(1) Médico Adjunto del Servicio de Cirugía Maxilofacial
(2) Médico Responsable del Servicio de Cirugía Maxilofacial. Hospital Neurotraumatológico. Complejo Hospitalario de Jaén

Address:
Juan Sánchez Jiménez
c/ Extremadura nº 2 portal 4, 8º H.
23008 Jaén.
Tlf: 953 256260 / 600916106
Fax: 953 008113
E-mail: juansanchezjimenez@andaluciajunta.es

Received: 22-11-2003 Accepted: 25-04-2004

INTRODUCTION

Metastatic tumours in the oral cavity are uncommon. They represent approximately 1% of all malignant tumours affecting the oral cavity (1). However, there are some authors who state that the possibility of finding metastatic tumours is higher. Metastatic tumours are the most common malignant neoplasms in the skeleton in general, and also, in the jaws (2,3).

Metastatic tumours in the oral cavity can be located in the soft perioral tissues, approximately 15% (4). Maxillary bones are the most common locations for metastasis (85%), the mandible being more often affected (80-90%) than the maxilla (10-20%). Within these the molar and premolar regions are the areas more commonly affected by metastases. Location in soft tissue is mainly adherent gums (1). Next more frequent is metastasis in the tongue, extremely uncommon, 1 out of 1000 metastatic tumours. They usually occur as gingival hyperplastic or reactive lesions with the clinical appearance of pyogenic granuloma or epulis, lesions with which they can be confused, even with the possibility of being multiple and bilateral lesions (5,6).

70% of oral metastases are manifested after the primary tumour becomes evident, while the remainder 30% are the first clinical sign of primitive tumour spreading (7). In most cases of metastasis to the oral cavity, the primary tumour is located below the level of the clavicles. Tumours which most frequently metastasize to oral cavity are primitive of breast, lung, kidney; as they cause 50% of all oral metastases (1,8). The remainder 50% is located in the prostate, thyroid, gut (mainly in Japan), suprarenales glandulae, uterus, bones, etc (1,2).

The mean age span of occurrence is the fifth and sixth decades, though it may occur at any age (1). There are no significant differences regarding sex, but it is clear that the primary tumour varies depending on the sex (9).

As for the clinical, oral metastases may present themselves in an asymptomatic manner or manifest as: tumour, pain, ulcer, paresthesia, haemorrhage, periodontitis, trismus, pathological fracture; establishing symptomatology in a short period of time. Under radiographic examination, it can be appreciated most frequently a radiolucent osteolytic lesion with ill-defined margins, although metastases from the prostate, breast, and lung may give sclerotic appearance (4), with the possibility of osteoblastic images in 5% of cases there will be no evident radiological changes (7).

The occurrence of a metastatic lesion to the oral cavity is, generally, a sign of bad prognosis, evidence of spread neoplasm disease indicating fatal evolution within a few months interval following diagnosis of oral metastasis. Lower than 10% of patients survive 4 years after diagnosis (2,7).

Treatment in oral metastases is usually palliative. Surgery is usually undertaken when the primary is under control and there is no evidence of other metastases. In such cases where the oral metastasis is the only one present, appropriate surgical treatment may improve lightly prognosis. When patients with soft tissue metastasis complain of pain, haemorrhage, overinfection or masticatory interference, palliative extirpation of lesion can be undertaken (10).

A CASE REPORT

Sixty-three-year-old male patient who comes for consultation presenting ulcer on adherent gums in the upper-left molar region with one month evolution and not improving after conservative treatment. Under oral examination the patient presented ulcer in the upper-left molar region, with necrotic fundus and swollen, erythematous edges 2 cm bigger diameter, along with swollen upper-left vestibular fundus and on the hard palate (left side). As previous medical history patient non-smoker or drinker admitted being operated on esophageal adenocarcinoma of lower third four months before. It was esophageal adenocarcinoma enteroid-type, poorly differentiated, with filtration into adipose periesophageal tissue, and little fibroblastic and lymphoid peritumoral reaction, as well as lymph node metastase, presenting extracapsular extension, with intense atypia, high mitotic rate and moderate necrosis. Patient did not have complemetary postoperative quimiotherapy or radiotherapy treatment as in postoperative presented gastroesophageal suture dehiscence and gastrocutaneous fistula which finished after parentheral nutrition.

Simple radiological study was undertaken by means of orthopantomography and Waters, showing osteolysis picture on upper-left maxillary bone and picture of occupation into left maxillary sinus. Biopsy was taken of intraoral lesion, reporting anatomo-pathologist of esophageal adenocarcinoma metastasis. Patient was admitted to hospital in order to dismiss further metastases and define the extension of metastasis to maxillary bone.

