Metastatic pulmonary ameloblastoma: An unusual case

doi:10.1016/S0266-4356(03)00046-9

British Journal of Oral and Maxillofacial Surgery

Volume 41, Issue 3, June 2003, Pages 194-196

https://doi.org/10.1016/S0266-4356(03)00046-9 Get rights and content

Abstract

We present a patient who had a large metastatic pulmonary ameloblastoma resected 25 years after removal of an apparently benign primary ameloblastoma of the jaw. It highlights three areas: problems with the histopathological diagnosis of pulmonary metastases using fine needle aspiration; a noted radiological improvement after a 5-year course of oral cyclophosphamide, in keeping with occasional patients who have responded to chemotherapy; and the technical difficulties of resection of a large pulmonary metastasis, particularly when it is adherent to the mediastinum.

Section snippets

INTRODUCTION

Ameloblastomas are rare odontogenic neoplasms, which account for about 1% of all oral tumours. They arise from the epithelium of the dental lamina, particularly in the mandible (80%), but also the maxilla (20%).¹ They are usually benign, but are locally aggressive with a high incidence of local recurrence. Malignant variants are usually reported as either ameloblastoma with metastases or widely infiltrating lesions with histological features of carcinoma. Metastases are rare, but can be found

CASE REPORT

A 46-year-old white man, who was a lifelong cigarette smoker, presented to the respiratory physicians at his local district general hospital with evidence of clubbing, purulent sputum and an abnormal sensation over his right chest. The chest radiograph showed a 1.5-cm opacity at the base the of the left lung. Computed tomogram (CT) scan showed mediastinal lymphadenopathy with an additional 7 cm mass in the right lower lobe that was indenting the left atrium, which had not been visible on the

HISTOPATHOLOGY

The original lesion in the jaw showed typical features of well-differentiated follicular ameloblastoma. The lesion from the left lung and three separate nodules in the right lower lobe all showed a similar histological picture except that the pulmonary disease was less well-differentiated with more basaloid areas. The tumour, although lacking the high grade malignant cytological features of a carcinoma, was clearly invasive, and had destroyed airway cartilage and adjacent lymph nodes. In some

DISCUSSION

Ameloblastomas are rare and only about 2% metastasise.³ They are usually treated adequately by local excision. Local recurrence is common (50–72%).¹ The mean age at presentation with ameloblastoma is 33 years, but malignant and metastasising ameloblastomas usually present about 10 years later.

Of the several varieties of ameloblastoma described, those most likely to metastasise are the plexiform and follicular types,³ otherwise the histopathological appearances have little ability to predict the

