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Primary carcinoid of the testis 25 years after contralateral testicular seminoma
CASE REPORTS
PRIMARY CARCINOID OF THE TESTIS 25 YEARS AFTER CONTRALATERAL TESTICULAR SEMINOMA ADAM J. SINGER
AND
KARL H. ANDERS
ABSTRACT We report a case of primary testicular carcinoid occurring coincidentally 25 years after contralateral radical orchiectomy for seminoma, which has not been previously reported. UROLOGY 57: 554xiii–554xiv, 2001. © 2001, Elsevier Science Inc.
C
arcinoid rarely affects the testis. We report the first case of testicular carcinoid occurring coincidentally 25 years after a contralateral orchiectomy for seminoma. CASE REPORT A 68-year-old man presented with left orchalgia of 3 months’ duration. He had undergone right radical orchiectomy and radiation for Stage A seminoma in 1973, 2 years after his vasectomy (Fig. 1). His medical history was significant for hypertension, hypercholesterolemia, diabetes mellitus, coronary artery disease, psoriasis, basal cell carcinoma, arthritis, colonic tubular adenomas, and carpal tunnel release. His medicines were nifedipine 60 mg, glyburide 3.75 mg, lovastatin 5 mg, and aspirin 81 mg daily. His physical examination was normal except for a 14.5-mm lower pole left intratesticular mass. Sonographically, it was isoechoic centrally with a hypoechoic rim (Fig. 2A). The complete blood count, electrolytes, creatinine, alpha-fetoprotein, beta-human chorionic gonadotropin, and prostate-specific antigen determination and urinalysis were normal. His serum testosterone level was 139 ng/dL (normal range 230 to 700 ng/dL). A left radical orchiectomy was done (Fig. 2B). The postoperative 24-hour urinary 5-hydroxy-indole acetic acid (5-HIAA) test, chest radi-
FIGURE 1. Right orchiectomy specimen revealed a pure seminoma. Sheets of large tumor cells were interspersed between lymphocytes and plasma cells. Neoplastic cells demonstrated abundant clear cytoplasm, distinct nuclear membranes, and frequent large central nucleoli positive for placental alkaline phosphatase and negative for chromogranin staining. Hematoxylin-eosin stain, original magnification ⫻400.
ography, upper/lower gastrointestinal series, and abdominal/pelvic computed tomography were normal. At the last follow-up visit, 3 years later, he was disease free. COMMENT
From the Departments of Urology and Pathology, Southern California Permanente Medical Group, Woodland Hills, California; and Department of Pathology and Laboratory Medicine, University of California, Los Angeles, School of Medicine, Los Angeles, California Address for correspondence: Adam J. Singer, M.D., Department of Urology, Southern California Permanente Medical Group, 5601 DeSota Avenue, Woodland Hills, CA 91365 Submitted: September 5, 2000, accepted (with revisions): October 30, 2000 © 2001, ELSEVIER SCIENCE INC. ALL RIGHTS RESERVED
First described by Simon in 1954, Zavala-Pompa and coworkers1 had 2 patients with primary testicular carcinoid and reviewed the 55 cases in the published reports. Few have been reported since then.2 Although seminoma and carcinoid was coincidental and metachronous in the present patient, 14 of 57 carcinoids were mixed within mature teratomas.1,2 Patients were 10 to 83 years old 0090-4295/01/$20.00 PII S0090-4295(00)01040-2 554xiii
FIGURE 2. (A) Ultrasound scan demonstrating a 14.5-mm mass within a solitary left testis (between caliper markings). The neoplasm was homogeneously isoechoic at its center and surrounded by a hypoechoic ring. (B) Left orchiectomy specimen revealing a pure carcinoid tumor. Distinct ribbons and nests of uniform, small, round cells retract from the surrounding stroma. No evidence of mature or immature teratomatous elements was seen. Tumor cells were positive for chromogranin and negative for placental alkaline phosphatase staining. Hematoxylin-eosin stain, original magnification ⫻400.
