Abstract
We report a case of malignant large cell calcifying Sertoli cell tumor with endocrine overactivity resulting in persistent gynecomastia. Synchronizing with the development of metastatic lesions, the urine titers of 17-ketosteroids and estradiol were markedly increased. To our knowledge our case is the first report of malignant large cell calcifying Sertoli cell tumor demonstrating overt endocrine overactivity. CASE REPORT A 29-year-old Japanese man presented with a left testicular mass and gynecomastia. Familial and personal histories were unremarkable. Imaging studies demonstrated metastatic lesions in the lung and retroperitoneal lymph nodes. The removed left 12 x 8 x 7 cm. testicular tumor revealed solid cut surfaces with scattered calcification, hemorrhage and necrosis. Microscopically the tumor was comprised of atypical polygonal cells with oval nuclei and abundant eosinophilic cytoplasm, which were arranged in tubular fashion (part A of figure). There were various sized calcifications scattered in the tumor, frequently showing concentric, mulberry-like features. The tumor cells demonstrated mitotic activity (a10 high power fields), extratesticular growth and vascular invasion. Diagnosis was malignant large cell calcifying Sertoli cell tumor. The tumor cells were immunoreactive for estradiol (part B of figure) and aneuploid deoxyribonucleic acid was apparent on histogram. No remarkable effect was obtained with systematic radiotherapy and chemotherapy. As the metastatic lesions progressed, urine secretion of 17-ketosteroids (up to 63.5 mg. daily, normal 2.0 to 7.6) and estradiol (up to 230.2 pg. daily, normal 0.1 to 3.0) increased markedly. Gynecomastia continued during the course of the disease. The patient died of lung metastases 27 months after onset of the primary lesion. Autopsy disclosed extensive metastatic lesions in the lung, right kidney, small intestine and systemic lymph nodes. The histopathology was nearly similar to that of the
Published Version
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