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Atypical ameloblastoma; report of 3 cases and a review of the literature
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L. Dorner et al.
A case of ameloblastoma carcinoma with pulmonary metastases
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Treatment efficacy and prognosis of pulmonary metastasizing ameloblastoma: a systematic review
2022, International Journal of Oral and Maxillofacial Surgery
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Three articles did not describe the patients’ history timeline1,35,37. Adverse events such as post-chemotherapy adverse reactions4,26, pulmonary recurrences16,29,34, and death10,11,20,36,38 were reported in several studies (Supplementary Material Table S1). Most cases were reported from the continent of Asia (11 cases)1,15,20,25,26,33,35,37,38, followed by Europe4,10,12,23,29,31,32 and North America3,11,16,17,34 (seven cases each).
The aim of this review was to integrate the current literature into a comprehensive evaluation of pulmonary metastasizing ameloblastoma (MA). Related articles, published since January 2000, were reviewed. The PubMed, Embase, Cochrane Database of Systematic Reviews, and Web of Science databases were searched based on the PRISMA guidelines. Twenty-four studies, including 28 case reports, met the eligibility criteria. The mean ± standard deviation disease-free interval after primary treatment was 12.1 ± 9.3 years. Adjuvant therapy (surgery and chemoradiotherapy) is considered appropriate for pulmonary MA patients presenting with rapid progression or apparent pulmonary symptoms. The proportion of patients undergoing simple follow-up (observation) has increased rapidly in the past 10 years. Meanwhile, among the three treatment groups (observation, surgery, and adjuvant therapy), no significant difference was found in the efficacy (P = 0.081) or progression-free survival (P = 0.263). The 5-year and 10-year overall survival rates were 72.4% and 52.8%, respectively. This study provides insights that could help surgeons with the management of patients presenting with pulmonary MA.
The use of radiation therapy in the definitive management of ameloblastic carcinoma: a case report
2019, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
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There are reports on the usefulness of adriamycin, cyclophosphamide, and doxorubicin to treat metastasizing ameloblastoma. However, unfortunately, it is not clear whether it is useful for AC or whether it contributes to survival and quality of life.22-25 In recent years, biotherapy using molecular targeting drugs have been developed.
Although rare, ameloblastic carcinoma (AC) is one of the most common malignant tumors occurring in the odontogenic epithelium. Evidenced-based medical guidelines for AC are not established because AC is a rare type of cancer. However, surgery for AC is the primary initial treatment modality. A few reports suggest that radiotherapy (RTx) is effective for AC; however, the tumor is generally considered to be radioresistant. Furthermore, if RTx is performed for AC, osteonecrosis may occur because most of the ACs arise from the jaw. Here, we report a case of an older patient with maxillary AC who had underlying medical problems and who received conventional RTx with good outcomes. Ten years after the treatment, no major adverse event accompanying irradiation, local recurrence, or metastasis was observed. We discuss the therapeutic effect and usefulness of RTx on AC. As indicated by our findings, RTx for AC may represent a definitive treatment strategy for patients with poor general conditions or those with tumors that cannot be resected completely.
Multiple nodular lung metastases with no obvious primary showing low positive 18-fluoro deoxyglucose (FDG) uptake on positron emission tomography (PET) scan: A unique case of metastatic ameloblastoma
2016, Cancer Treatment Communications
The incidental detection of multiple secondaries in the lung, in an asymptomatic individual calls for detailed evaluation, especially if the 18-fluoro-deoxyglucose positron emission tomography (FDG PET) scan results are inconclusive or unusual.
A 45 year old lady, asymptomatic, presented to us for evaluation of incidentally diagnosed, bilateral pulmonary metastases with a provisional diagnoses of advanced lung cancer. FDG PET scanning was inconclusive and revealed low positive FDG uptake. The biopsy revealed it to be metastatic ameloblastoma. She refused surgery but took only one cycle of chemotherapy with paclitaxel and carboplatin, which she then subsequently discontinued but continues to be asymptomatic at last followup.
She gave history of previous ameloblastoma of the left mandible 13 years back, and recurrences 3 and 6 years later, which had been resected. For the last seven years she had no evidence of disease. History of past illness including operative interventions done for apparently benign or locally aggressive lesions of the oral cavity such as an ameloblastoma is important in evaluation of such cases.
Pulmonary metastases arising from an ameloblastoma maybe associated with low FDG positive uptake, and even though they are rare, it is important to diagnose them, since the prognosis and management of lesions from lung or other primaries and those from ameloblastoma are markedly different.
Metastasizing Ameloblastoma - A perennial pathological enigma? Report of a case and review of literature
2014, Journal of Cranio-Maxillofacial Surgery
The Ameloblastoma is a slow growing locally invasive odontogenic epithelial neoplasm with a high recurrence rate and a low tendency to metastasize. Metastasis in Ameloblastoma was first described by Simmons and Emura in the 1920s. Slootweg and Muller proposed the term Malignant Ameloblastoma to describe a well-differentiated ameloblastoma that metastasizes but maintains the characteristic cytologic features of the original tumour and the term Ameloblastic Carcinoma to an ameloblastoma with malignant cytological features. About 2% of ameloblastomas undergo metastasis. So far there have only been two cases of Metastasizing Ameloblastoma reported from the Indian Subcontinent. We present the case of a 22-year-old male Indian patient, who presented with a diffuse swelling in the left posterior mandible. Radiographs revealed a multilocular radiolucency in the left mandible. On histopathological examination, the lesion was diagnosed as follicular ameloblastoma. Four years later the patient presented with a swelling in the left submandibular region. Histological examination revealed metastatic ameloblastoma within the cervical lymph node.
Ameloblastoma with varied sites of metastasis: Report of two cases and literature review
2014, Journal of Cranio-Maxillofacial Surgery
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.
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.
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.
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.
The variability and complexity of ameloblastoma: Carcinoma ex ameloblastoma or primary ameloblastic carcinoma
2013, Journal of Cranio-Maxillofacial Surgery
Citation Excerpt :
The ameloblastoma is the most common odontogenic tumor of the jaw. Although it is considered to be benign, it may show various biological behaviors ranging from cystic expansion to a more aggressive solid mass and even malignant transformation (Campbell et al., 2003; Hayakawa et al., 2004; Mendenhall et al., 2007). In the presented paper, a primary maxillary ameloblastoma showed an aggressive clinical course.
Ameloblastoma is characterized by slow-growing, local invasiveness and high incidence of local recurrence. It usually presents with a benign histological appearance. However, ameloblastoma occasionally demonstrates a clinical course that is characteristic of malignant transformation. Here, we present a case of ameloblastoma with an aggressive clinical course, including multiple recurrences, a short disease-free interval, pulmonary metastasis and extensive skull-base infiltration. With a careful re-evaluation of the histology and cytology of the specimens of primary and recurrent ameloblastoma in 2006 and 2007, malignant transformation was observed and carcinoma ex ameloblastoma was ultimately diagnosed.

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SHORT COMMUNICATIONMetastatic pulmonary ameloblastoma: An unusual case