Respiratory Medicine (1991) 85, 533-534
Case Reports
Spontaneous bilateral pneumothoraces from synovial cell sarcoma C. O'LEARY, M. EL SOUSS! AND J. C o w m
Plymouth Chest Clinic, Freedom Fields, Plymouth
Introduction Bilateral pneumothorax is a rare but recognized manifestation ofintrathoracic metastases. We present a case of spontaneous bilateral pneumothoraces in a young woman with synovial cell sarcoma.
Case Report A 25-year-old woman presented with a 3-week history of right sided chest pain and a 3-day history of left-sided chest pain. She had become dyspnoeic on exertion but was asymptomatic at rest. Fourteen months earlier she had an apparently enlarging painful ganglion excised from the dorsolateral aspect of her right wrist. Histological examination revealed this to be a poorly differentiated synovial cell sarcoma with a preponderance of spindle Received19July 1990and acceptedin revisedform27 February 1991.
cells (Plate I). She had been treated with local radiotherapy and reviewed regularly in the Oncology out-patient Department. She was otherwise well and a non-smoker. There was no history of asthma, intravenous drug abuse or recent trauma. She was not distressed but her chest X-ray (Plate 2) revealed a small right apical pneumothorax and a 40% left pneumothorax. In view of her history of malignancy, a CT scan of her chest was performed (Plate 3). This showed bilateral nodular pleural and intrapulmonary lesions. The left pneumothorax was initially aspirated but re-accumulated immediately. A chest drain was inserted and spontaneous pleurodesis achieved with difficulty. She was referred for chemotherapy (adriamycin, ifosfamide). At 3 months, she was continuing with chemotherapy, and there had been no recurrence of pneumothorax.
Plate I Histologic section showing a poorly differentiated synovial cell sarcoma with a preponderence of spindle ceils. 0954-6111/91/060533 +02 $03.00/0
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C. O ' L e a r y et al.
Spontaneous pneumothorax resulting from primary bronchial carcinoma is extremely rare ( < 0.05% cases) (3). One case o f spontaneous bilateral pneumothoraces due to metastatic cervical carcinoma has been reported (4). Only one case of spontaneous bilateral pneumothoraces due to pulmonary metastases from a synovial cell sarcoma has been reported (5). Various mechanisms have been suggested to exlain how a pneumothorax occurs, including rupture of necrotic tumour causing a communication between bronchus and pleural cavity, and tumour nodules in the periphery of lung causing a ball valve action, with overdistension of the lung forming subpleural bullae which rupture. Rarely has tumour spread to the pleura itself been demonstrated as the cause. CT scanning in this case demonstrated not only pulmonary deposits, but bilateral pleural deposits.
References Plate 2 Chest X-ray showing a small right apical pneumothorax and a 40% left pneumothorax.
Discussion
Spontaneous pneumothorax is an uncommon manifestation of malignant metastatic pulmonary disease. The most common primary tumour is a sarcoma, and of these the majority are osteosarcoma (1). Of 42 cases of spontaneous pneumothorax occurring in primary bone tumours, six presented with bilateral pneumothoraces (2). Other reported tumours, in decreasing frequency, are non-gestational choriocarcinoma, Ewing's tumour and malignant melanoma.
Plate 3
1. Stozenberg J, Clements JP. Bilateral simultaneous pneumothorax during radiation therapy for metastatic disease from osteogenic sarcoma. Radiol Clin Biol 1970; 39: 437-442. 2. Janetos GP, Ochsner SF. Bilateral pneumothorax in metastatic osteogenic sarcoma. Am Rev Resp Dis 1963; 88: 73. 3. Wright FW. Spontaneous pneumothorax and pulmonary malignant disease. A syndrome sometimes associated with ~cavitating tumour. Clin Radio11976; 27:211-222. 4. Lane S, Fascomo, Levitt, BrandsJ[etter, Letter. Spontaneous bilateral pneumothorax due to metastatic cervical carcinoma. Chest 1987; 91: 151-152. 5, Singh, Singh, Kaur. Bilateral spontaneous pneumothorax with pulmonary metastases from synovial cell sarcoma. Br J Dis Chest 1977; 71:211-212.
CT scan of the chest demonstrating bilateral nodular pleural and intrapulmonary lesions.