(mean age 45.8). The left testis was affected 62% of the time, and one was bilateral. The most common clinical presentations were a discrete mass (47%) and testicular enlargement (36%). Carcinoid syn-
554xiv
drome was present in 12% of patients, of whom four had metastasis and three did not. Larger tumors (average 7.3 cm) were more likely to metastasize than smaller tumors (average 2.9 cm), but the histologic examination could not distinguish between benignity and malignancy. On ultrasound, the carcinoids were nonspecific, although most were homogeneous and hypoechoic with or without acoustic shadowing from calcification. Treatments included orchiectomy (77%), surgery with radiotherapy and/or chemotherapy (16%), tumor excision (2%), and was unspecified in 5%. Three patients died of metastasis. Sutherland and associates3 outlined a treatment algorithm for primary testicular carcinoid. Because carcinoid is rarely suspected preoperatively, a 24hour urinary 5-HIAA determination serves as a baseline tumor marker after the diagnosis has been established. Abdominal computed tomography to evaluate for metastases and gastrointestinal contrast studies to search for primary midgut carcinoid are recommended, since metastasis may occur without an elevated urinary 5-HIAA. Radical orchiectomy is curative for organ-confined carcinoid. Retroperitoneal lymphadenectomy is not recommended unless computed tomography reveals metastasis. Resection of the metastasis should be considered, regardless of size, because remissions may occur, as carcinoids tend to grow slowly.3,4 Chemotherapy is minimally beneficial for metastatic disease.3,4 Follow-up should include history, physical examination, and 24-hour urinary 5-HIAA every 3 months for 1 year and then annually.3 REFERENCES 1. Zavala-Pompa A, Ro JY, El-Naggar A, et al: Primary carcinoid tumor of testis: immunohistochemical, ultrastructural, and DNA flow cytometric study of three cases with a review of the literature. Cancer 72: 1726 –1732, 1993. 2. Czopek J, and Frasik W: Carcinoid tumor as a part of mature teratoma of the testis. Pol J Pathol 47: 91–93, 1996. 3. Sutherland RS, Wettlaufer JN, and Miller GJ: Primary carcinoid tumors of the testicle: a case report and management schema. J Urol 148: 880 – 882, 1992. 4. Debas HT, and Orloff SL: Carcinoid tumors and the carcinoid syndrome, in Sabiston DC Jr, and Lyerly HK (Eds): Textbook of Surgery, 15th ed. Philadelphia, WB Saunders, 1997, pp 950 –954.
UROLOGY 57 (3), 2001
PRIMARY CARCINOID OF THE TESTIS 25 YEARS AFTER CONTRALATERAL TESTICULAR SEMINOMA ADAM J. SINGER
AND
KARL H. ANDERS
ABSTRACT We report a case of primary testicular carcinoid occurring coincidentally 25 years after contralateral radical orchiectomy for seminoma, which has not been previously reported. UROLOGY 57: 554xiii–554xiv, 2001. © 2001, Elsevier Science Inc.
C
arcinoid rarely affects the testis. We report the first case of testicular carcinoid occurring coincidentally 25 years after a contralateral orchiectomy for seminoma. CASE REPORT A 68-year-old man presented with left orchalgia of 3 months’ duration. He had undergone right radical orchiectomy and radiation for Stage A seminoma in 1973, 2 years after his vasectomy (Fig. 1). His medical history was significant for hypertension, hypercholesterolemia, diabetes mellitus, coronary artery disease, psoriasis, basal cell carcinoma, arthritis, colonic tubular adenomas, and carpal tunnel release. His medicines were nifedipine 60 mg, glyburide 3.75 mg, lovastatin 5 mg, and aspirin 81 mg daily. His physical examination was normal except for a 14.5-mm lower pole left intratesticular mass. Sonographically, it was isoechoic centrally with a hypoechoic rim (Fig. 2A). The complete blood count, electrolytes, creatinine, alpha-fetoprotein, beta-human chorionic gonadotropin, and prostate-specific antigen determination and urinalysis were normal. His serum testosterone level was 139 ng/dL (normal range 230 to 700 ng/dL). A left radical orchiectomy was done (Fig. 2B). The postoperative 24-hour urinary 5-hydroxy-indole acetic acid (5-HIAA) test, chest radi-
FIGURE 1. Right orchiectomy specimen revealed a pure seminoma. Sheets of large tumor cells were interspersed between lymphocytes and plasma cells. Neoplastic cells demonstrated abundant clear cytoplasm, distinct nuclear membranes, and frequent large central nucleoli positive for placental alkaline phosphatase and negative for chromogranin staining. Hematoxylin-eosin stain, original magnification ⫻400.
