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Painless Hematuria and Urethral Discharge Secondary to Ectopic Prostate
0022-5347/89/1426-1554$02.00/0 Vol. 142, December
THE JOURNAL OF UROLOGY
Copyright © 1989 by AMERICAN UROLOGICAL ASSOCIATION, INC.
Printed in U.S.A.
PAINLESS HEMATURIA AND URETHRAL DISCHARGE SECONDARY TO ECTOPIC PROSTATE L. CONGLETON, W. B. THOMASON, D. T. McMULLAN AND G. F. WORSHAM From the Departments of Urology and Pathology, Medical University of South Carolina and Roper Hospital, Charleston, South Carolina
ABSTRACT
We report a case of ectopic prostatic tissue found in the penile urethra of a 16-year-old boy. A literature review and discussion of this rare entity are presented. (J. Ural., 142: 1554-1555, 1989) Ectopic prostatic epithelium is an uncommon finding with most of the reported cases being found near the verumontanum. I - 14 Even more rare is the appearance of prostatic tissue in the penile urethra, with only 2 previous cases described in the literature. 11, 15 We report a case occurring in an adolescent and review the literature on aberrant prostatic tissue. CASE REPORT
A 16-year-old white boy was referred for recurrent gross hematuria and intermittent, bloody urethral discharge. The physical examination was normal including penile size. Facial and pubic hair was moderately developed and normal for age. Medical history was significant for an episode of toxoplasmosis involving only the eyes. The family history was notable for the father having Bright's disease as a child. All cultures of the urine were negative and excretory urography was normal. Cystourethroscopy revealed a 1 cm. thickened membrane at the penoscrotal junction of the penile urethra. The lesion extended from the 2 o'clock to 7 o'clock positions, and was gray and smooth in appearance. Antibiotics were begun to treat this assumed infectious process, although all cultures remained negative. The hematuria persisted, and computerized tomography of the abdomen and pelvis was normal. Upon repeat cystourethroscopy the lesion was visualized, resected and fulgurated. The patient has had no recurrent episodes. Microscopic examination of the tissue revealed a polypoid lesion composed of fibromuscular stroma surrounding benign glands with a convoluted pattern characteristic of prostate. Columnar and myoepithelial cells lined the glands with an occasional papillary pattern (see figure). Immunoperoxidase staining for prostate specific antigen confirmed that the tissue was prostatic in origin.
was from Butterick and associates, and involved 67 cases found near the verumontanum with 1 at the bladder neck. 4 Interestingly, most of these cases occurred in younger male patients, with 26% found in those 15 to 20 years old. All of these polyps were confirmed to contain prostatic epithelium by acid phosphatase stains. 4 There also have been 6 cases of aberrant prostatic tissue reported in the bulbous urethra,5, 6, 8,12,16,17 13 in the bladder l8-21 and 3 posterior to the bladder. 22-24 In 1982 Walker and associates reported the only case of malignancy found in ectopic prostate tissue. 25 The presenting symptom of ectopic prostatic tissue almost always is gross painless hematuria. 3,4, 6,9,10,14,20 However, symptoms of dysuria, frequency3, 4,10,16 and hematospermia9,10,13 have been described. Endoscopically, these ectopic prostatic lesions appear typically to be polypoid or papillary. The appearance in our case was atypically flat and smooth although 1 case with a similar appearance has been described. 17 Gutierrez and Nesbit proposed 3 possible etiologies for the existence of these lesions: 1) they could represent prostate buds that normally regress in the embryo but are stimulated to grow at puberty, 2) they simply may be misplaced nests of prostatic epithelium and 3) there may be metaplasia of transitional to glandular epithelium resembling prostate. 19 Although an uncommon lesion ectopic prostatic tissue always should be considered in the differential diagnosis of male patients with hematuria. The presence of this lesion in the penile urethra is even more unusual and could easily be overlooked in a cystoscopic evaluation. Treatment of these lesions is resection with fulguration and recu'rrence is rare.5,7
DISCUSSION
The first reported case of prostatic' tissue in the penile urethra was from Walker and associates in 1983 and was seen in combination with prostatic-type polyps in the posterior urethra. l1 A solitary ectopic prostatic lesion in the penile urethra then was described by Dejter and associates in 1988. 15 Both of these cases occurred in men more than 39 years old. We report a case of pendulous urethral prostatic tissue. Its appearance in an adolescent makes it even more unusual. The most common type of aberrant prostatic tissue is found in the posterior urethra with more than 100 cases reported worldwide. Randall reported 4 cases of benign glandular polyps in 19131 and Lazaruz reported a benign papilloma of the prostatic urethra in 1933. 2 In 1962 Nesbit reported 12 prostatic adenomas found near the verumontanum. 3 The largest series
Accepted for publication June 23, 1989.
