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LEYDIG CELL TUMORS PRESENTING AS AZOOSPERMIA
0022-5347/99/1615-1571/0 THEJOURNAL OF UROLOGY Copyright 0 1999 by AMERICAN UROLOGICAL ASSOCIATION, INC.
Vol. 161,1571-1572,May 1999 Printed in U.S.A.
LEYDIG CELL TUMORS PRESENTING AS AZOOSPERMIA MARK L. FALLICK,* WILLIAM W. LIN AND LARRY I. LIPSHULTZ From the Scott Department of Urology, Baylor College of Medicine, Houston, Texas
KEY WORDS:oligospermia, Leydig cell tumor, testis, testicular neoplasms Leydig cell tumors of the testis are rare, comprising 1to 3% of all testicular neoplasms.l.2 Approximately 10% of these tumors are malignant. They most commonly present as a testicular mass or with endocrine abnormalities, such as gynecomastia and loss of libido. We report on 2 patients with Leydig cell tumors who presented with the unusual symptoms of infertility and azoospermia. CASE REPORTS
Case 1. A 33-year-old man presented for evaluation of primary infertility 3 to 4 years in duration. History included a left maldescended testicle which spontaneously descended at age 2 years and prepubertal mumps without orchitis. Laboratory tests included 3 semen analyses showing azoospermia and pellet analysis confirming absence of sperm. Serum testosterone was 386 ng./dl. (normal 270 to 1,070), and serum a-fetoprotein, p-human chorionic gonadoFIG. 1. Microscopic view of testicular tissue in area away from tropin and lactic dehydrogenase were normal. Physical ex- tumor shows seminiferous tubules with Sertoli-cell-only and promiamination revealed a left testis measuring approximately 25 nent Leydig cells. H & E, reduced from x 100. cc with a large mass in the upper portion. The right testis was approximately 10 cc and soft. Ultrasonography confirmed a hyperechoic mass in the upper two-thirds of the left testis. Given the appearance of the mass, left radical orchiectomy was performed. Pathological examination revealed a Leydig cell tumor (figs. 1 and 2). Metastatic evaluation with abdominal and pelvic computerized tomography was negative. Case 2. A 32-year-old man presented for evaluation of 2.5 years of primary infertility. History included bilateral inguinal hernia repairs. Physical examination revealed bilateral severe testicular atrophy. Two semen analyses showed fructose positive azoospermia. Serum testosterone was normal but follicle-stimulating hormone (1,567 mlU/ml., normal 4 to 10) and luteinizing hormone (156 mlU/ml., normal 2 to 10) were elevated. Diagnostic biopsy of the right testis revealed late maturation arrest. The patient returned for additional evaluation 5 years later and a nodule was palpated in the left testis, which was FIG.2. Area of tumor shows sheets of Leydi cells, prominent confirmed to be solid on ultrasound. Serum hormones were and absence of seminiferous tubules. H E, reduced from without significant change from previous levels and serum stroma X400. tumor markers were negative. Left radical orchiectomy was performed, and pathological evaluation revealed Leydig cell tumor. Metastatic evaluation was negative. The patient was lost to followup for 9 years, when he returned for additional crine manifestations, azoospermia is not usually reported as infertility treatment. At that time, an 8 mm. mass was pal- a symptom of this tumor. In fact, 1study evaluating the effect pated in the solitary right testis. Despite ultrasound confir- of testicular tumors on spermatogenesis demonstrated sigmation of the mass, the patient has refused any intervention. nificant impairment in patients with malignant nongerm cell tumors but normal spermatogenesis in all men with Leydig cell tumors.3 DISCUSSION Both of our patients presented with azoospermia. This Leydig cell tumors are rare testicular neoplasms that most unusual manifestation of Leydig cell tumors is not typically commonly present as an asymptomatic mass or with mani- described for this lesion. In addition, these cases reinforce the festations of excessive testosterone or estrogen production.1.2 importance of urological evaluation of the infertile man. In They are malignant in approximately 10% of cases based on the era of intracytoplasmic sperm injection infertile couples the presence of metastases. Although they may have endo- with azoospermia may be successfully treated with testicular sperm extraction combined with intracytoplasmic sperm inAccepted for publication December 11, 1998. The presence of testicular tumors in these 2 patients * Current address: Center for Urologic Care, 100 Carnie Blvd., jection. reinforces the need for careful evaluation of the male partner Suite A-3, Voorhees, New Jersey, 08043.
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TESTICULAR LEYDIG CELL TUMORS PRESENTING AS AZOOSPERMIA
in an infertile couple before any sperm retrieval procedures are done. REFERENCES
1. Deshmukh, A. S.: Leydig cell tumor in undescended testis. Urology, 2 0 543, 1982.
2. Feldman, P. S., Kovacs, K., Horvath, E. and Adelson, G . L.: Malignant Leydig cell tumor: clinical, histologic, and electron microscopic features. Cancer, 49: 714, 1982. 3. Ho, G. T., Gardner, H.. Mostofi, K., DeWolf, W. C., Loughlin, K. R. and Morgentaler. A,: The effect of testicular nongerm cell tumors on local spermatogenesis. Fcrtil. Steril.. 62: 162, 1994.
