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Case reports

abstract

Received for publication: Apr. 11, 2006. Revised: May 26, 2006.

rEZUMat

Department of Oral and Maxillo-Facial Surgery, Faculty of Dental Medicine, Victor Babes University of Medicine and Pharmacy, Timisoara

Correspondence to:Serban Talpos, Department of Oral and Maxillo-Facial Surgery, 5 Take Ionescu Str., Timisoara, Tel. +40722434390Email: talpos@yahoo.com

IntrodUctIon

Oral metastatic lesions from distant tumours are uncommon, accounting for only 1% of all oral malignancies. They mainly involve the bony structures (particularly the mandible), whereas primary metastases to soft tissues are extraordinarily rare (only 0.1% of oral

UncoMMon EPULIs-LIKE GInGIVaL tUMoUr MEtastasIs

serban talpos, emilia Ianes, emanoil popa

Metastasis to the gingiva from lung cancer is extremely rare and, usually, has a rapid evolution with consecutive clinical symptoms which can seriously affect the quality of life. The present case report describes a 60-year-old man, who presented in our clinic with a large painless gingival mass, epulislike tumour, located on the anterior maxillary alveolar crest. One year before, the patient was referred to neurosurgery, when it was discovered a brain tumour. The investigations continued with chest X-ray, which showed the existence of a small tumour in the left lung. After brain tumour removal, the histological findings had proved to be a brain metastasis of the lung cancer. In our department, after one year from the first operation, chest X-ray and skull CT scan were repeated and revealed the primary massive tumour in the left lung and also a recurrence of the cerebral tumour. After gingival tumour removal, unkeratinized G3 scuamous cell carcinoma was histologically diagnosed, which was the same as that of the brain metastasis and led us to the conclusion that the gingival tumour represented the second metastasis, after the brain localization, of the primary lung tumour. Gingival metastasis is usually considered a late complication and is most often associated with multiple metastases in other organs. Chemotherapy and radiotherapy were insufficient but they had the role of improving the quality of patient's life for the lasts months, avoiding the functional problems of chewing, swallowing and speaking as well as local tumour bleeding.Key Words: gingival metastasis, epulis, lung cancer, surgical removal

Metastazele gingivale date de cancerul pulmonar sunt extrem de rare, iar atunci cnd apar, prezint\ o evolu]ie rapid\, simptomatologia consecutiv\ afectnd serios calitatea vie]ii pacientului. Cazul descris n articol este cel al unui pacient n vrst\ de 60 de ani, care s-a prezentat n Clinica noastr\ cu o forma]iune tumoral\ gingival\ masiv\, asem\n\toare unui epulis, nedureroas\, localizat\ la nivelul crestei alveolare maxilare anterioare. Cu un an n urm\, pacientul a fost internat n Clinica de Neurochirurgie, unde s-a descoperit o tumor\ cerebral\. Investiga]iile au continuat, efectundu-se o radiografie pulmonar\ ce a indicat o mic\ forma]iune tumoral\ la nivelul pl\mnului stng. Dup\ extirparea tumorii cerebrale, rezultatul examenului histopatologic a fost de metastaz\ cerebral\ dat\ de un cancer pulmonar. n Clinica noastr\, dup\ un an de la prima interven]ie chirugical\, au fost repetate radiografia pulmonar\ [i examenul CT cerebral, care au relevant prezen]a unei tumori masive pulmonare stngi [i recidiva tumorii cerebrale. Dup\ extirparea tumorii gingivale, aceasta a fost diagnosticat\ histologic carcinom scuamo-celular nekeratinizat G3, la fel ca [i n cazul metastazei cerebrale, ducnd la concluzia c\ tumora gingival\ reprezint\ o metastaz\ secundar\, dup\ cea cerebral\, a cancerului pulmonar. Metastaza gingival\ este considerat\ o complica]ie tardiv\ [i este deseori asociat\ cu multiple alte metastaze la nivelul altor organe. Chemoterapia [i radioterapia au fost insuficiente, ns\ ele au avut rolul de a mbun\t\]i calitatea vie]ii pacientului n ultimele luni, prin evitarea alter\rii func]iilor masticatorie, de degluti]ie [i de vorbire, precum [i prin evitarea snger\rilor locale tumorale.Cuvinte cheie: metastaz\ gingival\, epulis, cancer pulmonar, rezec]ie chirurgical\

malignancies). The most common sites of soft tissue involvement are the gingiva, tongue, lips, and the buccal and palatal mucosa. The primary tumours are mainly lung, breast, kidney and colon tumours, which represent about 70% of cases reported in the literature.1

