, transbronchial biopsy can be performed. Neck dissection is recommended for patients with lymph node metastases. Radiotherapy and chemotherapy are reserved for the palliative setting and for inoperable tumours (Laughlin, 1989). Platinum-based regimens are considered to be first line treatment as far as chemotherapy is concerned (Amzerin et al., 2011). Campbell reported a patient who received cyclophosphamide (50 mg t.i.d.), with accompanying decrease in size of the left lung lesion (Campbell et al., 2003). Nonetheless, the effect of these chemotherapy regimens is of limited value in the treatment of malignant ameloblastoma. 4. Conclusion
Mandible Mandible
1
From these findings, 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 confirm metastatic ameloblastoma. The treatment of ameloblastoma metastasis requires further research and practice. Conflict 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).
Please cite this article in press as: Lin Y, et al., Ameloblastoma with varied sites of metastasis: Report of two cases and literature review, Journal of Cranio-Maxillo-Facial Surgery (2013), http://dx.doi.org/10.1016/j.jcms.2013.10.010
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Y. Lin et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2013) 1e4
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Please cite this article in press as: Lin Y, et al., Ameloblastoma with varied sites of metastasis: Report of two cases and literature review, Journal of Cranio-Maxillo-Facial Surgery (2013), http://dx.doi.org/10.1016/j.jcms.2013.10.010
