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CASE REPORT
Year : 2006  |  Volume : 22  |  Issue : 3  |  Page : 262-263
 

Perforate Cowper's syringocele causing acute urinary retention: Report of a case with literature review


Department of Urology, Division of Pediatric Urology, University of Tennessee Health Science Center, Memphis, TN, USA

Correspondence Address:
J B Malcolm
Department of Urology, University of Tennessee Health Science Center, 956 Court Avenue, Room H216 Memphis, TN 38163
USA
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-1591.27637

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   Abstract 

We report a case of acute urinary retention in a 13-month-old boy, secondary to a perforate Cowper's syringocele. Diagnosis was established with voiding cystourethrogram, transperineal ultrasound and urethroscopy; definitive management involved endoscopic unroofing of the syringocele and resolution of the patient's obstructive signs and symptoms followed. Although novel diagnostic and therapeutic options have been reported, standard urethrographic imaging and endoscopic management is appropriate for the majority of symptomatic Cowper's syringoceles.


Keywords: Cowper′s syringocele, pediatric urology, urinary retention


How to cite this article:
Malcolm J B, Jerkins G R. Perforate Cowper's syringocele causing acute urinary retention: Report of a case with literature review. Indian J Urol 2006;22:262-3

How to cite this URL:
Malcolm J B, Jerkins G R. Perforate Cowper's syringocele causing acute urinary retention: Report of a case with literature review. Indian J Urol [serial online] 2006 [cited 2019 Sep 17];22:262-3. Available from: http://www.indianjurol.com/text.asp?2006/22/3/262/27637



   Introduction Top


The incidence of cystic abnormalities of Cowper's duct may be as high as 2.3% based on voiding cystourethrogram (VCUG) series and autopsy studies.[1],[2] However, most are asymptomatic[3] and clinically significant Cowper's syringoceles are rare. The true clinical significance of Cowper's syringocele lies in its potential to cause urethral obstruction. In reported cases, the majority of obstructing Cowper's syringoceles present with the insidious onset of obstructive voiding patterns.[3],[4],[5] To our knowledge, presentation of Cowper's syringocele with acute urinary retention has not previously been reported in a pediatric patient. We review the literature and report on developments in the diagnosis and management of this uncommon problem.


   Case Report Top


A 13 month-old male presented with acute onset of abdominal pain, suprapubic distention and decreased urine output. His past medical history was unremarkable. Physical examination revealed a tender, distended abdomen with palpable bladder fullness. Serum creatinine was 0.4 mg/dL. Sonographic and CT imaging of the abdomen and pelvis demonstrated moderate bilateral hydroureteronephrosis, a distended bladder and anterior displacement of a dilated posterior urethra by a 12 x 13 mm cyst-like structure that was noted on transperineal ultrasound to enlarge during voiding attempts.

A Foley catheter was placed with urine residual amounting to 90cc. After 48 h, further diagnostic imaging was obtained. Repeat ultrasound imaging demonstrated decreased bilateral hydronephrosis. Voiding cystourethrogram images were consistent with infravesical obstruction by a perforate Cowper's syringocele [Figure - 1] and urethroscopic evaluation confirmed this finding. After unroofing the syringocele [Figure - 2], a catheter was left to indwell for 48 h. Following catheter removal, the patient voided without difficulty.


   Discussion Top


Cowper's glands are believed to contribute to semen coagulation and urethral lubrication.[3] The glands are located within the urogenital diaphragm with ducts entering at the ventral surface of the bulbar urethra. In cases of congenital or acquired dilatation of the Cowper's gland ducts, lower urinary tract symptoms may develop. The majority of Cowper's syringoceles are congenital, possibly due to genetic misguidance of molecular mechanisms that result in a disturbance of stromal-epithelial interactions.[6] In adults, symptomatic Cowper's syringoceles are likely acquired, either by trauma or infection to the glandular duct which may cause ostial stenosis and subsequent ductal dilatation.[7],[8] Historically, Cowper's syringoceles have been diagnosed by urethrography and confirmed by endoscopy. More recently, perineal CT and MRI and transperineal ultrasound have demonstrated utility in diagnosing imperforate syringoceles when VCUG and endoscopy have not been diagnostic.[7],[9],[10] We used transperineal ultrasound in the radiographic work-up of our patient, which allowed real time demonstration of the dynamic nature of the syringocele during voiding attempts.

