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Urethral diverticulum with multiple calculi with presentation of urinary incontinence in a female — A case report and literature review
Urological Science 25 (2014) 129e131
Contents lists available at ScienceDirect
Urological Science journal homepage: www.urol-sci.com
Case report
Urethral diverticulum with multiple calculi with presentation of urinary incontinence in a female d A case report and literature review Te-Chi Lin a, b, Justin Ji-Yuen Siu a, b, Eric Chieh-Lung Chou a, b, * a b
Department of Urology, China Medical University Hospital, Taichung, Taiwan School of Medicine, China Medical University, Taichung, Taiwan
a r t i c l e i n f o
a b s t r a c t
Article history: Received 24 April 2014 Received in revised form 17 June 2014 Accepted 19 June 2014 Available online 30 July 2014
Urethral diverticulum with multiple calculi is a rare condition that is often difficult to diagnose. We present a 46-year-old female with initial symptoms of stress urinary incontinence and subconscious urine leakage. Magnetic resonance imaging revealed a large diverticulum with multiple stones in it. Transvaginal repair of the urethral diverticulum was performed. Some authors and textbooks suggest concomitant anti-incontinence surgery in patients with a similar condition, but we did not do so under the considerations of patient autonomy and risk of infection. The incontinence subsided spontaneously after diverticulectomy alone, against our expectations. Copyright © 2014, Taiwan Urological Association. Published by Elsevier Taiwan LLC. All rights reserved.
Keywords: diverticulum stone urethra calculi urethra diverticulum urethra stone
1. Introduction Urethral diverticulum (UD) with multiple calculi is a rare condition that is often difficult to diagnose. We present a 46-year-old female with initial symptoms of stress urinary incontinence (SUI) and subconscious urine leakage. Magnetic resonance imaging (MRI) revealed a large diverticulum with multiple stones in it. Transvaginal repair of the UD was performed, and her SUI subsided after diverticulectomy alone, against our expectations. 2. Case Report A 46-year-old female presented with SUI and subconscious urine leakage for 6 months that worsened shortly prior to her admission. She had not visited other physicians for this problem although this really troubled her. The patient had to change her trousers two to three times a day, and had to use two to three pads per day. She denied dysuria, dyspareunia, other lower urinary tract symptoms, or urinary tract infection. She had three pregnancies that resulted in two natural spontaneous deliveries and one cesarean section. The result of her urinalysis was within normal limits, and the pelvic examination revealed urine leak when
* Corresponding author. Department of Urology, China Medical University Hospital, 2 Yude Road, Taichung, 40447, Taiwan, ROC. E-mail address: [email protected] (E.C.-L. Chou).
coughing and no continuous urine leakage. In addition, a 2 cm 1 cm hard nodule was noted at the anterior urethra (Fig. 1). There was no discharge after urethral stripping. A cystourethroscopy did not reveal any specific finding. Her MRI scan revealed a 2-cm UD at the distal urethra 3 cm away from the bladder neck with four well-defined stones, and the wall of the diverticulum was smooth (Fig. 2). We suggested transvaginal urethral diverticulectomy with concomitant autologous pubovaginal fascial sling, but the patient decided not to undergo anti-incontinence surgery because of infection risk and postoperative pain. The patient was placed in a lithotomy position. The stones were exposed with a longitudinal incision (Fig. 3) to the ostium communicating with the diverticulum to the urethral lumen. An en bloc excision was performed, and all stones were removed (Fig. 4). Multilayered, watertight closure of the urethra and vagina wall was performed with absorbable sutures. At 2 weeks after the operation, the wound was well healed and her incontinence subsided after diverticulectomy alone. Her condition did not change at 6 months after the operation, against our expectations. 3. Discussion UD was first reported 2 centuries ago by Hey,1 but until the invention of positive-pressure urethrography in 1952 by Davis and Cian,2 only a few cases were discovered. Up to 20% of patients may be asymptomatic; therefore, the true incidence is unclear because
http://dx.doi.org/10.1016/j.urols.2014.06.002 1879-5226/Copyright © 2014, Taiwan Urological Association. Published by Elsevier Taiwan LLC. All rights reserved.
