Synchronous Testicular Seminoma and Adrenocortical Carcinoma: A Case Report
Takahiro Inoue, Akito Terai,* Toshiro Terachi, Takahito Souma, and Osamu Yoshida Department of Urology, Faculty of Medicine, Kyoto University, Kyoto, Japan
We report a rare case of synchronous testicular seminoma and adrenocortical carcinoma. A 57-year-old man had a left testicular seminoma (clinical stage IIIB) with metastases to the lung and paraaortic lymph node. A complete response was obtained after 3 courses of chemotherapy with single-agent carboplatin. However, a left adrenal tumor was detected 12 months later and demonstrated a tumor volume doubling time of 2.1 months. Chemotherapy with bleomycin, etoposide and cisplatin failed to stop the tumor growth. A laparoscopic adrenalectomy was performed and pathology revealed an adrenocortical carcinoma. The patient has been free of recurrence for 42 months postoperatively.
Int J Urol 1998;5:615-617
Key words: testicular cancer, seminoma, adrenocortical carcinoma, synchronous double cancers, laparoscopic adrenalectomy
INTRODUCTION
Adrenocortical carcinoma is a rare malignancy with an annual incidence of 0.1-0.2 per population of 100,000.1 We report a case of a rapidly growing adrenocortical carcinoma detected during the follow-up for testicular seminoma after chemotherapy with single-agent carbo- platin. The present case represents synchronous double cancers because the adrenal cancer was detected 12 months after the diagnosis of testicular cancer.
CASE REPORT
A 57-year-old man was referred in September 1992 for further treatment of a left testicular tumor following a left high orchiectomy. The histopathologic diagnosis was a pure seminoma. Preoperative serum levels of alpha- fetoprotein (AFP) and human choriogonadotropin-beta (HCG-ß) were normal, and the serum lactate dehydro- genase (LDH) level was 521 mU/mL (reference range, 150 to 450 mU/mL) preoperatively, but normalized soon after orchiectomy. CT scans demonstrated me- tastases to the left paraaortic lymph node just below the level of the left renal vein (3.5 cm in diameter) and to the right lung (1 cm in diameter). The patient was clinically diagnosed as having stage IIIB disease. He was treated with 3 courses of single-agent carboplatin
(400 mg/m2) during a 3-month period after informed consent was obtained. The pulmonary lesion disap- peared after 1 course of chemotherapy, and the size of the retroperitoneal mass was reduced to 0.5 cm after 3 courses of carboplatin therapy and became undetect- able in September 1993.
However, a follow-up CT scan demonstrated a solid adrenal tumor in April 1994 (Fig. 1D). A retro- spective review of all CT films revealed that an adrenal mass had been barely recognizable 7 months previously (Fig. 1B). The adrenal tumor showed a rapid growth rate with a doubling time of 2.1 months (Fig. 1B, C, D). Because the recurrence of seminoma was strongly suspected and the patient lacked a clinical presentation of steroid excess, a hormonal examination was not performed. Serum levels of AFP, HCG-ß and LDH had been normal throughout this period. Despite 2 courses of chemotherapy with bleomycin, etoposide and cisplatin (BEP regimen), tumor growth continued and was accompanied by a cystic change (Fig. 1E, F). The adrenal tumor was then resected laparoscopically in July 1994. Macroscopically, the resected tumor was well encapsulated, measured 6.3 x 5.5 x 5.0 cm and weighed 70 grams. On cross-section, central necrosis and peripherally compressed normal adrenal tissue was observed (Fig. 2A). The histopathologic diagnosis was an adrenocortical carcinoma (Fig. 2B). The patient has remained free of disease for 42 months.
DISCUSSION
According to data from the Surveillance, Epidemiol- ogy, and End Results (SEER) program from 1973 to 1987 in the United States, the age-adjusted incidence
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rates of testicular germ cell tumors and adrenocortical carcinoma were 3.2 to 4.0 per 100,000 and 0.1 to 0.2 per 100,000 for all races and both sexes, respectively.1,2 Cumulative results from several studies have shown that 26 of 160 (16%) patients with adrenocortical carci- noma had second primary carcinomas, 3-5 and only 1 of the 26 patients had adrenocortical carcinoma and semi- noma. Unfortunately, the clinical details of this patient were not described. Therefore, the present case dem- onstrates a very rare case of synchronous double can- cers consisting of a testicular germ cell tumor followed by an adrenocortical carcinoma.
This patient with a clinical stage IIIB seminoma was initially treated with single-agent carboplatin. Cisplatin-based combination chemotherapy had been established as the standard treatment for disseminated testicular germ cell tumors. However, on the basis of several preliminary reports that single-agent carboplatin was an effective and nontoxic treatment for advanced seminoma,6,7 we treated 5 patients with advanced semi- noma with single-agent carboplatin between May 1990 and August 1992.8 The present case was treated with single-agent carboplatin because the tumor burden was not massive (a 3.5 cm solitary lymph node metastasis and a 1 cm solitary pulmonary metastasis). Subsequently, we abandoned carboplatin therapy for advanced semi-
noma because it was shown that more than 25% of patients relapse and require cisplatin-based salvage chemotherapy during a follow-up period of 3 years or less.6,7 We suspected that the rapidly growing adrenal mass was an adrenal metastasis from the primary tes- ticular seminoma. The serum markers (AFP, HCG-ß and LDH) were normal during the follow-up period and were not helpful for a differential diagnosis. In our review of the literature, we could find no clinical case of seminomatous metastasis to the adrenal gland. On the other hand, autopsy has revealed that the adrenal gland is a relatively common site of metastatic involvement. Bredael et al.9 found that 29% of 154 patients with germ cell tumors of the testis had adrenal metastasis at autopsy. Also, Mostofi and Price10 noted in autopsy studies that the left adrenal gland was more often involved than the right (35% vs. 9%). Anatomical characteristics of lym- phatic drainage at the renal hilum may account for the predominance of left adrenal involvement. However, adrenal metastasis is generally considered a late event.
In our experience with 100 transperitoneal lap- aroscopic adrenalectomies, 2 adrenocortical cancers, including the present case, have been resected laparo- scopically.11 Using the anterior transperitoneal approach, the adrenal tumor itself can be identified easily through Gerota’s fascia. Early clipping of the inferior adrenal
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vein and intentional broad excision of the adrenal gland and the fatty tissue resulted in a safe adrenalectomy with negative surgical margins. Elashry et al.12 recently reported 2 successful laparoscopic adrenalectomies for a solitary adrenal metastasis from patients with renal cell carcinoma. However, there is the possibility of intra- operative tumor rupture because the adrenal tumor tends to be brittle and has a thin capsule. Laparoscopic adre- nalectomy for malignant diseases still remains an inves- tigative treatment and must be performed by highly
skilled laparoscopists who can properly decide when to convert to open surgery.
In summary, we describe a very rare case of double cancers in which an adrenocortical carcinoma was de- tected 12 months after single-agent carboplatin therapy for testicular seminoma. The preoperative diagnosis was confounded by the short interval between the appear- ance of the testicular and adrenal cancers, a rapid growth rate of the adrenal carcinoma and a lack of significant serum tumor markers.
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