Because many cases of Cowper's syringocele are asymptomatic, the true incidence of Cowper's duct anomalies is unknown. Of those cases that become symptomatic, common presentations include recurrent urinary tract infection, hematuria, enuresis, postvoid dribbling, perineal pain, perineal mass, weak stream and dysuria.[3],[4],[5],[8] Obstructive symptoms are reported not infrequently, however, our report of a case of severe obstruction with acute urinary retention illustrates an exceedingly rare outcome of Cowper's syringocele.

A classification scheme for Cowper's syringoceles has been proposed by Maizels et al , which differentiates simple, perforate, imperforate and ruptured syringoceles based on their urethrographic or endoscopic appearance.[5] An alternative system, proposed by Bevers et al , simply classifies Cowper's syringoceles as either open or closed, on the grounds that this distinction is more clinically significant.[8] In Bevers' classification scheme, open syringoceles are said to account for postvoid incontinence, while closed syringoceles may cause infravesical obstruction.[8] Our case of an open Cowper's syringocele presenting with severe lower tract obstruction suggests that, in fact, a clinical distinction between open and closed syringoceles may not be reliable as the entire spectrum of irritative and obstructive symptoms can occur across all morphologic types.

Regardless of classification, the management of symptomatic Cowper's syringoceles is typically straightforward, with endoscopic marsupialization providing relief from obstructive and irritative lower tract symptoms in most cases. A single report of laparoscopic management of a Cowper's syringocele demonstrated the feasibility of this approach.[9] However, the applicability of this management approach should be quite limited and endoscopic management will likely remain the gold standard for definitive management of symptomatic cases.

 
   References Top

1.Dewan PA. A study of the relationship between syringoceles and Cobb's collar. Eur Urol 1996;30:119-24.  Back to cited text no. 1    
2.Dhillon HK, Yeung CK, Duffy PG, Ransley PG. Cowper's glands cysts: A cause of transient intra-uterine bladder outflow obstruction? Fetal Diagn Ther 1993;8:51-5.  Back to cited text no. 2    
3.Brock WA, Kaplan GW. Lesions of Cowper's glands in children. J Urol 1979;122:121-3.  Back to cited text no. 3    
4.Campobasso P, Schieven E, Fernandes EC. Cowper's syringocele: An analysis of 15 consecutive cases. Arch Dis Child 1996;75:71-3.  Back to cited text no. 4    
5.Maizels M, Stephens FD, King LR, Firlit CF. Cowper's syringocele: A classification of dilatations of Cowper's gland duct based upon clinical characteristics of 8 boys. J Urol 1983;129:111-4.  Back to cited text no. 5    
6.Dunker N, Aumuller G. Transforming growth factor-beta 2 heterozygous mutant mice exhibit Cowper's gland hyperplasia and cystic dilations of gland ducts (Cowper's syringoceles). J Anat 2002;201:173-83.  Back to cited text no. 6    
7.Brandes SB. RE: Cowper's syringocele: Symptoms, classification and treatment of an unappreciated problem. J Urol 2000;164:1666-8.  Back to cited text no. 7    
8.Bevers RF, Abbekerk EM, Boon TA. Cowper's syringocele: Symptoms, classification and treatment of an unappreciated problem. J Urol 2000;163:782-4.  Back to cited text no. 8    
9.Cerqueira M, Xambre L, Silva V, Prisco R, Santo R, Lages R, et al . Imperforate syringocele of the Cowper's glands laparoscopic treatment. Actas Urol Esp 2004;28:535-8.  Back to cited text no. 9    
10.Kickuth R, Laufer U, Pannek J, Kirchner TH, Herbe E, Kirchner J. Cowper's syringocele: Diagnosis based on MRI findings. Pediatr Radiol 2002;32:56-8.  Back to cited text no. 10    


    Figures

  [Figure - 1], [Figure - 2]



 

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    Abstract
    Introduction
    Case Report
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    References
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