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Seminal vesicle cyst
SEMINAL
VESICLE
Containing
Mesonephroid
D. H. BAGLEY, N. JAVADPOUR, F. G. WITEBSKY, L. B. THOMAS,
CYST Tumor
M.D. M.D. M.D. M.D.
From the Surgery Branch and Laboratory of Pathology, National Cancer Institute, National Institutes of Health, Bethesda, Maryland
ABSTRACT -A case of seminal vesicle cyst containing mesonephroid tumor is reported. The radioand pathologic features and the management of this unusual entity are described.
Cystic structures originating from the mesonephric ducts and their derivatives, including the seminal vesicles, are rare. In 1969, Davidson and Beard1 reviewed the literature and found reports of only 17 cases of seminal vesicle cyst. There have been a few additional cases reported since that series. Some of these cysts have been associated variously with ipsilateral agenesis of the kidney, ureter, or trigone.2-5 None of these reported cysts has been associated with tumor. Tumors of the seminal vesicle also are rare. Primary adenocarcinoma has been found most frequently, 6.7 but sarcoma has also been reported. 8 We report a case of seminal vesicle cyst containing a mesonephroid tumor. Case Report A twenty-nine-year-old man was referred to this Institute for evaluation and treatment of a cystic pelvic tumor. The patient gave a history of episodes of urinary retention for eight months; he had also noted a little difficulty in maintaining an erection. There were no other symptoms referrable to the genitourinary tract. Physical examination by the referring physician had revealed a retrovesical mass superior to the prostate. Excretory urogram and barium enema delineated a mass between the bladder and the rectum, compressing both structures. A cysto-
UROLOGY
/ JANUARY 1975 / VOLUME V, NUMBER 1
urethrogram and cystoscopy showed compression and elongation of the prostatic urethra. The diagnosis of miillerian duct cyst was entertained. On surgical exploration, a cystic mass was found which contained 500 ml. of greenish brown fluid and some friable tissue. A frozen section of the tumor was interpreted as undifferentiated malignant disease. Therefore, the cystic structure and the abdominal incision were closed with a Penrose drain in place, and the patient was referred to this Institute. Physical examination of the patient showed he had a midline scar with no drainage, and a mass was palpable in the pelvis, which extended about 7 cm. above the pubic bone. On rectal examination, the soft, irregular mass was palpable retrovesically, superior to the prostate and extending to the left. The remainder of the physical examination was unremarkable. Blood chemistries, and results of liver and renal function tests, alphafetoprotein, urinalysis, and urine culture were within normal limits. Chromosomal analysis indicated a normal male karyotype. Whole body bone scan, as well as gallium, liver, and brain scans, and metastatic bone survey were unremarkable. Excretory urogram demonstrated a persistent extensive compression over the left lateral superior portion of the bladder (Fig. 1A).
147
-
-
--.-
. -.-
FIGURE 1. (A) Excretory urogram showing compression over left lateral, superior pcrrtion of bladder. i B j Left seminal vesiculogram demonstrating left vas deferens and diluted left seminal vesicle, communicating with cystic structure, and (Cj post-drainage f;lm showing retention of contrast material in cystic structure.
