Pulmonary Sarcoidosis Associated with Leydig Cell Testicular Neoplasm
The association between neoplastic diseases and sarcoidosis is well known. Among many types of tumor, some testicular neoplasms have also been associated with the development of intrathoracic sarcoid lesions. We report here the first case of association between Leydig cell testicular tumor and biopsy-proven pulmonary sarcoidosis.
A 33-year-old man came for observation in March, 1985 because of a moderate fever with exertional dyspnea and multiple bilateral opacities on chest roentgenogram (Fig 1). He was submitted in May, 1984 to a left orchiectomy at another hospital because of a painless mass, diagnosed as seminoma on frozen sections. However, the final diagnosis on routinely-processed sections was a Leydig cell tumor.
The slides were reviewed at the Department of Pathology, Paul-Brousse Hospital, Paris, where the diagnosis of a Leydig cell tumor was confirmed. Levels of beta-HCG and alpha-fetoprotein were within normal limits, while serum angiotensin-converting enzyme was at the upper normal level. Chest roentgenogram taken at admission showed bilateral multiple lung infiltrates interpreted as metastases, while retroperitoneal lymphangiogram raised some doubts about lymph node involvement. For this reason, and considering that a small percentage of Leydig-cell tumors are indeed malignant, treatment with adriamycin (VP-16) and cisplatin was started. Other treatments with cisplatin, vinblastine and bleomycin followed in November, 1984 and January, 1985, with no appreciable change on chest roentgenogram. A second retroperitoneal lymphangiogram was normal. buy actos online
At this point, lung biopsy (performed through a left thoracotomy) showed typical sarcoid tissue involving lung parenchyma free of metastatic tumor cells (Fig 2). We started treatment with prednisone 60 (mg/day, gradually tapered to 25 mg) and continued therapy for ten months.
Treatment was followed by a sharp improvement in both chest roentgenogram (Fig 3) and spirometric values, where the most striking effect was represented by the disappearance of diffusion troubles but persistence of moderate restriction. The patient is now in good health and chest roentgenogram is unchanged ten months after discontinuing steroid therapy.
Figure 1. Chest roentgenogram after four courses of chemotherapy, before corticosteroid treatment. Multiple bilateral parenchymal infiltrates are evident.
Figure 2. Lung biopsy specimen showing epithelioid nodules with giant cells and areas of fibrosis. There is no evidence of necrosis.
Figure 3. Chest roentgenogram after ten months of corticosteroid treatment. Regression of parenchymal infiltrates with residual fibrosis is evident.