British Journal of Oral Surgery (1981), 19, 38-42 @ The British Association
of Oral Surgeons
AMELOBLASTOMA WITH PULMONARY METASTASES. A CASE REPORT G.
Department of Dental and Oral Surgery, Mount Vernon Hospital, Northwood, Middlesex Summary. A case of ameloblastoma with metastases to the lungs is presented. The slow growing nature of the lesion traced over a period of 23 years is emphasised. The mechanisms of mode of spread are inconclusive although it is of interest to contemplate the possible role that packing may have played. The findings of Tsukada et al. (1965) are supported to a limited extent, the exceptions being the primary tumour was not extensive and few operations were performed before metastasis occurred.
Introduction The purpose of this paper is to report a case of ameloblastoma of the mandible with pulmonary metastases. Small and Waldron (1955) reviewed 1,036 cases of ameloblastoma which included 379 cases previously reported by Robinson (1937). They identified 33 possible cases of malignant ameloblastoma but most of the reports were poorly documented and lacked histopathological proof. They accepted three cases as showing evidence of metastasis, of which two cases were to the lungs (Waldron, 1966). Carr and Halprin (1968) further extended this review finding and described five additional cases. Spread of ameloblastoma occurs by way of the lymphatics, (Simmons, 1928), aspiration, (Vorzimer & Perla, 1932) or blood stream (Schweitzer & Barnfield, 1943). The commonest site for secondary deposits is in the lung. Many cases have been reported in which radio-opaque abnormalities have been apparent on radiographs of the lungs and these have been assumed to be secondary lesions of ameloblastoma. Histopathological examinations of the lung lesions, however, have not been carried out and so the identity of these lesions is speculative. Case report In 1956 a 31-year-old female had a swelling removed from the right side of the mandible in the premolar area which was reported as ameloblastoma. She remained symptomless until 1960 when in June she noted a further swelling in the same area. A cystic lesion was removed and the wound packed open. The histopathological report was as follows: Recurrent adamantimoma
The specimen is an irregular 5 x 1.5 x 1.75 cm tissue mainly formed of firm white tumour. (Received 3 March 1980; accepted 21 March 1980) * Present address: Suffolk.
of Oral Surgery, Ipswich Hospital, 38
Heath Road Wing, Ipswich,
This is an adamantinoma. The tumour clumps have an external layer of tall columnar cells and sometimes the central tissue is formed of stellate reticulum with some cystic spaces. The majority of the central masses however are formed of squamous epithelial cells with spinous processes. Although mitotic activity is very low, evidence of local bone infiltration is seen. (Fig. 1).
FIG. 1. Photomicrograph
the histopathological section of the ameloblastoma from the mandible (x 30).
In April 1962 the Grst molar region was re-explored after a radiograph showed a radiolucent area in this site. The histopathological reportconfirmed recurrent ameloblastoma. A year later the radiolucency had’extended to the lower border of the mandible (Fig. 2). The body of the mandible was resected subperiosteally from q to the retromolar region, where the tissues firmly adhered to the underlying bone: the dissection was taken supraperiosteally. Four weeks later the defect was reconstructed with a bone graft taken from the right iliac crest. The histopathological report contirmed that the lesion was an ameloblastoma. The patient remained well until May 1972 when a routine chest X-ray demonstrated radiopacities in the lungs (Fig. 3). Right and left thoracotomies were performed to remove the right middle lobe and a mass from the left lung base. The histopathological examination showed ameloblastoma similar to the mandibular primary tumour (Fig. 4). No further oral lesions were seen until June 1977, when a soft tissue swelling appeared in the right retromolar area and recurrent ameloblastoma was treated. In June 1978 a painless firm enlarging swelling was noticed lingually in the right molar region. The lesion, deep to the mucosa and superficial to the bone graft, was removed by way of intra-oral approach and histopathological examination confirmed recurrent ameloblastoma. Currently the patient is free from overt intraoral lesions.
FIG. 2. Lateral oblique radiograph of the right side of the mandible showing lobulated radiolucent area extending to the lower border.
FIG. 3. Chest radiograph
showing a rounded opacity in the field of the left lung base.
FIG. 4. Photomicrograph
of the histopathological section of the secondary deposit of ameloblastoma removed from the lung ( x 45).
Discussion Metastatic spread of tumours to the lungs is thought to occur either by way of bloodstream or by aspiration. When aspiration is considered it would be expected that the secondary deposits would be found in the right lung. In this case the lesions were found in the right middle lobe and left lung base, thus it is more probable that spread occurred via the bloodstream. However, spread by aspiration cannot be excluded especially when it is considered that at the second operation the wound was packed open and the dressing changed numerous times. It is therefore possible that it was during this procedure that seeding occurred not only to local tissue but also to the lungs. The factors influencing metastatic spread of ameloblastoma are uncertain, Tsukada et al. (1965) in their review of cases reported with pulmonary metastases point to some common findings, (1) (2) (3) (4)
The tumour is of long duration with metastases appearing late. There is extensive local spread at the time of metastasis. Patients have had more than one operation or radiation treatment. Metastases are limited to lymph nodes, lung and pleura.
In agreement with the above findings this case has the features of long duration with late metastases, the secondary deposits were found in the lung and the patient had had multiple operative procedures. Acknowledgements I would like to thank Mr B. W. Fickling for permission to report this case, Mr K. G. Boobyer for providing some of the information and Mr G. L. Fordyce for his help and encouragement in the
preparation of this paper. I am also grateful to Dr M. Bennet for his help in preparing the photomicrographs and reporting on the pathological material and to Prof. R. B. Lucas for confirming the nature of all the histopathological material. References Carr, R. F. & Halperin, V. (1968). Malignant ameloblastomas from 1953-1966. Review of the literature and a report of a case. Oral Surgery, Oral Medicine and Oral Pathology, 26, 514. Robinson, H. B. G. (1937). Ameloblastoma: a survey of 379 cases from the literature. Archives of Pathology, 23, 831. Schweitzer, F. G. & Barnfield, W. F. (1943). Ameloblastoma of the mandible with metastasis to the lungs-a report of a case. Journal of Oral Surgery, 1,287. Small, I. A. & Doldron, C. A. (1955). Ameloblastomas of the jaws. Oral Surgery, Oral Medicine and Oral Pathology, 8, 28 1. Simmons, C. C. (1928). Adamantinoma. Annals of Surgery, 88, 693. Tsukada, Y., de la Pava, S. & Pickren, J. W. (1965). Granular-cell ameloblastoma with metastases to the lungs. Report of a case and review of the literature. Cancer, 18, 916. Vorzimer, J. & Perla, D. (1932). An instance of adamantinoma of the jaw with metastases to the right lung. American Journal of Pathology, 8,445. Waldron, C. A. (1966). Ameloblastoma in perspective. Journal of Oral Surgery, 24, 331.