Cystourethroscopy showed a normal verumontanum with no evidence of a midline tumor. The posterior urethra was partially occluded by a lateral mass compressing and pushing the urethra and bladder neck to the right. The ureteral orifices were in normal position, and there was no evidence of lesions within the bladder. The ejaculatory ducts could not be cannulated but appeared to be normal. Through a left vasotomy, seminal vesiculography was performed. This revealed dilatation of the left seminal vesicle which communicated with a large rounded collection of contrast material. Irrigation of the bladder failed to remove this material, which was still present six hours later (Fig. 1B and C). The original pathologic slides were reviewed and interpreted as an unclassified cystic tumor, possibly of low-grade malignant disease. On the basis of the clinical studies, it appeared that the tumor was arising from the left seminal vesicle rather than from a miillerian duct remnant. Therefore, it was necessary to explore the abdomen for possible excision of the tumor. On exploration, no evidence of metastatic lesions or involvement of regional lymph nodes was found. A large, retrovesicular cystic mass, measuring 8 by 10 by 6 cm., was adherent to the
148
rectum and encompassed the left ureter and both seminal vesicles. The cystic structure was opened, and after drainage of 250 ml. of greenish brown fluid, a 2 by 3 cm., friable papillary tumor was removed from the base and submitted for frozen section. The rectum and bladder were freed from the mass, but the left ureter was so closely adherent that a portion was removed, with subsequent performance of ureteroureterostomy. The mass was removed in continuity with the left lateral lobes of the prostate and the seminal vesicles. The patient made an mleventful recovery. Excretory urogram twelve months postoperativel!, showed only minor postsurgical irregularity of the bladder with no evidence of recurrence of tumor. Pathologic
diclgnosis
The surgical specimen included the prostate gland, both seminal vesicles, and distal portions of the vasa deferentia (Fig. 2A). There was, iI1 the area of the left seminal vesicle, a large cyst measuring 8 cm. in length and 4 to 5 cm. in width (Fig. 1B). A normal portion of the left seminal vesicle could not be found grossly. The cyst of the left seminal vesicle had a 4 to 5 mm.thick wall and an irregular, reddish brown inner surface. The cyst wall was firmly attached to the
distal 3 to 4 cm. of the left vas deferens (Fig. 2B). The thickened left vas deferens could be separated from the fibrotic wall of the cyst of the seminal vesicle only by sharp dissection. _Microscopically the right seminal vesicle and the right vas deferens were normal. There was slight hyperplasia of prostatic epithelium. Multiple sections through the cyst wall showed dense,
fibrous connective tissue. relatively acellular, Most of the tumor covering the inner surface of the cyst had been removed at the time of surgery, but a few sections showed an inner lining of tumor cells (Fig. 3A). The pattern of the tumor consisted of solid nodules of epithelial cells and slitlike or tubular spaces lined by tumor cells. The cells were small-to-moderate size, with irregular, slightly oval nuclei and indistinct cytoplasmic The cytoplasm was faintly stained borders. and lacked vacuoles or granular material (Fig. 3B). Diastase-resistant, PAS-positive secretory material w-as present in some of the slit-like and tubular spaces, and the cytoplasm of the tumor cells stained faintly with PAS. Mitotic figures were rarely observed. Occasionally, glomeruloid structures projected into the spaces within the tumor (Fig. 3C). In most areas, the epithelial tumor was confined to the inner surface of the cyst. However, in the area of dense fibrous comrective tissue which fused the cyst wall to the left vas deferens, there was infiltration by tumor cells (Fig. 3D), Comment
FIGURE 2. Gross specimens. (A) Posterior dew shows opened cyst of left seminal vesicle (LSV), right vas deferens (RVD), right seminal vesicle JRSV), and prostate gland(P) are grossly normal. (B) Thick-walled cyst of left seminal vesicle dissected from left vas in distal deferens showing dense white area of$brosis pm-tion of left Gas deferens (L\7D): which was fused with
wall
UROLOGY
of cyst.
i
~.4NUARY 1975
i
\‘OI_IJMEV.
NUMBER
1
The seminal vesicles and the vasa deferentia are derived from the mesonephric duct. Absence of all mesonephric derivatives could result from a developmental error occurring at the fourth week, but anomalies of the seminal vesicles alone could be initiated as late as the third month.” Similarly, early anomalies of the mesonephric duct could prevent the outpouching of the ureteral bud and subsequent metanephric and renal development. The association of cysts of the seminal vesicle with renal and ureteral agenesis might thus be explained on the basis of embryologic development. The clinical presentation of patients with benign developmental pelvic cysts has generally been the result of compression from the cystic structure causing obstructive uropathy and epididymitis. 3~10$11Most benign cysts of the seminal vesicle have been found in male patients during the third decade of life. Tumors of the seminal vesicles have usually been reported in patients between sixty and eighty years of age.‘,l’ The presenting symptoms of such tumors include obstruction, but occasionally hematuria is seen in the ejaculated semen. Diagnosis ofcystic structures discovered in the pelvis on physical examination may be aided by radiologic studies. Excretory uro- and cystourethrograms may reveal extensive compression.
