Annals of Oncology 11: 887-889, 2000. © 2000 Kluwer Academic Publishers. Primed in the Netherlands.
Clinical case Spontaneous pneumothorax in malignancy: A case report and review of the literature S. Srinivas1 & G. Varadhachary2 1
Veterans Affairs Medical Center (11 lone), Palo Alto, California; 2Baylor College of Medicine, Houston, Texas, USA
thorax occurred with a variety of primary tumors. However it was always associated with lung metastases or lung involveBackground: Pneumothorax occurring in the absence of ob- ment with tumor. In certain cases the metastases were detected vious lung disease is denned as spontaneous pneumothorax. after the occurrence of pneumothorax. Conclusions: The occurrence of pneumothorax in a patient Spontaneous pneumothorax occurs in a variety of settings in with malignancy should prompt a search for lung metastases. patients with malignancies. Patients and methods: We present a case report of spontaneous pneumothorax in malignancy and review the literature. Results: No correlation was found between the occurrence Key words: chemotherapy, lung metastases, spontaneous of pneumothorax with age, sex or smoking history. Pneumo- pneumothorax Summary
Introduction
Pneumothorax occurring in the absence of obvious lung disease is denned as spontaneous primary pneumothorax. It is usually a disease affecting young men with a higher relative risk in smokers than non-smokers [1]. The mechanism is thought to be due to a rupture of a small apical subpleural emphysematous bleb that is either congenital or caused by bronchiolar inflammation and obstruction. However, secondary spontaneous pneumothorax occurs from a variety of diseases affecting the airways and parenchyma. It has been reported in patients with chronic obstructive lung disease, lung infections including abscess, tuberculosis, pneumocystis carinii pneumonia (PCP) and in malignancies. A more recently described category is spontaneous pneumothorax with malignancy [2]. We present a case of a patient with spontaneous pneumothorax, and review the literature and discuss the possible mechanisms.
(Figure 1). (3-HCG at that time was 130. Patient was started on chemotherapy with cisplatin, etoposide, and bleomycin (PEB) on 3 July 1996. Eight days later he
Case report A 54-year-old white male, smoker, underwent an orchiectomy for a right testicular pure seminoma in January 1995. He underwent adjuvant radiotherapy with 2500 cGy to the para aortic nodes and 2500 cGy to the pelvic area and scrotal scar in March 1995. He did well for 11 months until February 1996 when he presented with an increasing P-human chorionic gonadotropin (P-HCG) and a CT scan revealed extensive metastasis involving the liver, mediastinum, and multiple lung nodules
Figure 1. Multiple bilateral lung nodules.
888 Table 1. Causes of spontaneous pneumothorax in malignancy. Causes Tumor invasion Invasion of pleura Vascular invasion Tumor shrinkage Chemotherapy related Radiation Spontaneous necrosis Mechanical effects Bronchial obstruction Tumor embolus Contributing effects Defective repair mechanism Severe emesis Secondary infections Invasive aspergillosis, PCP Instrumentation Coincidental
Figure 2. Bilateral pneumothorax after chemotherapy.
developed sudden onset of chest pain and dyspnea and a chest X-ray revealed bilateral pneumothoraces requiring bilateral chest tubes, with more than 50% pneumothorax on the right and 30% on the left (Figure 2). His p-HCG at that time was 3.6. The chest X-ray showed no significant change in tumor size. Patient had not undergone any invasive chest diagnostic procedure. He subsequently received a total of four cycles of PEB uneventfully. He has had a complete response with no recurrence of disease or pnemothorax at the last follow-up.
