- Email: [email protected]
Anal tenesmus caused by seminal vesicle cyst
ELSEVIER
ANAL TENESMUS P. CALLEWAERT,
CAUSED BY SEMINAL
M. DE COSTER,
P. VUYLSTEKE,
R. DE MAN,
VESICLE CYST S. BRIJS,
AND
L. BAERT
ABSTRACT Congenital cysts of the seminal vesicles with ipsilateral renal aplasia or dysplasia are rare but have been well described in the literature. We report the first case where anal tenesmus was the only presenting symptom. Another unique feature was the combination of this anomaly with a duplication of the inferior vena cava. Copyright 1997 by Elsevier Science Inc. UROLOGY 49: 139-l 41, 1997.
A
21-year-old man presented with a history of increasing anal tenesmus during the last few months. He had no other complaints, and his previous medical history was unremarkable. On rectal examination, the prostate was normal, but a soft, fluctuating, nontender mass was palpated just proximal to it on the left side. Further clinical examination was normal. Transabdominal pelvic sonography demonstrated a retrovesical cystic mass extending to the left, with an estimated diameter of 6 cm. No left kidney could be visualized; the right kidney was hypertrophied. These findings were confirmed by urography. A barium enema was noncontributory. Although the pelvic computed tomography (CT) scan showed a polycystic massbetween the bladder and the rectum, it could not identify with certainty the organ of origin. The CT scan did, however, show a duplication of the inferior vena cava (Fig. l), with the left vena cava crossing to the right at a level just proximal to the renal arteries. The two veins were of equal size. Magnetic resonance imaging (MRI) clearly demonstrated the mass to be a seminal vesicle cyst (Fig. 2). At cystoscopy, no left ureteral orifice could be found. Because of the increasing symptoms, surgery was performed. An abdominal transperitoneal approach was used for the vesiculectomy. The rectovesical pouch was opened. Using sharp dissection, the cleavage plane between the anterior part
From the Departments of Surgery and Radiology, H. Hart Ziekenhuis, Roeselare, Belgium, and Department of Urology, University Hospital Gasthuisberg, Leuven, Belgium Reprint requests: P. Caflewaert, M.D., Department of Urology, University Hospital Gasthuisberg, Herestraat 49, 3000 Leaven, Belgium
Submitted: April 16, 1996, accepted (with revisions): July 9, 1996 COPYKIGHL
1997
ALL
RESERVED
RIGHTS
BY ELSEVIER
SCIENCE
INC.
of the rectum and the cyst was entered until the cyst was completely freed down to the deferent duct, without injuring the latter. Its insertion was ligated. A silicone drain was left in place, and the peritoneum was closed. On pathologic examination, the diagnosis of seminal vesicle cyst was confirmed. The cyst presented a thin fibrotic wall, was monolocular, and contained some pale brown viscid fluid. It was lined by a flattened cuboidal epithelium. The patient made an uneventful recovery and was asymptomatic on follow-up at 6 and 12 months. COMMENT The ureteral bud sprouts from the mesonephric (wolffian) duct at 4 weeks’ gestation.’ The seminal vesicle develops from the same structure only in week 13.2 The distal parts of the wolffian ducts and ureters are gradually absorbed by the urogenital sinus and eventually form the trigone, which is the only mesodermal part of the bladder. Sprouting of the ureteral bud too far caudally results in a ureter placed more laterally than normal, with possible reflux. Sprouting too cranially gives delayed absorption, with ectopic opening of the ureter in the trigone, bladder neck, urethra, ejaculatory duct, deferent duct, or seminal vesicle. In men, an ectopic ureter never drains below the level of the external sphincter, so incontinence does not ensue.3 The ureteral bud has to reach the metanephrogenic tissue punctually and centrally to induce normal development of the kidney.4 The closely related embryologic development of these structures explains why a defect in the wolffian duct and ureteral bud may give rise to an ectopic ureter, renal aplasia or dysplasia, a 0090-4295/97/$17.00 PII SOO90-4295(96)00366-4
139
FIGURE 1. A computed tomography scan at the level where the left vena cava crosses over to the right.