149
FIGURE 3. Photomicrographs. (A) Cyst walk inner surface covered by thickened layer of epithelial tumor cells; wall composed of dense, relatively acellular connective tissue. (B) Higher magni$cation of tumor lining cyst, epithelial cells form solid nodules with scattered slit-like and (C) Occasional glomeruloid tubular spaces. nodules of tumor cells project into spaces within tumor. (0) Left vas deferens and infiltrating tumor: wall of vas deferens thickened by fibrous connective tissue; portion of lumen of vas deferens seen on right; infiltrating tumor from cyst of left seminal vesicle on left. (Hematoxylin and eosin stains.)
Seminal vesi~n~ograp~y either by retrograde ejaculatory duct cannulation13 or by direct vasotomyr4 is essential in differentiating farge cysts of uct from those of the seminal vesicle. Miillerian cysts usuahy present as a midline structure. Seminal vesicle cysts have been treated by excision, drainage into the bladder, or periodic external aspiration. Treatment of most malignant tumors of the seminal vesicle has been limited
150
because of late discovery. However, it is reporte that 1 patient benefited from local excision with subsequent hormonal therapy. l5 In this patient, wide local excision was performed because of the previous surgical procedure, the equivocal histoand the uncertain biologic logic appearance, behavior of the tumor. The precise origin of this cystic neoplasm is not known. Its location suggests that it arose from the seminal vesicle or some malformed mesonephric
UROLOGY
/ JANUARY 1975 / VOLUME V, P\‘U;LaBER;
structure. It is uncertain whether the tumor is benign or malignant. Eighteen months postoperatively the patient remains clinically free of tumor or recurrent symptoms, with normal excretory urogram and metastatic studies. Bethesda, Maryland 20014 (DR. BAGLEY) ACKNOWLEDGMENT. F. K. Mostofi M.D., Armed Forces Institute of Pathology, for reviewing the case and for his valuable advice; L. Kenzie Edwards for the gross photographs; and Ralph L. Isenburg for the photomicrographs. References DAVIDSON, A. C., and BEARD, J. F. : A seminal vesicle cyst: in association with ipsilateral renal agenesis and lumbar scoliosis, S. Med. J. 63: 608 (1969). GREENBAUM, E., and PEARMAN, R. 0.: Vasovesiculography: cyst of the seminal vesicle associated with agenesis of the ipsilateral kidney, Radiol. 98: 363 (1971). HARBITZ, T. B., and LIAVAG, I.: Urogenital malformation with cyst of the seminal vesicle, ipsilateral dilated ureter and renal agenesis, Stand. J. Urol. Nephrol. 2: 217 (1968). HART, J. B.: A case of cyst of the seminal vesicle, J. Urol. 96: 247 (1966).
UROLOGY
/ JANUARY 1975 / VOLUMEV,
NUMBER 1
Cyst of the 5. REDDY, Y. N., and WINTER, C. C.: seminal vesicle: a case report and review of the literature, ibid. 198: 134 (1972). 6. HAJDU, S. I., and FARUQUE, A. A.: Adenocarcinoma of the seminal vesicle, ibid. 99: 798 (1968). 7. AWADALLA, O., HUNT, A. C., and MILLER, A.: Primary carcinoma of the seminal vesicle, Brit. J. Ural. 40: 574 (1968). Primary tumours of 8. BUCK, A. C., and SHAW, R. E.: the retrovesical region with special reference to the mesenchymal tumours of the seminal vesicles, ibid. 44: 47 (1972). Embryology 9. GRAY, S. W., and SKANDALAKIS, J. E.: for Surgeons, Philadelphia, W. B. Saunders Co., 1972, p. 665. Cyst of 10. HEETDERKS, D. R., and DELAMBRE, L. E.: the seminal vesicle, J. Urol. 93: 725 (1965). 11. HIMCHI, D., and WIESENFELD, A.: Cyst of the seminal vesicle associated with ipsilateral renal agenesis: case report, ibid. 89: 966 (1963). Seminal vesicle carcinoma, ibid. 89: 12. EWELL, G. H.: 908 (1963). 13. STEWART, B. L., and NICOLL, G. A.: Cyst of the seminal vesicle, ibid. 62: 189 (1949). 14. ABESHOUSE, B. S., HELLER, E., and SALIK, J. 0.: Vaso-epididymography and vas seminal vesiculography. ibid. 72: 983 (1954). 0. S.: Clinical improvement 15. RODRIGUEZ-KEES, following estrogenic therapy in a case of primary adenocarcinoma of the seminal vesicle, ibid. 91: 665 (1964).
