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Two cases of malignant interstitial cell carcinoma of the testis are reported. The first patient had no evidence of a virilizing syndrome. Basal plasma testosterone (T) was decreased, whereas plasma luteinizing hormone, estrone (E1), and androstenedione were elevated. These findings were diagnostic of a defect in T secretion as a result of a partial 17‐hydroxysteroid dehydrogenase deficiency as seen in male pseudohermaphroditism. In the second patient, showing gynecomastia and atrophic testis, endocrine studies revealed high plasma T and estradiol (E2); all measured Δ4 and Δ5 precursors of T were elevated resembling the pattern seen in virilizing adrenal carcinomas. Both patients were treated with radiotherapy without demonstrable effect. Chemotherapy—consisting of a combination of cis‐platinum, vinblastine, and bleomycin; then cyclophosphamide, doxorubicin, and vincristine; and finally o, p′‐DDD—was unsuccessful in reducing tumor bulk. Since malignant Leydig cell carcinomas are rare, this paper reviews the literature and makes recommendations concerning treatment.
Two cases of malignant interstitial cell carcinoma of the testis are reported. The first patient had no evidence of a virilizing syndrome. Basal plasma testosterone (T) was decreased, whereas plasma luteinizing hormone, estrone (E1), and androstenedione were elevated. These findings were diagnostic of a defect in T secretion as a result of a partial 17‐hydroxysteroid dehydrogenase deficiency as seen in male pseudohermaphroditism. In the second patient, showing gynecomastia and atrophic testis, endocrine studies revealed high plasma T and estradiol (E2); all measured Δ4 and Δ5 precursors of T were elevated resembling the pattern seen in virilizing adrenal carcinomas. Both patients were treated with radiotherapy without demonstrable effect. Chemotherapy—consisting of a combination of cis‐platinum, vinblastine, and bleomycin; then cyclophosphamide, doxorubicin, and vincristine; and finally o, p′‐DDD—was unsuccessful in reducing tumor bulk. Since malignant Leydig cell carcinomas are rare, this paper reviews the literature and makes recommendations concerning treatment.
Leydig cell carcinoma of the testicle is a rare tumor with 16 well-documented cases in the medical literature. Radiotherapy and chemotherapy have not been successful in treating patients with metastatic disease. The patient described in this report had widely metastatic Leydig cell carcinoma associated with hypertension, hypokalemic alkalosis, and an elevated serum concentration of desoxycorticosterone. He experienced an objective clinical remission and disappearance of pulmonary metastases while receiving o,p'-DDD as the sole chemotherapeutic agent. This response was similar to that found in patients with adrenal cortical carcinoma. The regression of this tumor with an adrenolytic agent, in conjunction with the similar morphologic and biochemical features of these two types of tumors, provides additional evidence of the close similarity between Leydig cell carcinoma and adrenal cortical carcinoma.
A case of malignant interstitial cell tumor of the testis is reported and the literature is reviewed. Malignant interstitial cell tumors occur exclusively in adults. Gynecomastia was noted in 12% of the cases. Elevated plasma levels or increased urinary excretion of estrogen, 17-ketosteroid, or testosterone were demonstrated in 64% of the patients. Vessel invasion, which occurred in 74% of the patients, appears to be the only reliable histologic feature for predicting malignant behavior. There is no proved sensitivity to radiation or chemotherapeutic regimens in patients with disseminated tumor. Once the diagnosis of malignancy is histologically established, inguinal and retroperitoneal lymph node dissections, even in the absence of clinical evidence of metastasis, may be beneficial in providing early staging of the tumor and, perhaps, in preventing subsequent lymphatic spread. Case ReportA 75-year-old man was seen with a two-month history of painless left scrotal enlargement. Physical examination showed enlargement of the left scrotal content to three times normal size. The right testis was of normal size. There was no gynecomastia. There was no history of trauma. Left suprainguinal exploration and orchiectomy were performed. A diagnosis of interstitial cell tumor with histologic features suggesting malignancy was made on pathologic examination. The ultrasound scan of the abdomen, intravenous pyelography, chest radiograph, serum alpha-fetoprotein level, serum beta H C G level, and urinary 17-ketosteroid excretion performed shortly after the surgery showed no abnormalities.Eight months later productive cough and left flank pain developed. The chest radiograph showed mutliple pulmonary Accepted for publication January 20, 1981. nodules. A computed tomography of the abdomen showed periaortic adenopathy. Physical examination revealed no peripheral lymphadenopathy or hepatosplenomegaly. There was no weight loss. The hemogram showed a hematocrit of 55.9%, a hemoglobin of 19.2 g/dl, an erythrocyte count of 6.08 X 103/mm3, a leukocyte count of 9100/mm3 with a normal differential count, and a platelet count of 252 X 103/mm3. Serum chemistry studies were normal except for an elevated alkaline phosphatase level (194 mU/ml). Blood volume studies showed an erythrocyte mass of 47.19 ml/kg (normal 20.0-36.5 ml/kg), and a plasma volume of 41.5 ml/kg (normal 23.4-43.8 ml/kg). The arterial blood gas studies performed at the same time showed no abnormalities. The serum alphafetoprotein, beta HCG, pituitary gonadotropin, and testosterone levels were normal. The patient was treated with Adriamycin (50 mg/m2) and/ Cytoxan (600 mg/m2). No response was observed after three cycles of therapy. The treatment was then changed to Cisplatinum (100 mg/m2). After two courses, the pulmonary nodules decreased slightly in size, but there was no change of the retroperitoneal metastasis. The patient died seven months after the detection of the metastasis. An autopsy was not performed. Pathologic FindingsThe removed left testis measured 5.5 X 4...
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