FIGURE 2. The polycystic seminal vesicle on the left is shown in detail by magnetic resonance imaging.
seminal vesicle cyst, or any combination of these. The combination of renal agenesis and Gartner’s cyst in the female has been reported as well. One study found 235 cases of renal agenesis in a total of 280,000 children during renal ultrasound screening. Of those, 13 were associated with a seminal vesicle cyst or Gartner’s cyst.5 A few cases of an absent testis or vas deferens associated with a seminal vesicle cyst also have been recorded.6 Cysts of the seminal vesicles also can be acquired following blockage caused by inflammation or calculus formation in the ejaculatory duct.7 Symptoms related to seminal vesicle cysts usually develop between the ages of 20 and 40 years, the period of greatest sexual activity.* Most patients have a history suggesting chronic prostatitis or recurrent epididymitis with unsuccessful anti140
biotic treatment. The clinical features include prostatism (urgency, frequency, hesitancy), perineal discomfort, epididymal pain, painful ejaculation, and, infrequently, pain on defecation or constipation. Impaired fertility also can be the reason for investigations leading to the diagnosis of these anomalies.8 To our knowledge, this case is the first to be reported where anal tenesmus is the only presenting symptom. Diagnosis usually is made by a combination of history, physical examination, and transrectal and abdominal ultrasonography or CT scan. Other useful studies are intravenous urography, cystoscopy, and MRL9 Vasovesiculography is possible but carries the risk of (further) impairing fertility and causing infection. Rectal or perineal aspiration under ultrasonographic guidance, with injection of contrast media, offers an alternative. In our experience, MRI gave the best noninvasive visualization of the seminal vesicle cyst and the surrounding organs. Treatment should be restricted to symptomatic cases. Several options are available.3 Aspiration (repeated if necessary) has been used with success, but possible recurrence and infection are the disadvantages. Transurethral resection with unroofing of the cyst can be considered. Most authors prefer complete removal of the cyst through an open procedure, using either an abdominal transperitoneal, transvesical, or retroperitoneal approach, which also permits resection of a possible dysplastic kidney. Pathologic examination shows an ectatic and atrophied seminal vesicle with possible inflammation, typically filled with a brown fluid containing spermatozoa, leukocytes, and red blood cells.8 Seminal vesicle carcinoma is extremely rare.l’ Duplication of the inferior vena cava has an incidence of 0.2% to 3.0%.” This is the result of aberrations in a complex process of embryogenesis.‘* The clinical relevance of this anomaly usually is limited to surgical problems during retroperitoneal procedures.13 Ours is the first report of inferior vena cava duplication in combination with a seminal vesicle cyst. In conclusion, our patient’s history proves that seminal vesicle cysts should also be considered in the differential diagnosis of anal tenesmus. Whether there is an embryologic link with the associated duplication of the vena cava remains an open question. REFERENCES 1. Tanagho EA: Embryology of the genitourinary system, in Tanagho EA, and McAninch JW (Eds): Smith’s General Urology. Norwalk, Conn, Appleton & Lange, 1995, pp 1730. UROLOGY
49 (I), 1997
2. Zaontz MR, and Kass EJ: Ectopic ureter opening into seminal vesicle cyst associated with ipsilateral renal agenesis. Urology 29: 523-525, 1987. 3. Rohrborn C, Schneider HJ, Alles JU, and Engstfeld J: Embryologic and diagnostic aspects of associated malformation of upper urinary and genital tracts. Urology 22: 588593, 1983. 4. Caldamone AA: Embryology, in Sant GR (Ed): Pathophysiologic Principles of Urology. Boston, Blackwell Scientific Publications, 1994, pp l-29. 5. Sheih CP, Hung CS, Wei CF, and Lin CY: Cystic dilatations within the pelvis in patients with ipsilateral renal agenesis or dysplasia. J Urol 144: 324-327, 1990. 6. Omstein MH, and Kershaw DR: Cysts of the seminal vesicle are Mtillerian in origin. J R Sot Med 78: 1050-1051, 1985. 7. Conn IG, Peeling WB, and Clements R: Complete resolution of a seminal vesicle cyst-evidence for an obstructive aetiology. Br J Urol 69: 636-639, 1992.
UROLOGY
49 (I), I997
8. Rohrborn CG, Schneider HJ, Rugendorff EW, and Hamann W: Embryological and diagnostic aspects of seminal vesicle cysts associated with upper urinary tract malformation. J Urol 135: 1029-1032, 1986. 9. Rappe BMJ, Meuleman EJH, and Debruyne FMJ: Seminal vesicle cyst with ipsilateral renal agenesis. Urol Int 50: 54-56, 1993. 10. Okada Y, Tanaka H, Takeuchi H, and Yoshida 0: Papillary adenocarcinoma in a seminal vesicle cyst associated with ipsilateral renal agenesis: a case report. J Urol 148: 15431545,1992. 11. Giordano JM, and Trout HH III: Anomalies of the inferior vena cava. J Vast Surg 3: 924-928, 1986. 12. Hinman F: Venous system, in Hinman F (Ed): Atlas of Urosurgical Anatomy. Philadelphia, WB Saunders, 1993, pp 15-28. 13. Bartle EJ, Pearce WH, Sun JH, and Rutherford RB: Infrarenal venous anomalies and aortic surgery: avoiding vascular injury. J Vast Surg 6: 590-593, 1987.