130
T.-C. Lin et al. / Urological Science 25 (2014) 129e131
Fig. 1. A 2 cm 1 cm hard nodule over the anterior urethra. Fig. 3. En bloc excision was performed and the urethral wall repaired.
of missed diagnoses, although the accepted prevalence is about 0.5e6%.3 UD patients present with diverse symptoms, but the classical symptoms of “3Ds” are dysuria, dyspareunia, and postvoiding dribbling. Other symptoms associated with UD are frequency, urgency, hematuria, SUI, and persistent pyuria. The most important findings on physical examination are palpable vaginal mass and discharge after stripping. However, in a series with 46 female patients with urethral diverticula, the mean time from occurrence of symptoms to diagnosis is 5.2 years, despite the fact that 52% of those patients had palpable vaginal masses3das in the case of our patient, who did not notice the vaginal mass but sought medical advice because of symptoms of SUI. Double balloon positive pressure urethrography is a good diagnostic tool but is not widely available. Compared to voiding cystourethrography and ultrasonography, high-resolution MRI has higher sensitivity and specificity, especially when differential diagnoses of urethral masses other than UD are present.4,5
Fig. 2. Four smooth stones inside the urethral diverticulum.
UD presenting with urolithiasis is uncommon, with an incidence of 1e10%. The etiology may be deposition, stasis of infected urine, or stone migration. To our knowledge, studies seldom compare the diagnosis and management between UD with or without stones. Almost all UD with stone present with palpable hard vaginal masses, and patients tend to receive surgical excision rather than conservative treatment. The management of UD depends on whether it is symptomatic. Low-dose antibiotics and urethral stripping after voiding may be used in asymptomatic patients or patients with smaller diverticula. Some case reports revealed that malignancies such as squamous cell carcinoma, adenocarcinoma, and clear cell carcinoma may originate from the diverticulum, but the risk is very low, and only about 100 cases have been reported so far.6 This should be made known to the patient, and regular follow-up should be advised. For symptomatic patients, surgical repair is indicated. Complete excisional diverticulectomy with multilayer watertight closure showed the highest efficacy in most patients with small defects. In patients with larger defects, poor tissue quality, or suboptimal blood supply, Martius flap is a reliable procedure with a low complication rate. Up to 30% of patients have SUI prior to diverticulectomy, and about 15e49% had de novo SUI after
Fig. 4. All the stones were removed.
T.-C. Lin et al. / Urological Science 25 (2014) 129e131
surgery.3,4,7,8 For patients with UD and preoperative SUI, antiincontinence surgery such as pubovaginal fascial sling resulted in a significant decrease in postoperative SUI. The use of slings is safe and does not increase infection risk, and it is considered the “gold standard” treatment modality.3,9,10 The synthetic midurethral sling used for this condition is not well studied, and most authors would avoid using it owing to the potentially increased infection risk. Our patient had a large UD with stones with concomitant SUI, and she opted not to undergo anti-incontinence surgery. However, her SUI was cured after diverticulectomy, which was against our expectations. In another series, 89 patients with UD received diverticulectomy with or without Martius flap but not anti-incontinence surgery; 32 patients had preoperational SUI, and 20 of them were cured after diverticulectomy without sling insertion. De novo SUI was noted in 13 patients, and a total of 25 patients had postoperational SUI. Twelve out of 25 patients had mild symptoms and did not require further management.4 Another study of 50 patients with UD reported an incidence of de novo SUI rate as 49%, but most patients had mild symptoms and only five patients required subsequent anti-incontinence surgery.8 The mechanism of how diverticulectomy cures SUI is unclear. One hypothesis is that a large diverticulum may compromise urethral support and cause SUI. Another hypothesis is that the preoperational SUI reported in certain patients was not true SUI, but was actually paradoxical stress incontinence, that is, the loss of retained urine in the diverticulum during coughing. In conclusion, in patients with UD and concomitant SUI, diverticulectomy with anti-incontinence surgery is accepted and performed by most clinicians; however, staged operation is another alternative that may prevent unnecessary treatment.