Vol. 161,1571-1572,May 1999 Printed in U.S.A.
LEYDIG CELL TUMORS PRESENTING AS AZOOSPERMIA MARK L. FALLICK,* WILLIAM W. LIN AND LARRY I. LIPSHULTZ From the Scott Department of Urology, Baylor College of Medicine, Houston, Texas
KEY WORDS:oligospermia, Leydig cell tumor, testis, testicular neoplasms Leydig cell tumors of the testis are rare, comprising 1to 3% of all testicular neoplasms.l.2 Approximately 10% of these tumors are malignant. They most commonly present as a testicular mass or with endocrine abnormalities, such as gynecomastia and loss of libido. We report on 2 patients with Leydig cell tumors who presented with the unusual symptoms of infertility and azoospermia. CASE REPORTS
Case 1. A 33-year-old man presented for evaluation of primary infertility 3 to 4 years in duration. History included a left maldescended testicle which spontaneously descended at age 2 years and prepubertal mumps without orchitis. Laboratory tests included 3 semen analyses showing azoospermia and pellet analysis confirming absence of sperm. Serum testosterone was 386 ng./dl. (normal 270 to 1,070), and serum a-fetoprotein, p-human chorionic gonadoFIG. 1. Microscopic view of testicular tissue in area away from tropin and lactic dehydrogenase were normal. Physical ex- tumor shows seminiferous tubules with Sertoli-cell-only and promiamination revealed a left testis measuring approximately 25 nent Leydig cells. H & E, reduced from x 100. cc with a large mass in the upper portion. The right testis was approximately 10 cc and soft. Ultrasonography confirmed a hyperechoic mass in the upper two-thirds of the left testis. Given the appearance of the mass, left radical orchiectomy was performed. Pathological examination revealed a Leydig cell tumor (figs. 1 and 2). Metastatic evaluation with abdominal and pelvic computerized tomography was negative. Case 2. A 32-year-old man presented for evaluation of 2.5 years of primary infertility. History included bilateral inguinal hernia repairs. Physical examination revealed bilateral severe testicular atrophy. Two semen analyses showed fructose positive azoospermia. Serum testosterone was normal but follicle-stimulating hormone (1,567 mlU/ml., normal 4 to 10) and luteinizing hormone (156 mlU/ml., normal 2 to 10) were elevated. Diagnostic biopsy of the right testis revealed late maturation arrest. The patient returned for additional evaluation 5 years later and a nodule was palpated in the left testis, which was FIG.2. Area of tumor shows sheets of Leydi cells, prominent confirmed to be solid on ultrasound. Serum hormones were and absence of seminiferous tubules. H E, reduced from without significant change from previous levels and serum stroma X400. tumor markers were negative. Left radical orchiectomy was performed, and pathological evaluation revealed Leydig cell tumor. Metastatic evaluation was negative. The patient was lost to followup for 9 years, when he returned for additional crine manifestations, azoospermia is not usually reported as infertility treatment. At that time, an 8 mm. mass was pal- a symptom of this tumor. In fact, 1study evaluating the effect pated in the solitary right testis. Despite ultrasound confir- of testicular tumors on spermatogenesis demonstrated sigmation of the mass, the patient has refused any intervention. nificant impairment in patients with malignant nongerm cell tumors but normal spermatogenesis in all men with Leydig cell tumors.3 DISCUSSION Both of our patients presented with azoospermia. This Leydig cell tumors are rare testicular neoplasms that most unusual manifestation of Leydig cell tumors is not typically commonly present as an asymptomatic mass or with mani- described for this lesion. In addition, these cases reinforce the festations of excessive testosterone or estrogen production.1.2 importance of urological evaluation of the infertile man. In They are malignant in approximately 10% of cases based on the era of intracytoplasmic sperm injection infertile couples the presence of metastases. Although they may have endo- with azoospermia may be successfully treated with testicular sperm extraction combined with intracytoplasmic sperm inAccepted for publication December 11, 1998. The presence of testicular tumors in these 2 patients * Current address: Center for Urologic Care, 100 Carnie Blvd., jection. reinforces the need for careful evaluation of the male partner Suite A-3, Voorhees, New Jersey, 08043.
f
1571
1572
TESTICULAR LEYDIG CELL TUMORS PRESENTING AS AZOOSPERMIA
in an infertile couple before any sperm retrieval procedures are done. REFERENCES
1. Deshmukh, A. S.: Leydig cell tumor in undescended testis. Urology, 2 0 543, 1982.
2. Feldman, P. S., Kovacs, K., Horvath, E. and Adelson, G . L.: Malignant Leydig cell tumor: clinical, histologic, and electron microscopic features. Cancer, 49: 714, 1982. 3. Ho, G. T., Gardner, H.. Mostofi, K., DeWolf, W. C., Loughlin, K. R. and Morgentaler. A,: The effect of testicular nongerm cell tumors on local spermatogenesis. Fcrtil. Steril.. 62: 162, 1994.