Photomicrograph of prostatic tissue in penile urethra. Reduced from X400.
1554
1. Randall, A study of benign polyps of the male urethra. Surg., Gynec. & Obst., 548, 1913. 2. Lazaruz, J. A.: Primary tumors of the urethra: report of three cases. U rol. Cutan. 604, 1933. 3. Nesbit, R. M.: The genesis of benign polyps in the prostatic urethra. J. Urol., 87: 416, 1962. 4. Butterick, J. D., Schnitzer, B. and Abell, M. R.: Ectopic prostatic tissue in urethra: a clinico-pathological entity and a significant cause of hematuria. J. Urol., 105: 97, 1971. 5. Mostofi, E. K. and Price, E. B., Jr.: Tumors of the male genital system. In: Atlas of Tumor Pathology. Washington, D. C.: Armed Forces Institute of Pathology, 2nd series, fasc. 8, p. 265, 1973. 6. Craig, J. R. and Hart, W. R.: Benign polyps with prostatic-type epithelium of the urethra. Amer. J. Clin. Path., 63: 343, 1975. 7. Mori, K., Spiro, L. H., Hecht, H. and Orkin, L. A.: Recurrent intraurethral proliferation of ectopic prostatic tissue associated with hematuria. J. Urol., 114: 316, 1975. 8. Murad, T., Robinson, L. H. and Bueschen, A. J.: Villous polyps of the urethra: a report of two cases. Hum. Path., 10: 478,1979. 9. Stein, A. J., Prioleau, P. G. and Catalona, W. J.: Adenomatous polyps of the prostatic urethra: a cause of hematospermia. J. Urol., 124: 298, 1980. 10. Goldstein, A. M. B., Bragin, S. D., Terry, R. and Yoell, J. H.: Prostatic urethral polyps in adults: histopathologic variations and clinical manifestations. J. Urol., 126: 129, 1981. 11. Walker, A. N., Mills, S. E., Fechner, R. E. and Perry, J. M.: Epithelial polyps of the prostatic urethra. A light-microscopic and immunohistochemical study. Amer. J. Surg. Path., 351, 1983. 12. Remick, D. G., Jr. and Kumar, N. B.: Benign polyps with prC)stcltIC type eipthelium of the urethra and the urinary A sug-
L:ll't~HH.L:i::U studies. 13. Fan, K., Schaefer, evidence 14. S., Lurie, urethra. 15. Dejter, S. W., Zuckerman, E. and Lynch, J. villous with prostatic type epithelium of the penile J. 139: 590, 1988. 16. Hicks, C. Nicholas, E. M. and Morgan, J. W.: Hematuria from ectopic prostatic tissue in bulbous urethra. Urology, 50, 1977. 17. Heyderman, E., Kadow C., Mandaliya, K. Bultitude, M. I. and O'Donnell, P. J.: Ectopic prostatic in bulbar urethra immunoperoxidase study. Urology, 76, 1987. 18. Goodale, R. H.: Cystadenoma of the bladder from aberrant prostatic gland. Arch. Path., 6: 210, 1928. 19. Gutierrez, J. and Nesbit, R. M.: Ectopic prostatic tissue in bladder. J. Urol., 98: 474, 1967. 20. Klein, H. Z. and Rosenberg, M. L.: Ectopic prostatic tissue in bladder trigone: distinctive cause of hematuria. Urology, 81, 1984. 21. Ewing, R., Harnden-Mayor, P., Mason, M. K. and Anderson, C. K.: Extra-urethral ectopic prostate. Brit. J. Urol., 60: 433, 1987. 22. Gledhill, A.: Ectopic prostatic tissue. J. Urol., 133: 110, 1985. 23. Spiro, L. H. and Levine, B.: Ectopic subvesical prostatic tissue. J. Urol., 631, 1974. 24. Yasukawa, S., Aoshi, H. and Takamatsu, M.: Ectopic prostatic adenoma in retrovesical space. Urol., 998, 1987. E. and J. M.: 25. Walker, A. N., Mills, S. E., Fechner, "Endometrial" adenocarcinoma of the prostatic arISIng in a villous A microscopic and immunoperoxidase study. Arch. 624, 1982.