151
VESICLE
Containing
Mesonephroid
D. H. BAGLEY, N. JAVADPOUR, F. G. WITEBSKY, L. B. THOMAS,
CYST Tumor
M.D. M.D. M.D. M.D.
From the Surgery Branch and Laboratory of Pathology, National Cancer Institute, National Institutes of Health, Bethesda, Maryland
ABSTRACT -A case of seminal vesicle cyst containing mesonephroid tumor is reported. The radioand pathologic features and the management of this unusual entity are described.
Cystic structures originating from the mesonephric ducts and their derivatives, including the seminal vesicles, are rare. In 1969, Davidson and Beard1 reviewed the literature and found reports of only 17 cases of seminal vesicle cyst. There have been a few additional cases reported since that series. Some of these cysts have been associated variously with ipsilateral agenesis of the kidney, ureter, or trigone.2-5 None of these reported cysts has been associated with tumor. Tumors of the seminal vesicle also are rare. Primary adenocarcinoma has been found most frequently, 6.7 but sarcoma has also been reported. 8 We report a case of seminal vesicle cyst containing a mesonephroid tumor. Case Report A twenty-nine-year-old man was referred to this Institute for evaluation and treatment of a cystic pelvic tumor. The patient gave a history of episodes of urinary retention for eight months; he had also noted a little difficulty in maintaining an erection. There were no other symptoms referrable to the genitourinary tract. Physical examination by the referring physician had revealed a retrovesical mass superior to the prostate. Excretory urogram and barium enema delineated a mass between the bladder and the rectum, compressing both structures. A cysto-
UROLOGY
/ JANUARY 1975 / VOLUME V, NUMBER 1
urethrogram and cystoscopy showed compression and elongation of the prostatic urethra. The diagnosis of miillerian duct cyst was entertained. On surgical exploration, a cystic mass was found which contained 500 ml. of greenish brown fluid and some friable tissue. A frozen section of the tumor was interpreted as undifferentiated malignant disease. Therefore, the cystic structure and the abdominal incision were closed with a Penrose drain in place, and the patient was referred to this Institute. Physical examination of the patient showed he had a midline scar with no drainage, and a mass was palpable in the pelvis, which extended about 7 cm. above the pubic bone. On rectal examination, the soft, irregular mass was palpable retrovesically, superior to the prostate and extending to the left. The remainder of the physical examination was unremarkable. Blood chemistries, and results of liver and renal function tests, alphafetoprotein, urinalysis, and urine culture were within normal limits. Chromosomal analysis indicated a normal male karyotype. Whole body bone scan, as well as gallium, liver, and brain scans, and metastatic bone survey were unremarkable. Excretory urogram demonstrated a persistent extensive compression over the left lateral superior portion of the bladder (Fig. 1A).
147
-
-
--.-
. -.-
FIGURE 1. (A) Excretory urogram showing compression over left lateral, superior pcrrtion of bladder. i B j Left seminal vesiculogram demonstrating left vas deferens and diluted left seminal vesicle, communicating with cystic structure, and (Cj post-drainage f;lm showing retention of contrast material in cystic structure.