131
Conflicts of interest The authors declare that they have no financial or non-financial conflicts of interest related to the subject matter or materials discussed in the manuscript.
Sources of funding No funding was received for the work described in this article.
References 1. Hey W. Practical observations in surgery: illustrated with cases. Philadelphia: James Humphreys; 1805. 2. Davis HJ, Cian LG. Positive pressure urethrography: a new diagnostic method. J Urol 1952;68:611e6. 3. Romanzi LJ, Groutz A, Blaivas JG. Urethral diverticulum in women: diverse presentations resulting in diagnostic delay and mismanagement. J Urol 2000;164:428e33. 4. Reeves FA, Inman RD, Chapple CR. Management of symptomatic urethral diverticula in women: a single-centre experience. Eur Urol 2014;66(1):164e72. 5. Singla P, Long SS, Long CM, Genadry RR, Macura KJ. Imaging of the female urethral diverticulum. Clin Radiol 2013;68:e418e25. 6. Thomas AA, Rackley RR, Lee U, Goldman HB, Vasavada SP, Hansel DE. Urethral diverticula in 90 female patients: a study with emphasis on neoplastic alterations. J Urol 2008;180:2463e7. 7. Ganabathi K, Leach GE, Zimmern PE, Dmochowski R. Experience with the management of urethral diverticulum in 63 women. J Urol 1994;152:1445e52. 8. Lee UJ, Goldman H, Moore C. Rate of de novo stress urinary incontinence after urethral diverticulum repair. J Urol 2008;71(5):849e53. 9. Faerber GJ. Urethral diverticulectomy and pubovaginal sling for simultaneous treatment of urethral diverticulum and intrinsic sphincter deficiency. Tech Urol 1998;4:192e7. 10. Bass JS, Leach GE. Surgical treatment of concomitant urethral diverticulum and stress incontinence. Urol Clin North Am 1991;18:365e73.
Contents lists available at ScienceDirect
Urological Science journal homepage: www.urol-sci.com
Case report
Urethral diverticulum with multiple calculi with presentation of urinary incontinence in a female d A case report and literature review Te-Chi Lin a, b, Justin Ji-Yuen Siu a, b, Eric Chieh-Lung Chou a, b, * a b
Department of Urology, China Medical University Hospital, Taichung, Taiwan School of Medicine, China Medical University, Taichung, Taiwan
a r t i c l e i n f o
a b s t r a c t
Article history: Received 24 April 2014 Received in revised form 17 June 2014 Accepted 19 June 2014 Available online 30 July 2014
Urethral diverticulum with multiple calculi is a rare condition that is often difficult to diagnose. We present a 46-year-old female with initial symptoms of stress urinary incontinence and subconscious urine leakage. Magnetic resonance imaging revealed a large diverticulum with multiple stones in it. Transvaginal repair of the urethral diverticulum was performed. Some authors and textbooks suggest concomitant anti-incontinence surgery in patients with a similar condition, but we did not do so under the considerations of patient autonomy and risk of infection. The incontinence subsided spontaneously after diverticulectomy alone, against our expectations. Copyright © 2014, Taiwan Urological Association. Published by Elsevier Taiwan LLC. All rights reserved.
Keywords: diverticulum stone urethra calculi urethra diverticulum urethra stone
1. Introduction Urethral diverticulum (UD) with multiple calculi is a rare condition that is often difficult to diagnose. We present a 46-year-old female with initial symptoms of stress urinary incontinence (SUI) and subconscious urine leakage. Magnetic resonance imaging (MRI) revealed a large diverticulum with multiple stones in it. Transvaginal repair of the UD was performed, and her SUI subsided after diverticulectomy alone, against our expectations. 2. Case Report A 46-year-old female presented with SUI and subconscious urine leakage for 6 months that worsened shortly prior to her admission. She had not visited other physicians for this problem although this really troubled her. The patient had to change her trousers two to three times a day, and had to use two to three pads per day. She denied dysuria, dyspareunia, other lower urinary tract symptoms, or urinary tract infection. She had three pregnancies that resulted in two natural spontaneous deliveries and one cesarean section. The result of her urinalysis was within normal limits, and the pelvic examination revealed urine leak when
* Corresponding author. Department of Urology, China Medical University Hospital, 2 Yude Road, Taichung, 40447, Taiwan, ROC. E-mail address: [email protected] (E.C.-L. Chou).