Discussion Spontaneous pneumothorax has been reported in association with many malignancies. It is usually seen with primary or metastatic lung lesions, occasionally presenting even before detection of the underlying malignancy. The most common malignancies with which it has been described are metastatic germ cell tumors, osteogenic and soft tissue sarcomas and primary lung cancers [3-5, 10]. The various possible causes and mechanisms of malignancy associated pneumothorax are listed in Table 1. When the small airways are narrowed by cancer invasion they act as a check valve, causing air trapping and dilation of distal alveolar spaces and eventual rupture
Possible mechanism
Broncho-pleural fistula
Shrinkage of a subpleural Unclear, reported with Hodgkin's metastases Check valve mechanism and rupture of subpleural bleb
[7]. Bronchopleural fistula can be caused by either direct tumor invasion or develop secondary to necrosis of a peripheral tumor from effective chemotherapy, or spontaneous vascular occlusion within the tumor itself. Direct invasion of the pleura by tumor is also a possible mechanism though relatively uncommon [8]. Pneumothorax following radiation therapy and following bleomycin lung have been described. A medline search of the English literature from 1966 to the present identified numerous cases of pneumothorax associated with chemotherapy administration in a variety of malignancies (Table 2). In our present review no correlation was found with age, sex or smoking history. It was not restricted to chemotherapy sensitive tumors nor was it associated with any one chemotherapy regimen. However it was always associated with lung involvement either with metastases or lung primaries, including cases where the metastases were detected after the occurrence of pneumothorax. While this is a small review with limited follow-up, it appears that the occurrence of pneumothorax itself does not affect prognosis, with survival being dependent on the underlying tumor. Treatment of pneumothorax should be based on the extent of pneumothorax as well as the nature of the underlying disease.
Conclusions Any patient with lung metastases is at risk of developing spontaneous pneumothorax. The occurrence of pneumothorax in patients with malignancies should prompt a search for lung metastases or be followed closely for the occurrence of a lung primary.
889 Table 2. Occurrence of pneumothorax in association with chemotherapy. Age/sex
Tumor
Therapy
Site
Resp toTx
Reference
14/F
Wilms Wilms Uterine Uterine Seminoma Lung cancer Lung cancer Lung cancer Ovarian Testicular Synovial cell
C/XRT C/XRT
Bilateral Unilateral Bilateral Bilateral Unilateral Unilateral Bilateral Bilateral Bilateral Bilateral Bilateral Bilateral Bilateral Bilateral Bilateral
None None None Progression
AJR 1980; 134: 1231 AJR 1980; 134: 1231 AJOb/Gy; 1982; 142: 705 AJob/gyn; 1982;142: 705 Cancer 1995; 752710 Chest 1992; 102: 628-9 Chest 1992; 102: 628-9 Chest 1991; 100:853-5 Chest 1975; 75: 194-6 Chest 1975; 75: 194-6 Mayo 1973; 48: 541-4 Mayo 1973; 48: 541^t Cancer 1975; 35: 936-45 Cancer 1981; 47: 1743-54 Ann Oncol 1992; 3: 297
6/M
56/F 70/F 49/M 57/M 67/M 47/M 19/F 21/M 20/M 18/M 15/M 33/M 29/F
OGS OGS
Synovial cell Trophoblastic
C C C C C C C C C C C C C
Yes Yes Yes
None Yes Yes Yes
Not known Yes Yes
Not known
Abbreviations: C - chemotherapy; OGS - osteogenic sarcoma.
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mechanism as a cause of bilateral spontaneous pneumothorax complicating bronchioalveolar cell carcinoma. Chest 1991; 100: 853. O'Connor BM, Zeigler P, Spaulding MB. Spontaneous pneumothorax in small-cell lung cancer. Chest 1992; 102: 628-9. Doll DC. Fatal pneumothorax associated with bleomycin-induced pulmonary fibrosis. Cancer Chemother Pharmacol 1986; 17 (3): 294-5. 10. Lote K, Dahl O, Vigander T. Pneumothorax during combination chemotherapy. Cancer 1981; 47: 1743-5. Received 12 April 1999; accepted 14 May 1999.
Correspondence to: S. Srinivas, MD Veterans Affairs Medical Center (11 lone) 3801 Miranda Avenue Palo Alto, CA 94304 USA E-mail:
[email protected]