THE JOURNAL OF UROLOGY
Copyright © 1989 by AMERICAN UROLOGICAL ASSOCIATION, INC.
Printed in U.S.A.
PAINLESS HEMATURIA AND URETHRAL DISCHARGE SECONDARY TO ECTOPIC PROSTATE L. CONGLETON, W. B. THOMASON, D. T. McMULLAN AND G. F. WORSHAM From the Departments of Urology and Pathology, Medical University of South Carolina and Roper Hospital, Charleston, South Carolina
ABSTRACT
We report a case of ectopic prostatic tissue found in the penile urethra of a 16-year-old boy. A literature review and discussion of this rare entity are presented. (J. Ural., 142: 1554-1555, 1989) Ectopic prostatic epithelium is an uncommon finding with most of the reported cases being found near the verumontanum. I - 14 Even more rare is the appearance of prostatic tissue in the penile urethra, with only 2 previous cases described in the literature. 11, 15 We report a case occurring in an adolescent and review the literature on aberrant prostatic tissue. CASE REPORT
A 16-year-old white boy was referred for recurrent gross hematuria and intermittent, bloody urethral discharge. The physical examination was normal including penile size. Facial and pubic hair was moderately developed and normal for age. Medical history was significant for an episode of toxoplasmosis involving only the eyes. The family history was notable for the father having Bright's disease as a child. All cultures of the urine were negative and excretory urography was normal. Cystourethroscopy revealed a 1 cm. thickened membrane at the penoscrotal junction of the penile urethra. The lesion extended from the 2 o'clock to 7 o'clock positions, and was gray and smooth in appearance. Antibiotics were begun to treat this assumed infectious process, although all cultures remained negative. The hematuria persisted, and computerized tomography of the abdomen and pelvis was normal. Upon repeat cystourethroscopy the lesion was visualized, resected and fulgurated. The patient has had no recurrent episodes. Microscopic examination of the tissue revealed a polypoid lesion composed of fibromuscular stroma surrounding benign glands with a convoluted pattern characteristic of prostate. Columnar and myoepithelial cells lined the glands with an occasional papillary pattern (see figure). Immunoperoxidase staining for prostate specific antigen confirmed that the tissue was prostatic in origin.
was from Butterick and associates, and involved 67 cases found near the verumontanum with 1 at the bladder neck. 4 Interestingly, most of these cases occurred in younger male patients, with 26% found in those 15 to 20 years old. All of these polyps were confirmed to contain prostatic epithelium by acid phosphatase stains. 4 There also have been 6 cases of aberrant prostatic tissue reported in the bulbous urethra,5, 6, 8,12,16,17 13 in the bladder l8-21 and 3 posterior to the bladder. 22-24 In 1982 Walker and associates reported the only case of malignancy found in ectopic prostate tissue. 25 The presenting symptom of ectopic prostatic tissue almost always is gross painless hematuria. 3,4, 6,9,10,14,20 However, symptoms of dysuria, frequency3, 4,10,16 and hematospermia9,10,13 have been described. Endoscopically, these ectopic prostatic lesions appear typically to be polypoid or papillary. The appearance in our case was atypically flat and smooth although 1 case with a similar appearance has been described. 17 Gutierrez and Nesbit proposed 3 possible etiologies for the existence of these lesions: 1) they could represent prostate buds that normally regress in the embryo but are stimulated to grow at puberty, 2) they simply may be misplaced nests of prostatic epithelium and 3) there may be metaplasia of transitional to glandular epithelium resembling prostate. 19 Although an uncommon lesion ectopic prostatic tissue always should be considered in the differential diagnosis of male patients with hematuria. The presence of this lesion in the penile urethra is even more unusual and could easily be overlooked in a cystoscopic evaluation. Treatment of these lesions is resection with fulguration and recu'rrence is rare.5,7
DISCUSSION
The first reported case of prostatic' tissue in the penile urethra was from Walker and associates in 1983 and was seen in combination with prostatic-type polyps in the posterior urethra. l1 A solitary ectopic prostatic lesion in the penile urethra then was described by Dejter and associates in 1988. 15 Both of these cases occurred in men more than 39 years old. We report a case of pendulous urethral prostatic tissue. Its appearance in an adolescent makes it even more unusual. The most common type of aberrant prostatic tissue is found in the posterior urethra with more than 100 cases reported worldwide. Randall reported 4 cases of benign glandular polyps in 19131 and Lazaruz reported a benign papilloma of the prostatic urethra in 1933. 2 In 1962 Nesbit reported 12 prostatic adenomas found near the verumontanum. 3 The largest series
Accepted for publication June 23, 1989.