coughing and no continuous urine leakage. In addition, a 2 cm 1 cm hard nodule was noted at the anterior urethra (Fig. 1). There was no discharge after urethral stripping. A cystourethroscopy did not reveal any specific finding. Her MRI scan revealed a 2-cm UD at the distal urethra 3 cm away from the bladder neck with four well-defined stones, and the wall of the diverticulum was smooth (Fig. 2). We suggested transvaginal urethral diverticulectomy with concomitant autologous pubovaginal fascial sling, but the patient decided not to undergo anti-incontinence surgery because of infection risk and postoperative pain. The patient was placed in a lithotomy position. The stones were exposed with a longitudinal incision (Fig. 3) to the ostium communicating with the diverticulum to the urethral lumen. An en bloc excision was performed, and all stones were removed (Fig. 4). Multilayered, watertight closure of the urethra and vagina wall was performed with absorbable sutures. At 2 weeks after the operation, the wound was well healed and her incontinence subsided after diverticulectomy alone. Her condition did not change at 6 months after the operation, against our expectations. 3. Discussion UD was first reported 2 centuries ago by Hey,1 but until the invention of positive-pressure urethrography in 1952 by Davis and Cian,2 only a few cases were discovered. Up to 20% of patients may be asymptomatic; therefore, the true incidence is unclear because
http://dx.doi.org/10.1016/j.urols.2014.06.002 1879-5226/Copyright © 2014, Taiwan Urological Association. Published by Elsevier Taiwan LLC. All rights reserved.
130
T.-C. Lin et al. / Urological Science 25 (2014) 129e131
Fig. 1. A 2 cm 1 cm hard nodule over the anterior urethra. Fig. 3. En bloc excision was performed and the urethral wall repaired.
of missed diagnoses, although the accepted prevalence is about 0.5e6%.3 UD patients present with diverse symptoms, but the classical symptoms of “3Ds” are dysuria, dyspareunia, and postvoiding dribbling. Other symptoms associated with UD are frequency, urgency, hematuria, SUI, and persistent pyuria. The most important findings on physical examination are palpable vaginal mass and discharge after stripping. However, in a series with 46 female patients with urethral diverticula, the mean time from occurrence of symptoms to diagnosis is 5.2 years, despite the fact that 52% of those patients had palpable vaginal masses3das in the case of our patient, who did not notice the vaginal mass but sought medical advice because of symptoms of SUI. Double balloon positive pressure urethrography is a good diagnostic tool but is not widely available. Compared to voiding cystourethrography and ultrasonography, high-resolution MRI has higher sensitivity and specificity, especially when differential diagnoses of urethral masses other than UD are present.4,5
Fig. 2. Four smooth stones inside the urethral diverticulum.
UD presenting with urolithiasis is uncommon, with an incidence of 1e10%. The etiology may be deposition, stasis of infected urine, or stone migration. To our knowledge, studies seldom compare the diagnosis and management between UD with or without stones. Almost all UD with stone present with palpable hard vaginal masses, and patients tend to receive surgical excision rather than conservative treatment. The management of UD depends on whether it is symptomatic. Low-dose antibiotics and urethral stripping after voiding may be used in asymptomatic patients or patients with smaller diverticula. Some case reports revealed that malignancies such as squamous cell carcinoma, adenocarcinoma, and clear cell carcinoma may originate from the diverticulum, but the risk is very low, and only about 100 cases have been reported so far.6 This should be made known to the patient, and regular follow-up should be advised. For symptomatic patients, surgical repair is indicated. Complete excisional diverticulectomy with multilayer watertight closure showed the highest efficacy in most patients with small defects. In patients with larger defects, poor tissue quality, or suboptimal blood supply, Martius flap is a reliable procedure with a low complication rate. Up to 30% of patients have SUI prior to diverticulectomy, and about 15e49% had de novo SUI after
Fig. 4. All the stones were removed.
