Caso raro de pneumotórax: adamantinoma metastático

Abstract

A 26-year-old Caucasian male with a history of local trauma had been experiencing an increase in left leg volume, accompanied by pain, for eight months. An X-ray of the leg revealed an osteolytic tumor in the tibial diaphysis. We performed tumor resection with histologically tumorfree margins. Seven years later, the patient presented local recurrence and underwent below-knee amputation of the affected limb. Three years later, he presented dyspnea upon moderate exertion, and, at that time, right pneumothorax was diagnosed, and nodular images, some of which were cystic, were seen in both lungs (Figure 1). Those images subsequently proved to be lung metastases of adamanti- IntroductionAdamantinoma is a rare tumor that affects long bones, and it is estimated to account for 0.1 to 0.5% of all primary bone tumors.(1) It is named for its histological similarity to the tumor of the mandible (ameloblastoma).Adamantinoma is a low-grade neoplasm that presents an indolent course, is locally aggressive, and rarely metastasizes. The secondary sites most frequently affected are the lungs, lymph nodes, and bones. When it is present in the lungs, hemoptysis can occur.Although cases of pneumothorax secondary to other metastatic tumors have been described, there are, in the literature, only three references to metastatic adamantinoma of the lung as the etiology of spontaneous pneumothorax.(2,3,4) Here, we report two cases followed by the authors. Here, we describe two cases of lung metastasis of adamantinoma of long bones, a low-grade bone neoplasm that rarely metastasizes. In both cases, the clinical presentation of the metastases was characterized by spontaneous pneumothorax secondary to tumor cavitation, a phenomenon described in only three of the studies reviewed in the literature. Clinical, radiological, and anatomopathological findings, as well as the procedures adopted in the two cases, are described.Keywords: Adamantinoma; Pneumothorax; Neoplasm metastasis; Medical records. ResumoDescrevem-se dois casos de metástases pulmonares de adamantinoma de ossos longos, o qual é uma neoplasia óssea de baixo grau que raramente metastatiza. Nos dois casos a apresentação clínica das metástases se deu por pneumotórax espontâneo secundário a escavação tumoral, fenômeno descrito em apenas três dos trabalhos consultados na literatura. São descritos os achados clínicos, radiológicos e anatomopatológicos, bem como os procedimentos adotados nos dois casos.

Interventional management of extensive pulmonary metastasis in adamantinoma

“…There have been several reports of secondary spontaneous pneumothorax due to either pleural metastasis or rupture of a peripherally placed lung mass. [7] Management in the cases encountered had ranged from metastasectomy to chest tube drainage and pleurodesis. [78] In our case, pleurodesis was the only option feasible due to extensive nature of the pulmonary disease.…”

“…[7] Management in the cases encountered had ranged from metastasectomy to chest tube drainage and pleurodesis. [78] In our case, pleurodesis was the only option feasible due to extensive nature of the pulmonary disease. Bronchoscopy of the two cases with suspected endobronchial metastasis had confirmed the same, and these patients subsequently succumbed to respiratory insufficiency.…”