THE JOURNAL
Vol. 114, August
OF UROLOGY
Printed in U.S.A.
Copyright © 1975 by The Williams & Wilkins Co.
RECURRENT INTRAURETHRAL PROLIFERATION OF ECTOPIC PROSTATIC TISSUE ASSOCIATED WITH HEMATURIA KEN MORI, LAURENCE H. SPIRO,* HUBERT HECHT AND LAZARUS A. ORKIN
From the Departments of Pathology and Urology, Beth Israel Medical Center and the Mt. Sinai School of Medicine, New York, New York
ABSTRACT
The first reported case of recurrent intraurethral proliferation of ectopic prostatic tissue associated with hematuria is presented. physical examination the prostate gland was described as 1 plus, non-tender and smooth. Multiple urinalyses revealed many erythrocytes but no leukocytes. Multiple urine cultures were negative. An excretory urogram was within normal limits. A retrograde urethrogram was normal. Cystoscopy revealed a normal bladder and clear efflux was seen from both orifices. Panendoscopy showed a hyperemic verumontanum. The prostatic utricle was gaping and papillary tissue presented at the utricular orifice. The papillary tissue was biopsied, resected and its base was fulgurated. The hematuria ceased and the patient was subsequently discharged from the hospital. The biopsied tissue, received in formalin, measured 0.5 cm. in aggregate. Microscopic examination revealed orderly tubular glands of equal size that showed frequent intraluminal folds with delicate connective tissue cores (fig. 1). The epithelial cells were columnar, devoid of cilia and occasionally pseudostratified. Small, dark nuclei were usually located near the thin basement membrane. The cytoplasm was pale and finely granular in hematoxylin and eosin staining (fig. 1, B). Some granules were stained by periodic acid, Schiff (PAS) before and after diastase treatment. 5 Basophilic PAS positive intracytoplasmic bodies, described by Butterick and associates, and corpora amylacea were absent. The intervening loose stroma contained varying amounts of smooth muscle. No inflammatory leukocytic infiltration was found. In areas, the scalloping epithelium ap- · peared to be on the urethral surface instead of facing the glandular lumen. The biopsy was diagnosed as prostatic tissue. The patient was followed for 3 months after CASE REPORT discharge from the hospital and was free of all E. K., 303035, a 16-year-old white boy, was hos- symptoms. However, he was rehospitalized 7 pitalized complaining of dysuria and gross, painless months later because of recurrent, terminal painterminal hematuria 18 months in duration. The less hematuria. Again the prostate gland was patient denied having hematospermia, nocturia, described as 1 plus, non-tender and smooth. All frequency, burning on urination, excessive mastur- laboratory data were normal except for repeated bation, anal intercourse or urethral discharge. On urinalyses showing many erythrocytes. Panendoscopy revealed a recurrent papillary growth at the Accepted for publication December 6, 1974. *Requests for reprints: Department of Urology, Beth utricular orifice. The growth was resected at its base. Care was taken not to injure the ejaculatory Israel Medical Center, New York, New York 10003.
In 1913 Randall divided benign polyps of the prostatic urethra into fibrous, villous and glandular types. 1 He postulated that the glandular type might have arisen from a tuft of aberrant prostatic tissue as a result of early prostatic hypertrophy. Similarly, Stueber and Persky classified urethral polyps into 3 categories: 1) inflammatory pseudopolyp, 2) solid polyp of children and 3) ectopic prostatic tissue.2 Cases of Randall's glandular type and of the third category of Stueber and Persky's classification were further studied by Nesbit in 1962 3 and more extensively by Butterick and associates in 1971. • Nesbit reported on 12 patients with aberrant prostatic tissue in the prostatic urethra. In 2 of his patients the prostatic tissue was described as arising from the verumontanum. All except 1 of these patients presented with either gross or microscopic hematuria. Butterick and associates reported on 68 new cases of ectopic prostatic tissue in the prostatic urethra. The majority of these patients were 16 to 35 years old and 63 of them presented with hematuria. In 27 of these patients the lesions were described as arising from the verumontanum. The most frequently used terms to describe the gross appearance of the lesions in situ were frond-like, filiform, polyp, mass or papillomatous. We recently encountered a 16-year-old boy who had recurrent episodes of gross hematuria. On both occasions a papillary growth consisting of prostatic tissue was found arising from the prostatic utricle, and was excised and fulgurated with immediate disappearance of the hematuria. Recurrence of the rare lesion prompted this report.