T.-C. Lin et al. / Urological Science 25 (2014) 129e131
surgery.3,4,7,8 For patients with UD and preoperative SUI, antiincontinence surgery such as pubovaginal fascial sling resulted in a significant decrease in postoperative SUI. The use of slings is safe and does not increase infection risk, and it is considered the “gold standard” treatment modality.3,9,10 The synthetic midurethral sling used for this condition is not well studied, and most authors would avoid using it owing to the potentially increased infection risk. Our patient had a large UD with stones with concomitant SUI, and she opted not to undergo anti-incontinence surgery. However, her SUI was cured after diverticulectomy, which was against our expectations. In another series, 89 patients with UD received diverticulectomy with or without Martius flap but not anti-incontinence surgery; 32 patients had preoperational SUI, and 20 of them were cured after diverticulectomy without sling insertion. De novo SUI was noted in 13 patients, and a total of 25 patients had postoperational SUI. Twelve out of 25 patients had mild symptoms and did not require further management.4 Another study of 50 patients with UD reported an incidence of de novo SUI rate as 49%, but most patients had mild symptoms and only five patients required subsequent anti-incontinence surgery.8 The mechanism of how diverticulectomy cures SUI is unclear. One hypothesis is that a large diverticulum may compromise urethral support and cause SUI. Another hypothesis is that the preoperational SUI reported in certain patients was not true SUI, but was actually paradoxical stress incontinence, that is, the loss of retained urine in the diverticulum during coughing. In conclusion, in patients with UD and concomitant SUI, diverticulectomy with anti-incontinence surgery is accepted and performed by most clinicians; however, staged operation is another alternative that may prevent unnecessary treatment.
131
Conflicts of interest The authors declare that they have no financial or non-financial conflicts of interest related to the subject matter or materials discussed in the manuscript.
Sources of funding No funding was received for the work described in this article.
References 1. Hey W. Practical observations in surgery: illustrated with cases. Philadelphia: James Humphreys; 1805. 2. Davis HJ, Cian LG. Positive pressure urethrography: a new diagnostic method. J Urol 1952;68:611e6. 3. Romanzi LJ, Groutz A, Blaivas JG. Urethral diverticulum in women: diverse presentations resulting in diagnostic delay and mismanagement. J Urol 2000;164:428e33. 4. Reeves FA, Inman RD, Chapple CR. Management of symptomatic urethral diverticula in women: a single-centre experience. Eur Urol 2014;66(1):164e72. 5. Singla P, Long SS, Long CM, Genadry RR, Macura KJ. Imaging of the female urethral diverticulum. Clin Radiol 2013;68:e418e25. 6. Thomas AA, Rackley RR, Lee U, Goldman HB, Vasavada SP, Hansel DE. Urethral diverticula in 90 female patients: a study with emphasis on neoplastic alterations. J Urol 2008;180:2463e7. 7. Ganabathi K, Leach GE, Zimmern PE, Dmochowski R. Experience with the management of urethral diverticulum in 63 women. J Urol 1994;152:1445e52. 8. Lee UJ, Goldman H, Moore C. Rate of de novo stress urinary incontinence after urethral diverticulum repair. J Urol 2008;71(5):849e53. 9. Faerber GJ. Urethral diverticulectomy and pubovaginal sling for simultaneous treatment of urethral diverticulum and intrinsic sphincter deficiency. Tech Urol 1998;4:192e7. 10. Bass JS, Leach GE. Surgical treatment of concomitant urethral diverticulum and stress incontinence. Urol Clin North Am 1991;18:365e73.