Cystourethroscopy showed a normal verumontanum with no evidence of a midline tumor. The posterior urethra was partially occluded by a lateral mass compressing and pushing the urethra and bladder neck to the right. The ureteral orifices were in normal position, and there was no evidence of lesions within the bladder. The ejaculatory ducts could not be cannulated but appeared to be normal. Through a left vasotomy, seminal vesiculography was performed. This revealed dilatation of the left seminal vesicle which communicated with a large rounded collection of contrast material. Irrigation of the bladder failed to remove this material, which was still present six hours later (Fig. 1B and C). The original pathologic slides were reviewed and interpreted as an unclassified cystic tumor, possibly of low-grade malignant disease. On the basis of the clinical studies, it appeared that the tumor was arising from the left seminal vesicle rather than from a miillerian duct remnant. Therefore, it was necessary to explore the abdomen for possible excision of the tumor. On exploration, no evidence of metastatic lesions or involvement of regional lymph nodes was found. A large, retrovesicular cystic mass, measuring 8 by 10 by 6 cm., was adherent to the
148
rectum and encompassed the left ureter and both seminal vesicles. The cystic structure was opened, and after drainage of 250 ml. of greenish brown fluid, a 2 by 3 cm., friable papillary tumor was removed from the base and submitted for frozen section. The rectum and bladder were freed from the mass, but the left ureter was so closely adherent that a portion was removed, with subsequent performance of ureteroureterostomy. The mass was removed in continuity with the left lateral lobes of the prostate and the seminal vesicles. The patient made an mleventful recovery. Excretory urogram twelve months postoperativel!, showed only minor postsurgical irregularity of the bladder with no evidence of recurrence of tumor. Pathologic
diclgnosis
The surgical specimen included the prostate gland, both seminal vesicles, and distal portions of the vasa deferentia (Fig. 2A). There was, iI1 the area of the left seminal vesicle, a large cyst measuring 8 cm. in length and 4 to 5 cm. in width (Fig. 1B). A normal portion of the left seminal vesicle could not be found grossly. The cyst of the left seminal vesicle had a 4 to 5 mm.thick wall and an irregular, reddish brown inner surface. The cyst wall was firmly attached to the
distal 3 to 4 cm. of the left vas deferens (Fig. 2B). The thickened left vas deferens could be separated from the fibrotic wall of the cyst of the seminal vesicle only by sharp dissection. _Microscopically the right seminal vesicle and the right vas deferens were normal. There was slight hyperplasia of prostatic epithelium. Multiple sections through the cyst wall showed dense,
fibrous connective tissue. relatively acellular, Most of the tumor covering the inner surface of the cyst had been removed at the time of surgery, but a few sections showed an inner lining of tumor cells (Fig. 3A). The pattern of the tumor consisted of solid nodules of epithelial cells and slitlike or tubular spaces lined by tumor cells. The cells were small-to-moderate size, with irregular, slightly oval nuclei and indistinct cytoplasmic The cytoplasm was faintly stained borders. and lacked vacuoles or granular material (Fig. 3B). Diastase-resistant, PAS-positive secretory material w-as present in some of the slit-like and tubular spaces, and the cytoplasm of the tumor cells stained faintly with PAS. Mitotic figures were rarely observed. Occasionally, glomeruloid structures projected into the spaces within the tumor (Fig. 3C). In most areas, the epithelial tumor was confined to the inner surface of the cyst. However, in the area of dense fibrous comrective tissue which fused the cyst wall to the left vas deferens, there was infiltration by tumor cells (Fig. 3D), Comment
FIGURE 2. Gross specimens. (A) Posterior dew shows opened cyst of left seminal vesicle (LSV), right vas deferens (RVD), right seminal vesicle JRSV), and prostate gland(P) are grossly normal. (B) Thick-walled cyst of left seminal vesicle dissected from left vas in distal deferens showing dense white area of$brosis pm-tion of left Gas deferens (L\7D): which was fused with
wall
UROLOGY
of cyst.
i
~.4NUARY 1975
i
\‘OI_IJMEV.
NUMBER
1
The seminal vesicles and the vasa deferentia are derived from the mesonephric duct. Absence of all mesonephric derivatives could result from a developmental error occurring at the fourth week, but anomalies of the seminal vesicles alone could be initiated as late as the third month.” Similarly, early anomalies of the mesonephric duct could prevent the outpouching of the ureteral bud and subsequent metanephric and renal development. The association of cysts of the seminal vesicle with renal and ureteral agenesis might thus be explained on the basis of embryologic development. The clinical presentation of patients with benign developmental pelvic cysts has generally been the result of compression from the cystic structure causing obstructive uropathy and epididymitis. 3~10$11Most benign cysts of the seminal vesicle have been found in male patients during the third decade of life. Tumors of the seminal vesicles have usually been reported in patients between sixty and eighty years of age.‘,l’ The presenting symptoms of such tumors include obstruction, but occasionally hematuria is seen in the ejaculated semen. Diagnosis ofcystic structures discovered in the pelvis on physical examination may be aided by radiologic studies. Excretory uro- and cystourethrograms may reveal extensive compression.