316
INTRAURETHRAL PROLIFERATION OF PROSTATIC TISSUE ASSOCIATED WITH HEMATURIA
317
FIG, L A, representative fragment of first biopsy, Glands in left upper and right lower portions suggest branching, Same histological pattern prevailed in second biopsy, H & E, reduced from x40, B, cross section of glands, Note epithelial folds, small nuclei and pale cytoplasm of epithelial cells, indistinct basal lamina and loose stroma, Smooth muscle is absent in this field, H & E, reduced from x200,
ducts, The hematuria ceased and the patient was discharged from hospitaL He has been followed fm 8 months and has been free of all symptoms, The tissue removed measured less than 0,5 cm, in aggregate, Histological examination disclosed structures identical to the first except for an abundance of smooth muscle, In a flat transitional of the DISCUSSION
In the most turia in young male established the lesion as Our case shows a s}rr,i1ar
to date But-
en-
because it appears to be the first case of recurrence of the lesion and hematuria trans urethral
nant transformation", heavily towards an opmental
incriminating a develas indicated by their title,' excision of aberrant tissue may result in local recurrence, it would be to examine the µv00,u,u of neoVvhile intraurethral carcmomas presumducts or urethral have been rP,~rrrt••rl in the past, •-a of the utricle have been rare, In 1971 Melicow and Tannenbaum collected 6 cases of adenocarcinorna at the utricle and coined a term endometrial carcinoma of uterus masculinus since the ,,:-;~ic~e consider2d to be a in normal men. 9 In 1973 1nlillerian. duct
318
MORI AND ASSOCIATES
Controversy over anatomical and embryological boundaries of the prostate poses a semantic problem for the lesion. For example, McNeal emphasized a sharp distinction of the urethral glands from the prostate gland and proposed a new term, benign periurethral hyperplasia, instead of the traditional benign prostatic hyperplasia. 14 In this context some of the cases reported by Butterick and associates may be reclassified as hyperplasia of the periurethral glands since extent and location of the abnormalities in the series were not described individually or summarized in terms of topographical distribution. In our report of a utricular lesion we purposely selected a descriptive designation, intra urethral proliferation of prostatic tissue, rather than an interpretive term such as ectopic prostatic tissue. Undoubtedly, the lesion described herein should be seriously considered as a cause of hematuria in young male subjects despite its obscure etiology and semantic ambiguity.
REFERENCES
Fm. 2. Surface fragment of second biopsy. Upper portion of tissue is partially covered by transitional epithelium. In lower portion glandular epithelium appears to be continuous with surface linings. Abundant smooth muscle is seen on left side. Ill-defined dark patches in subepithelial zone are foci of hemorrhage. H & E, reduced from x 40.
Young and Lagios added another case. 10 In our case the histological and cytological characteristics of biopsied tissues were so consistent with the configuration of non-neoplastic prostatic tissue that the possibility of neoplasm or mullerian duct vestige appeared extremely remote. An inflammatory pathogenesis should be considered since a minority of patients reviewed by Butterick and others presented with urinary infection. While inflammatory reaction of the urethral tissue may develop a papillary growth (proliferative papillary urethritis), 11 the predominant tissue element in the pseudotumor is connective tissue, not the glandular structures so abundant in our biopsy material. Furthermore, histological absence of inflammatory reaction as well as clinical absence of urinary infection in our patient makes it unlikely that the abnormality was of an inflammatory or infectious origin. Likewise, cellular constituents of the urethral tunica propria, not of the glands, are overwhelmingly present in fibrous polyps of the congenital type. 2 • 12 • 13 These urethral polyps differ from our patient in many other aspects since they are usually solid, pedunculated and associated with urinary obstruction in children. In short, the lesion reported herein appears to be best described as a clinicopathological entity of undetermined etiology.