141
ANAL TENESMUS P. CALLEWAERT,
CAUSED BY SEMINAL
M. DE COSTER,
P. VUYLSTEKE,
R. DE MAN,
VESICLE CYST S. BRIJS,
AND
L. BAERT
ABSTRACT Congenital cysts of the seminal vesicles with ipsilateral renal aplasia or dysplasia are rare but have been well described in the literature. We report the first case where anal tenesmus was the only presenting symptom. Another unique feature was the combination of this anomaly with a duplication of the inferior vena cava. Copyright 1997 by Elsevier Science Inc. UROLOGY 49: 139-l 41, 1997.
A
21-year-old man presented with a history of increasing anal tenesmus during the last few months. He had no other complaints, and his previous medical history was unremarkable. On rectal examination, the prostate was normal, but a soft, fluctuating, nontender mass was palpated just proximal to it on the left side. Further clinical examination was normal. Transabdominal pelvic sonography demonstrated a retrovesical cystic mass extending to the left, with an estimated diameter of 6 cm. No left kidney could be visualized; the right kidney was hypertrophied. These findings were confirmed by urography. A barium enema was noncontributory. Although the pelvic computed tomography (CT) scan showed a polycystic massbetween the bladder and the rectum, it could not identify with certainty the organ of origin. The CT scan did, however, show a duplication of the inferior vena cava (Fig. l), with the left vena cava crossing to the right at a level just proximal to the renal arteries. The two veins were of equal size. Magnetic resonance imaging (MRI) clearly demonstrated the mass to be a seminal vesicle cyst (Fig. 2). At cystoscopy, no left ureteral orifice could be found. Because of the increasing symptoms, surgery was performed. An abdominal transperitoneal approach was used for the vesiculectomy. The rectovesical pouch was opened. Using sharp dissection, the cleavage plane between the anterior part
From the Departments of Surgery and Radiology, H. Hart Ziekenhuis, Roeselare, Belgium, and Department of Urology, University Hospital Gasthuisberg, Leuven, Belgium Reprint requests: P. Caflewaert, M.D., Department of Urology, University Hospital Gasthuisberg, Herestraat 49, 3000 Leaven, Belgium
Submitted: April 16, 1996, accepted (with revisions): July 9, 1996 COPYKIGHL
1997
ALL
RESERVED
RIGHTS
BY ELSEVIER
SCIENCE
INC.
of the rectum and the cyst was entered until the cyst was completely freed down to the deferent duct, without injuring the latter. Its insertion was ligated. A silicone drain was left in place, and the peritoneum was closed. On pathologic examination, the diagnosis of seminal vesicle cyst was confirmed. The cyst presented a thin fibrotic wall, was monolocular, and contained some pale brown viscid fluid. It was lined by a flattened cuboidal epithelium. The patient made an uneventful recovery and was asymptomatic on follow-up at 6 and 12 months. COMMENT The ureteral bud sprouts from the mesonephric (wolffian) duct at 4 weeks’ gestation.’ The seminal vesicle develops from the same structure only in week 13.2 The distal parts of the wolffian ducts and ureters are gradually absorbed by the urogenital sinus and eventually form the trigone, which is the only mesodermal part of the bladder. Sprouting of the ureteral bud too far caudally results in a ureter placed more laterally than normal, with possible reflux. Sprouting too cranially gives delayed absorption, with ectopic opening of the ureter in the trigone, bladder neck, urethra, ejaculatory duct, deferent duct, or seminal vesicle. In men, an ectopic ureter never drains below the level of the external sphincter, so incontinence does not ensue.3 The ureteral bud has to reach the metanephrogenic tissue punctually and centrally to induce normal development of the kidney.4 The closely related embryologic development of these structures explains why a defect in the wolffian duct and ureteral bud may give rise to an ectopic ureter, renal aplasia or dysplasia, a 0090-4295/97/$17.00 PII SOO90-4295(96)00366-4
139
FIGURE 1. A computed tomography scan at the level where the left vena cava crosses over to the right.