Cervical examination in order to value the state of cervical lymph nodes proved negative. Orofacial CT was undertaken, showing destruction of upper-left maxilla with hard palate involvement, pterygoid apophysis (figure 1), left maxillary sinus, with back and internal wall fracture of left maxillary sinus (figure 2); left masticatory area also being involved and tumoral lesion being quite close to base of left orbit. Ecographic abdominal study and conventional radiolography thorax study were taken reporting as normal. Studies were completed with osseous gammagraphy, showing osseous lesions suggesting malignancy in upper-left maxillary bone, right pelvis, seventh dorsal vertebra, and right costal parry (figure 3). When the study was finally completed, (two weeks time between first consultation and intraoral biopsy) intraoral lesion had grown considerably, presenting necrotic areas, and involving the whole upper-left vestibular fundus, gums, left hard palate: presenting functional inability in left leg which prevented correct mobility. Patient died suddenly after massive haemorrhage in oral cavity.

DISCUSSION AND CONCLUSIONS

Primary esophageal adenocarcinoma is a relatively uncommon tumour, approximately 1 to 50% of all malignant esophageal tumours, depending on the geographical location (quite common in China-Japan). In the USA, it used to be unfrequent the past decades, with 10% incidence of esophageal cancer. However, progressive increase has been reported of Barret´s esophagus incidence associated with esophageal adenocarcinoma, 9% annual increase in men and 5% women. This tumour comprises, at the present time, 34% of malignant esophageal tumours and 80% of lower third esophagus (11). Reasons for this increase in frequency are unknown, though advances in diagnostic methods (echography, endoscopic echography, computed tomography,magnetic resonance imaging…) may be partly responsible. Reflux, alcohol consumption, smoking have been suggested as etiological factors for esophageal adenocarcinoma (12).

Esophageal adenocarcinoma tends to grow locally, invading surrounding tissue causing local complications which may lead to death. Approximately 80% present invasion of esophageal muscles into periadventicial tissues and then to surrounding tissues. Most common invasion is into trachebronchial tree, presenting a tracheoesophageal or bronchoesophageal fistula. Mediastinum and lung are the secondly involved sites, presenting pneumonia, mediastinitis,and abscess formation. Other organs posibly involved include: aorta, pericardium, liver, recurrent nerves , spinal cord, etc. Lymphatic spreading is present in 70% of patients, especially with the occurrence of ganglionic metastases in mediastinum, abdominal and cervical. Ganglionic involvement of left supraclavicular fossa is traditional (Virchow´s ganglion). Lymphatic extension, along with submucosa invasion, is also responsible for intraesophageal tumour extension. Hematic dissemination occurs at a later stage and is less frequent, liver and lung being the most common locations (13).

There exist three criteria for considering a malignant neoplasm to be metastatic (9):

1. There must be a histological verified primary.

2. The secondary lesion must be histologically similar to the primary.

3. The possibility of direct extension from the primary must be excluded.

Clausen and Poulsen have proposed a set of criteria for definition of metastasis to maxillary bones (14):

1. Lesion must be located on maxillary bone, excluding the possibility of primary tumour both on maxillary bone and oral mucosa.

2. Lesion must be confirmed microscopically.

3. Primary tumour must be identified.

Additionally, for diagnosis of metastatic gingival tumour four criteria are required (15):

1. Primary tumour must be known.

2. No evidence of direct tumoral extension between primary and secondary tumour.

3. No involvement of underlying bone.

4. Histopathological pattern similar to primary tumour.

The incidence of maxillary bone metastases is difficult to assess accurately.

Probably, maxillary bone metastases are more frequent than it has been reported in medical literature.

The use of gammagraphy, magnetic resonance imaging (MRI) and computed tomography (CT), may be very useful methods in evaluating the extent of the metastatic process to the jawbones. Osseous gammagraphy is a very useful method of examination to determine the existence of metastasis, as the marker accumulates in areas of greater metabolic activity. The frequency of micrometastases in histologic sections has been found to be higher than that observed by radiological methods (16). In the mandible, microscopical deposits of metastatic tumour cells have been found in 16% of autopsied carcinoma cases (3).

The route of metastases in oral cavity is mainly via the lymphatics, blood, via the sputum or following surgical intervention. As it is generally believed that the jaws do not contain lymphatic vessels, the only practical route for metastasis remains the blood vessels but the lymphatic vessels may be responsible for metastases in the rest of perioral soft tisúes (10,17).