1. Randall, A.: A study of the benign polyps of the male urethra. Surg., Gynec. & Obst., 17: 548, 1913. 2. Stueber, P. J. and Persky, L.: Solid tumors of the urethra and bladder neck. J. Urol., 102: 205, 1969. 3. Nesbit, R. M.: The genesis of benign polyps in the prostatic urethra. J. Urol., 87: 416, 1962. 4. Butterick, J. A., Schnitzer, B. and Abell, M. R.: Ectopic prostatic tissue in urethra: a clinicopathological entity and a significant cause of hematuria. J. Urol., 105: 97, 1971. 5. Brandes, D. and Bourne, G. H.: Histochemistry of the human prostate: normal and neoplastic. J. Path. Bact., 71: 33, 1956. 6. Dube, V. E., Farrow, G. M. and Greene, L. F.: Prostatic adenocarcinoma of ductal origin. Cancer, 32: 402, 1973. 7. Ende, N., Woods, L. P. and Shelley, H. S.: Carcinoma originating in ducts surrounding the prostatic urethra. Amer. J. Clin. Path., 40: 183, 1963. 8. Grabstald, H.: Tumors of the urethra in men and women. Cancer, 32: 1236, 1973. 9. Melicow, M. M. and Tannenbaum, M.: Endometrial carcinoma of uterus masculinus (prostatic utricle). Report of 6 cases. J. Urol., 106: 892, 1971. 10. Young, B. W. and Lagios, M. D.: Endometrial (papillary) carcinoma of prostatic utricle-response to orchiectomy, a case report. Cancer, 32: 1293, 1973. 11. Schinella, R., Thurm, J. and Feiner, M.: Papillary pseudotumor of the prostatic urethra: proliferative papillary urethritis. J. Urol., 111: 38, 1974. 12. Downs, R. A.: Congenital polyps of prostatic urethra. A review of the literature and report of two cases. Brit. J. Urol., 42: 76, 1970. 13. Kippusami, M. D. and Moors, D. E.: Fibrous polyp of verumontanum. Canad. J. Surg., 11: 388, 1968. 14. McNeal, J. E.: The prostate and prostatic urethra: a morphological synthesis. J. Urol., 107: 1008, 1972.
Vol. 114, August
OF UROLOGY
Printed in U.S.A.
Copyright © 1975 by The Williams & Wilkins Co.
RECURRENT INTRAURETHRAL PROLIFERATION OF ECTOPIC PROSTATIC TISSUE ASSOCIATED WITH HEMATURIA KEN MORI, LAURENCE H. SPIRO,* HUBERT HECHT AND LAZARUS A. ORKIN
From the Departments of Pathology and Urology, Beth Israel Medical Center and the Mt. Sinai School of Medicine, New York, New York
ABSTRACT
The first reported case of recurrent intraurethral proliferation of ectopic prostatic tissue associated with hematuria is presented. physical examination the prostate gland was described as 1 plus, non-tender and smooth. Multiple urinalyses revealed many erythrocytes but no leukocytes. Multiple urine cultures were negative. An excretory urogram was within normal limits. A retrograde urethrogram was normal. Cystoscopy revealed a normal bladder and clear efflux was seen from both orifices. Panendoscopy showed a hyperemic verumontanum. The prostatic utricle was gaping and papillary tissue presented at the utricular orifice. The papillary tissue was biopsied, resected and its base was fulgurated. The hematuria ceased and the patient was subsequently discharged from the hospital. The biopsied tissue, received in formalin, measured 0.5 cm. in aggregate. Microscopic examination revealed orderly tubular glands of equal size that showed frequent intraluminal folds with delicate connective tissue cores (fig. 1). The epithelial cells were columnar, devoid of cilia and occasionally pseudostratified. Small, dark nuclei were usually located near the thin basement membrane. The cytoplasm was pale and finely granular in hematoxylin and eosin staining (fig. 1, B). Some granules were stained by periodic acid, Schiff (PAS) before and after diastase treatment. 5 Basophilic PAS positive intracytoplasmic bodies, described by Butterick and associates, and corpora amylacea were absent. The intervening loose stroma contained varying amounts of smooth muscle. No inflammatory leukocytic infiltration was found. In areas, the scalloping epithelium ap- · peared to be on the urethral surface instead of facing the glandular lumen. The biopsy was diagnosed as prostatic tissue. The patient was followed for 3 months after CASE REPORT discharge from the hospital and was free of all E. K., 303035, a 16-year-old white boy, was hos- symptoms. However, he was rehospitalized 7 pitalized complaining of dysuria and gross, painless months later because of recurrent, terminal painterminal hematuria 18 months in duration. The less hematuria. Again the prostate gland was patient denied having hematospermia, nocturia, described as 1 plus, non-tender and smooth. All frequency, burning on urination, excessive mastur- laboratory data were normal except for repeated bation, anal intercourse or urethral discharge. On urinalyses showing many erythrocytes. Panendoscopy revealed a recurrent papillary growth at the Accepted for publication December 6, 1974. *Requests for reprints: Department of Urology, Beth utricular orifice. The growth was resected at its base. Care was taken not to injure the ejaculatory Israel Medical Center, New York, New York 10003.