Lung cancer represents an important cause of death in our country and it has a clearly consistent pattern of metastasis. The common sites of metastasis of lung cancer are: regional lymph nodes, brain, bone, adrenal gland and lung.2 Metastasis to the brain is the most feared complication of systemic cancer and the most common intracranial tumours in adults. Approximately 40% of intracranial neoplasms are metastatic.3

Metastasis to the gingiva from lung cancer is extremely rare. Metastatic tumours in the gingiva usually exhibit rapid growth and tend to cause various clinical symptoms seriously affecting the quality of life.2

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However, there have been few reports focusing on the management of gingival metastasis. So, we consider of a great importance to review our clinical records and the literature to determine the clinical characteristics of patients with lung cancer who develop gingival metastasis.

Between 1998 and 2006, we found only one case of gingival metastasis from among our patients with lung cancer. Of course, there are numerous cases when the primary tumour is represented by gingival tumour with lung metastases.

casE rEPort

The present case report describes a 60-year-old man, B. V., who was directed in our Department of Maxillo-Facial Surgery from ENT, who showed a large painless gingival mass, epulis like tumour, nut-sized, with approximate 4 cm in diameter, located on the anterior maxillary alveolar crest, with deep red crusts partially covered, sessile, having well-defined borders. (Fig. 1) Also, the patient presented a lot of maxillary untreated dental roots and massive deposits of tartar and bacterial plaque.

Figure 1. The massive epulislike gingival tumour.

The extraoral examination revealed the presence of the gingival tumour which hardly pushed the upper lip and determined functional and aesthetic consequences. (Fig. 2) Also the facial skin colour was modified, with a pale aspect of the face.

In this case, the patient’s medical history was very important and proved to be an important instrument in diagnosis. Hereby, the patient presented approximately one year ago symptoms like headache, nausea, vomiting, confusion and lethargy and decided to go to the hospital where he was directed to neurosurgery. Neurosurgeons discovered a brain tumour after performing a cerebral CT scan.

Figure 2. Extraoral examination: frontal and lateral view.

The investigations continued with a chest X-ray, which showed the existence of a small tumour in the left lung. According to Gaspar (2004), approximately one third of patients with lung carcinoma eventually develop intracranial metastases, and also 50% of brain metastases result from this type of cancer.4 That is why the suspicions that the lung tumour could be the primary tumour with consecutive brain metastasis are legitimate.

Neurosurgeons decided to remove the brain tumour, after clinical and laboratory investigations. On histological examination, the tumor had proved to be a brain metastasis from a lung cancer, more specifically, the brain tumour was an epidermoid carcinoma. After neurosurgery, patient underwent chemotherapy associated with 3 sessions of radiotherapy on the right temporo-parietal area.

As we mentioned before, approximately one year from the first operation, the patient was referred to our clinic because of the presence of a gingival epulislike tumour, weight-loss and anorexia.

The patient reported that the gingival tumour had begun to grow approximately 4 months ago, reaching to actual sizes and leading to functional problems in the act of mastication, swallowing and speaking. Also, sometimes, the tumour moderately bled, especially when the patient ate.

CT scan of the skull was performed, and revealed a brain tumour, consisting in a solid mass lesion of one cm diameter in the right cerebral hemisphere, with prominent surrounding edema. (Fig. 3) In the light of previous patient’s history of a brain tumour approximately one year ago, we concluded that a cerebral recurrence had developed.

We performed also a chest X-ray, which was very suggestive, showing the primary tumour, a massive round mass in the left lung, with a diameter of 10 cm, and some 1-cm diameter shadows in the right lung, which also

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indicated also several lung metastases. (Fig. 4)The CT scan in the axial plane of the upper jaw

revealed the gingival tumour (metastatic mass) of approximately 4 cm in diameter, which is well defined, adjacent to upper frontal alveolar crest, without bone resorbtion. (Fig. 5)

Figure 3. Cerebral CT scan shows a solid mass lesion of 1 cm diameter in the right emisphere.

Figure 4. Pulmonary X-ray film which reveals a large left lung tumour, with 10 cm diameter and, in the right lung, some mass shadows of 1 cm diameter.

Figure 5. Upper jaw CT scan shows a massive mass in front of the anterior maxilla.