ography, upper/lower gastrointestinal series, and abdominal/pelvic computed tomography were normal. At the last follow-up visit, 3 years later, he was disease free. COMMENT
From the Departments of Urology and Pathology, Southern California Permanente Medical Group, Woodland Hills, California; and Department of Pathology and Laboratory Medicine, University of California, Los Angeles, School of Medicine, Los Angeles, California Address for correspondence: Adam J. Singer, M.D., Department of Urology, Southern California Permanente Medical Group, 5601 DeSota Avenue, Woodland Hills, CA 91365 Submitted: September 5, 2000, accepted (with revisions): October 30, 2000 © 2001, ELSEVIER SCIENCE INC. ALL RIGHTS RESERVED
First described by Simon in 1954, Zavala-Pompa and coworkers1 had 2 patients with primary testicular carcinoid and reviewed the 55 cases in the published reports. Few have been reported since then.2 Although seminoma and carcinoid was coincidental and metachronous in the present patient, 14 of 57 carcinoids were mixed within mature teratomas.1,2 Patients were 10 to 83 years old 0090-4295/01/$20.00 PII S0090-4295(00)01040-2 554xiii
FIGURE 2. (A) Ultrasound scan demonstrating a 14.5-mm mass within a solitary left testis (between caliper markings). The neoplasm was homogeneously isoechoic at its center and surrounded by a hypoechoic ring. (B) Left orchiectomy specimen revealing a pure carcinoid tumor. Distinct ribbons and nests of uniform, small, round cells retract from the surrounding stroma. No evidence of mature or immature teratomatous elements was seen. Tumor cells were positive for chromogranin and negative for placental alkaline phosphatase staining. Hematoxylin-eosin stain, original magnification ⫻400.
(mean age 45.8). The left testis was affected 62% of the time, and one was bilateral. The most common clinical presentations were a discrete mass (47%) and testicular enlargement (36%). Carcinoid syn-
554xiv
drome was present in 12% of patients, of whom four had metastasis and three did not. Larger tumors (average 7.3 cm) were more likely to metastasize than smaller tumors (average 2.9 cm), but the histologic examination could not distinguish between benignity and malignancy. On ultrasound, the carcinoids were nonspecific, although most were homogeneous and hypoechoic with or without acoustic shadowing from calcification. Treatments included orchiectomy (77%), surgery with radiotherapy and/or chemotherapy (16%), tumor excision (2%), and was unspecified in 5%. Three patients died of metastasis. Sutherland and associates3 outlined a treatment algorithm for primary testicular carcinoid. Because carcinoid is rarely suspected preoperatively, a 24hour urinary 5-HIAA determination serves as a baseline tumor marker after the diagnosis has been established. Abdominal computed tomography to evaluate for metastases and gastrointestinal contrast studies to search for primary midgut carcinoid are recommended, since metastasis may occur without an elevated urinary 5-HIAA. Radical orchiectomy is curative for organ-confined carcinoid. Retroperitoneal lymphadenectomy is not recommended unless computed tomography reveals metastasis. Resection of the metastasis should be considered, regardless of size, because remissions may occur, as carcinoids tend to grow slowly.3,4 Chemotherapy is minimally beneficial for metastatic disease.3,4 Follow-up should include history, physical examination, and 24-hour urinary 5-HIAA every 3 months for 1 year and then annually.3 REFERENCES 1. Zavala-Pompa A, Ro JY, El-Naggar A, et al: Primary carcinoid tumor of testis: immunohistochemical, ultrastructural, and DNA flow cytometric study of three cases with a review of the literature. Cancer 72: 1726 –1732, 1993. 2. Czopek J, and Frasik W: Carcinoid tumor as a part of mature teratoma of the testis. Pol J Pathol 47: 91–93, 1996. 3. Sutherland RS, Wettlaufer JN, and Miller GJ: Primary carcinoid tumors of the testicle: a case report and management schema. J Urol 148: 880 – 882, 1992. 4. Debas HT, and Orloff SL: Carcinoid tumors and the carcinoid syndrome, in Sabiston DC Jr, and Lyerly HK (Eds): Textbook of Surgery, 15th ed. Philadelphia, WB Saunders, 1997, pp 950 –954.
UROLOGY 57 (3), 2001