In 1913 Randall divided benign polyps of the prostatic urethra into fibrous, villous and glandular types. 1 He postulated that the glandular type might have arisen from a tuft of aberrant prostatic tissue as a result of early prostatic hypertrophy. Similarly, Stueber and Persky classified urethral polyps into 3 categories: 1) inflammatory pseudopolyp, 2) solid polyp of children and 3) ectopic prostatic tissue.2 Cases of Randall's glandular type and of the third category of Stueber and Persky's classification were further studied by Nesbit in 1962 3 and more extensively by Butterick and associates in 1971. • Nesbit reported on 12 patients with aberrant prostatic tissue in the prostatic urethra. In 2 of his patients the prostatic tissue was described as arising from the verumontanum. All except 1 of these patients presented with either gross or microscopic hematuria. Butterick and associates reported on 68 new cases of ectopic prostatic tissue in the prostatic urethra. The majority of these patients were 16 to 35 years old and 63 of them presented with hematuria. In 27 of these patients the lesions were described as arising from the verumontanum. The most frequently used terms to describe the gross appearance of the lesions in situ were frond-like, filiform, polyp, mass or papillomatous. We recently encountered a 16-year-old boy who had recurrent episodes of gross hematuria. On both occasions a papillary growth consisting of prostatic tissue was found arising from the prostatic utricle, and was excised and fulgurated with immediate disappearance of the hematuria. Recurrence of the rare lesion prompted this report.
316
INTRAURETHRAL PROLIFERATION OF PROSTATIC TISSUE ASSOCIATED WITH HEMATURIA
317
FIG, L A, representative fragment of first biopsy, Glands in left upper and right lower portions suggest branching, Same histological pattern prevailed in second biopsy, H & E, reduced from x40, B, cross section of glands, Note epithelial folds, small nuclei and pale cytoplasm of epithelial cells, indistinct basal lamina and loose stroma, Smooth muscle is absent in this field, H & E, reduced from x200,
ducts, The hematuria ceased and the patient was discharged from hospitaL He has been followed fm 8 months and has been free of all symptoms, The tissue removed measured less than 0,5 cm, in aggregate, Histological examination disclosed structures identical to the first except for an abundance of smooth muscle, In a flat transitional of the DISCUSSION
In the most turia in young male established the lesion as Our case shows a s}rr,i1ar
to date But-
en-
because it appears to be the first case of recurrence of the lesion and hematuria trans urethral
nant transformation", heavily towards an opmental
incriminating a develas indicated by their title,' excision of aberrant tissue may result in local recurrence, it would be to examine the µv00,u,u of neoVvhile intraurethral carcmomas presumducts or urethral have been rP,~rrrt••rl in the past, •-a of the utricle have been rare, In 1971 Melicow and Tannenbaum collected 6 cases of adenocarcinorna at the utricle and coined a term endometrial carcinoma of uterus masculinus since the ,,:-;~ic~e consider2d to be a in normal men. 9 In 1973 1nlillerian. duct
318
MORI AND ASSOCIATES
Controversy over anatomical and embryological boundaries of the prostate poses a semantic problem for the lesion. For example, McNeal emphasized a sharp distinction of the urethral glands from the prostate gland and proposed a new term, benign periurethral hyperplasia, instead of the traditional benign prostatic hyperplasia. 14 In this context some of the cases reported by Butterick and associates may be reclassified as hyperplasia of the periurethral glands since extent and location of the abnormalities in the series were not described individually or summarized in terms of topographical distribution. In our report of a utricular lesion we purposely selected a descriptive designation, intra urethral proliferation of prostatic tissue, rather than an interpretive term such as ectopic prostatic tissue. Undoubtedly, the lesion described herein should be seriously considered as a cause of hematuria in young male subjects despite its obscure etiology and semantic ambiguity.