FIGURE 2. The polycystic seminal vesicle on the left is shown in detail by magnetic resonance imaging.
seminal vesicle cyst, or any combination of these. The combination of renal agenesis and Gartner’s cyst in the female has been reported as well. One study found 235 cases of renal agenesis in a total of 280,000 children during renal ultrasound screening. Of those, 13 were associated with a seminal vesicle cyst or Gartner’s cyst.5 A few cases of an absent testis or vas deferens associated with a seminal vesicle cyst also have been recorded.6 Cysts of the seminal vesicles also can be acquired following blockage caused by inflammation or calculus formation in the ejaculatory duct.7 Symptoms related to seminal vesicle cysts usually develop between the ages of 20 and 40 years, the period of greatest sexual activity.* Most patients have a history suggesting chronic prostatitis or recurrent epididymitis with unsuccessful anti140
biotic treatment. The clinical features include prostatism (urgency, frequency, hesitancy), perineal discomfort, epididymal pain, painful ejaculation, and, infrequently, pain on defecation or constipation. Impaired fertility also can be the reason for investigations leading to the diagnosis of these anomalies.8 To our knowledge, this case is the first to be reported where anal tenesmus is the only presenting symptom. Diagnosis usually is made by a combination of history, physical examination, and transrectal and abdominal ultrasonography or CT scan. Other useful studies are intravenous urography, cystoscopy, and MRL9 Vasovesiculography is possible but carries the risk of (further) impairing fertility and causing infection. Rectal or perineal aspiration under ultrasonographic guidance, with injection of contrast media, offers an alternative. In our experience, MRI gave the best noninvasive visualization of the seminal vesicle cyst and the surrounding organs. Treatment should be restricted to symptomatic cases. Several options are available.3 Aspiration (repeated if necessary) has been used with success, but possible recurrence and infection are the disadvantages. Transurethral resection with unroofing of the cyst can be considered. Most authors prefer complete removal of the cyst through an open procedure, using either an abdominal transperitoneal, transvesical, or retroperitoneal approach, which also permits resection of a possible dysplastic kidney. Pathologic examination shows an ectatic and atrophied seminal vesicle with possible inflammation, typically filled with a brown fluid containing spermatozoa, leukocytes, and red blood cells.8 Seminal vesicle carcinoma is extremely rare.l’ Duplication of the inferior vena cava has an incidence of 0.2% to 3.0%.” This is the result of aberrations in a complex process of embryogenesis.‘* The clinical relevance of this anomaly usually is limited to surgical problems during retroperitoneal procedures.13 Ours is the first report of inferior vena cava duplication in combination with a seminal vesicle cyst. In conclusion, our patient’s history proves that seminal vesicle cysts should also be considered in the differential diagnosis of anal tenesmus. Whether there is an embryologic link with the associated duplication of the vena cava remains an open question. REFERENCES 1. Tanagho EA: Embryology of the genitourinary system, in Tanagho EA, and McAninch JW (Eds): Smith’s General Urology. Norwalk, Conn, Appleton & Lange, 1995, pp 1730. UROLOGY
49 (I), 1997
2. Zaontz MR, and Kass EJ: Ectopic ureter opening into seminal vesicle cyst associated with ipsilateral renal agenesis. Urology 29: 523-525, 1987. 3. Rohrborn C, Schneider HJ, Alles JU, and Engstfeld J: Embryologic and diagnostic aspects of associated malformation of upper urinary and genital tracts. Urology 22: 588593, 1983. 4. Caldamone AA: Embryology, in Sant GR (Ed): Pathophysiologic Principles of Urology. Boston, Blackwell Scientific Publications, 1994, pp l-29. 5. Sheih CP, Hung CS, Wei CF, and Lin CY: Cystic dilatations within the pelvis in patients with ipsilateral renal agenesis or dysplasia. J Urol 144: 324-327, 1990. 6. Omstein MH, and Kershaw DR: Cysts of the seminal vesicle are Mtillerian in origin. J R Sot Med 78: 1050-1051, 1985. 7. Conn IG, Peeling WB, and Clements R: Complete resolution of a seminal vesicle cyst-evidence for an obstructive aetiology. Br J Urol 69: 636-639, 1992.
UROLOGY
49 (I), I997
8. Rohrborn CG, Schneider HJ, Rugendorff EW, and Hamann W: Embryological and diagnostic aspects of seminal vesicle cysts associated with upper urinary tract malformation. J Urol 135: 1029-1032, 1986. 9. Rappe BMJ, Meuleman EJH, and Debruyne FMJ: Seminal vesicle cyst with ipsilateral renal agenesis. Urol Int 50: 54-56, 1993. 10. Okada Y, Tanaka H, Takeuchi H, and Yoshida 0: Papillary adenocarcinoma in a seminal vesicle cyst associated with ipsilateral renal agenesis: a case report. J Urol 148: 15431545,1992. 11. Giordano JM, and Trout HH III: Anomalies of the inferior vena cava. J Vast Surg 3: 924-928, 1986. 12. Hinman F: Venous system, in Hinman F (Ed): Atlas of Urosurgical Anatomy. Philadelphia, WB Saunders, 1993, pp 15-28. 13. Bartle EJ, Pearce WH, Sun JH, and Rutherford RB: Infrarenal venous anomalies and aortic surgery: avoiding vascular injury. J Vast Surg 6: 590-593, 1987.
141