In the skeleton, bones with red marrow are the preferential sites for metastatic deposits. Hematopoietically active marrow bones represent an attractive site for metastatic development. The bones with greater medullar activity are: the trunk, ribs, skull, femur and humerus. The marrow contains growth factors which may enhance colonization of metastatic cells (18). Jawbones, especially in old age, are poor in active marrow, which is usually found in the posterior part of the mandible and it is believed that these hematopoietic areas in the mandible and slowingdown of the circulation in this area appear to favour the entrapment of the metastatic cells (10).

Pathogenically, at gingival level the swelling of adherent gums must be considered in the attraction of metastatic cells, which might be entrapped by rich vascular neoformation of chronically swollen gums which contain neocapillaries with a fragmented basal membrane more easily penetrated by tumour cells than mature vessels (19).

In a patient diagnosed with neoplasm disease, presenting lesion in oral cavity, a metastatic lesion should be suspected. However, in 30% of cases oral metastasis is the first indication of distant malignant tumour (1,15), which should be strongly considered to establish diagnosis, especially in those cases where metastases may present a histopathological appearance similar to other primary intraoral lesions, for example, primary intraoral epidermoid carcinoma versus epidermoid lung carcinoma, etc (7).

BIBLIOGRAFÍA        [ Links ]

2. Nihismura Y, Yakata T. Metastatic tumours of the mouth and jaw. J Maxillofac Surg 1982;10:253-7.         [ Links ]

3. Hashimoto N, Kurihama K. Pathological characteristics of metastatic carcinoma in the human mandible. J Oral Pathol 1987;16:362-7.         [ Links ]

4. Al-Anis S. Metastatic tumors to the mouth: report of two cases. J Oral Surg 1973;31:120-5.         [ Links ]

5. Grace A, Drake-Lee A, Conn P. Metastatic lingual carcinoma masquerading as Ludwig´s angina. J Laryngol Otol 1984;98:535-7.         [ Links ]

6. Rass T, Prein J. Differential diagnosis of epulis-type changes in the mouth. Swiss Dent 1992;13:21-7.         [ Links ]

7. Hirhberg A, Leibovich P, Buchner A. Metastatic tumors to the jaws: analysis of 390 cases. J Oral Pathol Med 1994;23:337-41.         [ Links ]

8. Rodado C, González J, Huguet P, Ávila M, Raspall G. Metástasis Orales como forma de presentación de un adenocarcinoma de pulmón. Med Oral 1997;2:315-20.         [ Links ]

9. Zachariades N, Papanicolaoy S. Breast cancer metastatic to the mandible. J Oral Maxillofac Surg 1982;40:813-6.         [ Links ]

10. Van der Kwast. Jaw metastases. Oral Surg 1974;37:850-4.         [ Links ]

11. Devesa SS, Blot WJ, Fraumeni JF. Changing patterns in th incidence of esophageal and gastric carcinoma in the United States. Cancer 1998;83:2049-53.         [ Links ]

12. Smith RR, Hamilton SR, Boitnott JK, Rogers EL. The spectrum of carcinoma arising in Barrett´s esophagus. A clinicopathologic study of 26 patients. Am J Surg Pathol 1984;8:563-73.         [ Links ]

13. Feith M, Stein HJ, Siewert JR. Pattern of lymphatic spread of Barrett´s cancer. World J Surg 2003;27:1052-7.         [ Links ]

14. Clausen F, Poulsen H. Metastasis from carcinoma to the jaws. Acta Pathol Microbiol Scand 1963;57:361-5.         [ Links ]

15. Peris K, Cerroni L, Paoloni M. Gingival metastases as first sign of an undiffenciated carcinoma of the lung. J Dermatol Surg Oncol 1994;20:407-9.         [ Links ]

16. Lindemann F, Schlimok G, Dirscheld P. Prognostic significance of micrometastatic tumour cells in bone marrow of colorectal cancer patients. Lancet 1992;340:685-9.         [ Links ]

17. Wolujewicz MA. Condylar metastasis from a carcinoma of the prostate gland. Brit J Oral Surg 1980;18:175-8.         [ Links ]

18. Hirshberg A, Leibovich P, Buchner A. Metastatic tumors to the jawbones: analisis of 390 cases. J Oral Pathol Med 1994;23:337-41.         [ Links ]

19. Gerlach KL, Horch H, Lacroix WF. Condylar metastasis from bronchial carcinoma. J Maxillofac Surg 1982;10:250-4.        [ Links ]