The patient underwent biopsy of the gingival mass and unkeratinized G3 scuamous cell carcinoma was histologically diagnosed. We decided to do a palliative operation consisting in tumour removal, that was sent then for biopsy. Intraoperatively, there were no problems of persistent bleeding or of a primary closure. The patient received three courses of combination chemotherapy consisting of docetaxel and irinotecan followed by concurrent chemoradiotherapy consisting of cisplatin and thoracic irradiation.

The histology of the gingival tumour was the same as that of the brain metastasis which led us to the conclusion that the gingival tumour represented the second metastasis, after the brain localization, of the primary lung tumour.

An abdominal ecography was also performed but revealed no liver or other metastasis at that time of investigation.

dIscUssIons

Metastasis of cancer to the oral cavity is unusual.5 Furthermore, tumours metastasizing only to the oral mucosa were extremely rare and accounted for only about 0.1% of all oral malignancies. Metastatic tumours to the oral region may occur in the soft tissues or jawbones.6 More cases have been published on metastases in the jawbone than in oral soft tissues.7 From all tumors, lung cancer is believed to most commonly metastasize to the oral cavity.3 Several cases of metastatic lung carcinoma involving the periodontium were also reported in the literature.8 However, the real prevalence of gingival metastasis by lung cancer remains unclear.9 Hematogenous spread represents the mechanism of metastasis for this tumour. The brain metastasis are explained by arterial blood which must pass through the lungs before entering the brain, and larger aggregates of tumour cells are filtered out in the capillaries, and many emboli travelling to the brain.4

We retrospectively reviewed our clinical records of 146 patients with lung cancer hospitalized between 1998 and 2006. Among these patients, only one (0.68%) developed gingival metastasis during the clinical course of lung cancer. This patient, who previously developed brain metastasis, is the subject of the present article.

Epulis like gingival tumours metastases from lung cancer occur very rarely, especially with the dimensions and localization such as that found in the presented case and they could raise diagnostic and therapeutic difficulties. The bigest error could be to misjudgethe clinical lesion based on the objective clinical signs,

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as a benign tumour (epulis). Thus, patient’s medical history, sistemic investigations as well as investigation of target organs are very important and mandatory. Actually, a simple pulmonary X-ray had showed the main aspect of the case, the primary tumour, which had an insidious development.

Gingival metastasis is usually considered a late complication and is most often associated with multiple metastases in other organs. Furthermore, gingival metastasis progressively results in diverse clinical symptoms such as: mechanical disturbance of chewing, pain, or intermittent bleeding that seriously affect the quality of life of these patients with lung cancer.10 It may thus be a bad prognostic sign and death usually occurs within a few weeks or months caused by the progression of the primary tumour or by the spreading of cerebral metastases.11,12 Sometimes death occurs suddenly of unknown causes. Unfortunately, our patient’s condition deteriorated rapidly and he died of pneumonia three months later. Histopathologic study at autopsy confirmed that malignant cells of the gingival mass were identical to those in the primary lung tumour.

Chemotherapy and radiotherapy were insufficient but they had the role of improving the quality of life for the last months. The management of gingival metastases and also brain metastases is clearly a difficult problem and the therapeutic strategies summarized in the present article have not led to major improvements in survival, despite progress in several areas. Clearly, the most important aspect of management remains that of quality of life, symptom control and effective palliation, a major component of which is neurocognitive function (brain metastases).13

In our case, chemotherapy and radiotherapy seemed to have some benefits after primary operation of the brain tumour with a period of symptomatic relief for approximate 6 months. After that, the patient condition deteriorated progressively, firstly with the appearance of the gingival epulislike tumour and secondly, after tumor excision, followed by chemotherapy and local radiotherapy, when progressive alteration of the general status occured until death from pneumonia 3 months later.

concLUsIons

In conclusion, gingival metastases to lung cancer patients are extremely rare representing a late

complication and most often is associated with other organs metastases, such in our case. Early detection and appropriate treatment of gingival metastasis are very important because it significantly influences the quality of life. The removal of the gingival metastasis represents nevertheless a palliative treatment, but this could block the appearance of some functional problems in chewing, speaking, swallowing and avoid the local tumour bleeding.

There were case reportsof complete regression in small gingival metastases from lung cancer after receiving of one or some courses of combination chemotherapy consisting of ifosfamide, cisplatin, and vindesine.9,14 This fact represents an improvement of local control of the disease, without functional damages but, unfortunately, with a poor prognosis.

The clinical characters of the gingival mass were of an epulislike benign tumour, which could create confusion in diagnosis. Medical patient’s history as well as some target organs investigations, such as brain, lungs, liver, became mandatory to establish the correct diagnostic.

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