149
FIGURE 3. Photomicrographs. (A) Cyst walk inner surface covered by thickened layer of epithelial tumor cells; wall composed of dense, relatively acellular connective tissue. (B) Higher magni$cation of tumor lining cyst, epithelial cells form solid nodules with scattered slit-like and (C) Occasional glomeruloid tubular spaces. nodules of tumor cells project into spaces within tumor. (0) Left vas deferens and infiltrating tumor: wall of vas deferens thickened by fibrous connective tissue; portion of lumen of vas deferens seen on right; infiltrating tumor from cyst of left seminal vesicle on left. (Hematoxylin and eosin stains.)
Seminal vesi~n~ograp~y either by retrograde ejaculatory duct cannulation13 or by direct vasotomyr4 is essential in differentiating farge cysts of uct from those of the seminal vesicle. Miillerian cysts usuahy present as a midline structure. Seminal vesicle cysts have been treated by excision, drainage into the bladder, or periodic external aspiration. Treatment of most malignant tumors of the seminal vesicle has been limited
150
because of late discovery. However, it is reporte that 1 patient benefited from local excision with subsequent hormonal therapy. l5 In this patient, wide local excision was performed because of the previous surgical procedure, the equivocal histoand the uncertain biologic logic appearance, behavior of the tumor. The precise origin of this cystic neoplasm is not known. Its location suggests that it arose from the seminal vesicle or some malformed mesonephric
UROLOGY
/ JANUARY 1975 / VOLUME V, P\‘U;LaBER;
structure. It is uncertain whether the tumor is benign or malignant. Eighteen months postoperatively the patient remains clinically free of tumor or recurrent symptoms, with normal excretory urogram and metastatic studies. Bethesda, Maryland 20014 (DR. BAGLEY) ACKNOWLEDGMENT. F. K. Mostofi M.D., Armed Forces Institute of Pathology, for reviewing the case and for his valuable advice; L. Kenzie Edwards for the gross photographs; and Ralph L. Isenburg for the photomicrographs. References DAVIDSON, A. C., and BEARD, J. F. : A seminal vesicle cyst: in association with ipsilateral renal agenesis and lumbar scoliosis, S. Med. J. 63: 608 (1969). GREENBAUM, E., and PEARMAN, R. 0.: Vasovesiculography: cyst of the seminal vesicle associated with agenesis of the ipsilateral kidney, Radiol. 98: 363 (1971). HARBITZ, T. B., and LIAVAG, I.: Urogenital malformation with cyst of the seminal vesicle, ipsilateral dilated ureter and renal agenesis, Stand. J. Urol. Nephrol. 2: 217 (1968). HART, J. B.: A case of cyst of the seminal vesicle, J. Urol. 96: 247 (1966).
UROLOGY
/ JANUARY 1975 / VOLUMEV,
NUMBER 1
Cyst of the 5. REDDY, Y. N., and WINTER, C. C.: seminal vesicle: a case report and review of the literature, ibid. 198: 134 (1972). 6. HAJDU, S. I., and FARUQUE, A. A.: Adenocarcinoma of the seminal vesicle, ibid. 99: 798 (1968). 7. AWADALLA, O., HUNT, A. C., and MILLER, A.: Primary carcinoma of the seminal vesicle, Brit. J. Ural. 40: 574 (1968). Primary tumours of 8. BUCK, A. C., and SHAW, R. E.: the retrovesical region with special reference to the mesenchymal tumours of the seminal vesicles, ibid. 44: 47 (1972). Embryology 9. GRAY, S. W., and SKANDALAKIS, J. E.: for Surgeons, Philadelphia, W. B. Saunders Co., 1972, p. 665. Cyst of 10. HEETDERKS, D. R., and DELAMBRE, L. E.: the seminal vesicle, J. Urol. 93: 725 (1965). 11. HIMCHI, D., and WIESENFELD, A.: Cyst of the seminal vesicle associated with ipsilateral renal agenesis: case report, ibid. 89: 966 (1963). Seminal vesicle carcinoma, ibid. 89: 12. EWELL, G. H.: 908 (1963). 13. STEWART, B. L., and NICOLL, G. A.: Cyst of the seminal vesicle, ibid. 62: 189 (1949). 14. ABESHOUSE, B. S., HELLER, E., and SALIK, J. 0.: Vaso-epididymography and vas seminal vesiculography. ibid. 72: 983 (1954). 0. S.: Clinical improvement 15. RODRIGUEZ-KEES, following estrogenic therapy in a case of primary adenocarcinoma of the seminal vesicle, ibid. 91: 665 (1964).
151