Photomicrograph of prostatic tissue in penile urethra. Reduced from X400.
1554
1. Randall, A study of benign polyps of the male urethra. Surg., Gynec. & Obst., 548, 1913. 2. Lazaruz, J. A.: Primary tumors of the urethra: report of three cases. U rol. Cutan. 604, 1933. 3. Nesbit, R. M.: The genesis of benign polyps in the prostatic urethra. J. Urol., 87: 416, 1962. 4. Butterick, J. D., Schnitzer, B. and Abell, M. R.: Ectopic prostatic tissue in urethra: a clinico-pathological entity and a significant cause of hematuria. J. Urol., 105: 97, 1971. 5. Mostofi, E. K. and Price, E. B., Jr.: Tumors of the male genital system. In: Atlas of Tumor Pathology. Washington, D. C.: Armed Forces Institute of Pathology, 2nd series, fasc. 8, p. 265, 1973. 6. Craig, J. R. and Hart, W. R.: Benign polyps with prostatic-type epithelium of the urethra. Amer. J. Clin. Path., 63: 343, 1975. 7. Mori, K., Spiro, L. H., Hecht, H. and Orkin, L. A.: Recurrent intraurethral proliferation of ectopic prostatic tissue associated with hematuria. J. Urol., 114: 316, 1975. 8. Murad, T., Robinson, L. H. and Bueschen, A. J.: Villous polyps of the urethra: a report of two cases. Hum. Path., 10: 478,1979. 9. Stein, A. J., Prioleau, P. G. and Catalona, W. J.: Adenomatous polyps of the prostatic urethra: a cause of hematospermia. J. Urol., 124: 298, 1980. 10. Goldstein, A. M. B., Bragin, S. D., Terry, R. and Yoell, J. H.: Prostatic urethral polyps in adults: histopathologic variations and clinical manifestations. J. Urol., 126: 129, 1981. 11. Walker, A. N., Mills, S. E., Fechner, R. E. and Perry, J. M.: Epithelial polyps of the prostatic urethra. A light-microscopic and immunohistochemical study. Amer. J. Surg. Path., 351, 1983. 12. Remick, D. G., Jr. and Kumar, N. B.: Benign polyps with prC)stcltIC type eipthelium of the urethra and the urinary A sug-
L:ll't~HH.L:i::U studies. 13. Fan, K., Schaefer, evidence 14. S., Lurie, urethra. 15. Dejter, S. W., Zuckerman, E. and Lynch, J. villous with prostatic type epithelium of the penile J. 139: 590, 1988. 16. Hicks, C. Nicholas, E. M. and Morgan, J. W.: Hematuria from ectopic prostatic tissue in bulbous urethra. Urology, 50, 1977. 17. Heyderman, E., Kadow C., Mandaliya, K. Bultitude, M. I. and O'Donnell, P. J.: Ectopic prostatic in bulbar urethra immunoperoxidase study. Urology, 76, 1987. 18. Goodale, R. H.: Cystadenoma of the bladder from aberrant prostatic gland. Arch. Path., 6: 210, 1928. 19. Gutierrez, J. and Nesbit, R. M.: Ectopic prostatic tissue in bladder. J. Urol., 98: 474, 1967. 20. Klein, H. Z. and Rosenberg, M. L.: Ectopic prostatic tissue in bladder trigone: distinctive cause of hematuria. Urology, 81, 1984. 21. Ewing, R., Harnden-Mayor, P., Mason, M. K. and Anderson, C. K.: Extra-urethral ectopic prostate. Brit. J. Urol., 60: 433, 1987. 22. Gledhill, A.: Ectopic prostatic tissue. J. Urol., 133: 110, 1985. 23. Spiro, L. H. and Levine, B.: Ectopic subvesical prostatic tissue. J. Urol., 631, 1974. 24. Yasukawa, S., Aoshi, H. and Takamatsu, M.: Ectopic prostatic adenoma in retrovesical space. Urol., 998, 1987. E. and J. M.: 25. Walker, A. N., Mills, S. E., Fechner, "Endometrial" adenocarcinoma of the prostatic arISIng in a villous A microscopic and immunoperoxidase study. Arch. 624, 1982.