REFERENCES
Fm. 2. Surface fragment of second biopsy. Upper portion of tissue is partially covered by transitional epithelium. In lower portion glandular epithelium appears to be continuous with surface linings. Abundant smooth muscle is seen on left side. Ill-defined dark patches in subepithelial zone are foci of hemorrhage. H & E, reduced from x 40.
Young and Lagios added another case. 10 In our case the histological and cytological characteristics of biopsied tissues were so consistent with the configuration of non-neoplastic prostatic tissue that the possibility of neoplasm or mullerian duct vestige appeared extremely remote. An inflammatory pathogenesis should be considered since a minority of patients reviewed by Butterick and others presented with urinary infection. While inflammatory reaction of the urethral tissue may develop a papillary growth (proliferative papillary urethritis), 11 the predominant tissue element in the pseudotumor is connective tissue, not the glandular structures so abundant in our biopsy material. Furthermore, histological absence of inflammatory reaction as well as clinical absence of urinary infection in our patient makes it unlikely that the abnormality was of an inflammatory or infectious origin. Likewise, cellular constituents of the urethral tunica propria, not of the glands, are overwhelmingly present in fibrous polyps of the congenital type. 2 • 12 • 13 These urethral polyps differ from our patient in many other aspects since they are usually solid, pedunculated and associated with urinary obstruction in children. In short, the lesion reported herein appears to be best described as a clinicopathological entity of undetermined etiology.
1. Randall, A.: A study of the benign polyps of the male urethra. Surg., Gynec. & Obst., 17: 548, 1913. 2. Stueber, P. J. and Persky, L.: Solid tumors of the urethra and bladder neck. J. Urol., 102: 205, 1969. 3. Nesbit, R. M.: The genesis of benign polyps in the prostatic urethra. J. Urol., 87: 416, 1962. 4. Butterick, J. A., Schnitzer, B. and Abell, M. R.: Ectopic prostatic tissue in urethra: a clinicopathological entity and a significant cause of hematuria. J. Urol., 105: 97, 1971. 5. Brandes, D. and Bourne, G. H.: Histochemistry of the human prostate: normal and neoplastic. J. Path. Bact., 71: 33, 1956. 6. Dube, V. E., Farrow, G. M. and Greene, L. F.: Prostatic adenocarcinoma of ductal origin. Cancer, 32: 402, 1973. 7. Ende, N., Woods, L. P. and Shelley, H. S.: Carcinoma originating in ducts surrounding the prostatic urethra. Amer. J. Clin. Path., 40: 183, 1963. 8. Grabstald, H.: Tumors of the urethra in men and women. Cancer, 32: 1236, 1973. 9. Melicow, M. M. and Tannenbaum, M.: Endometrial carcinoma of uterus masculinus (prostatic utricle). Report of 6 cases. J. Urol., 106: 892, 1971. 10. Young, B. W. and Lagios, M. D.: Endometrial (papillary) carcinoma of prostatic utricle-response to orchiectomy, a case report. Cancer, 32: 1293, 1973. 11. Schinella, R., Thurm, J. and Feiner, M.: Papillary pseudotumor of the prostatic urethra: proliferative papillary urethritis. J. Urol., 111: 38, 1974. 12. Downs, R. A.: Congenital polyps of prostatic urethra. A review of the literature and report of two cases. Brit. J. Urol., 42: 76, 1970. 13. Kippusami, M. D. and Moors, D. E.: Fibrous polyp of verumontanum. Canad. J. Surg., 11: 388, 1968. 14. McNeal, J. E.: The prostate and prostatic urethra: a morphological synthesis. J. Urol., 107: 1008, 1972.
