Journal of Andrology
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Published-Ahead-of-Print July 3, 2008, DOI:10.2164/jandrol.108.005496
Journal of Andrology, Vol. 29, No. 6, November/December 2008
Copyright © American Society of Andrology
DOI: 10.2164/jandrol.108.005496

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Laparoscopic Management of Müllerian Duct Remnants: Four Case Reports and Review of the Literature

JUN-HANG LUO*, WEI CHEN*, JUN-JIE SUN{dagger}, DAN XIE{ddagger}, JIA-CONG MO{dagger}, LI ZHOU{dagger} AND JIAN LU*

From the Departments of * Urology and {dagger} Paediatric Surgery, the First Affiliated Hospital, and the {ddagger} State Key Laboratory of Oncology in South China, Cancer Center, Sun Yat-sen University, Guangzhou, China.

Correspondence to: Wei Chen, Department of Urology, the First Affiliated Hospital, Sun Yat-Sen University, No. 58, ZhongShan 2nd Road, Guangzhou, China (e-mail: chenw3{at}mail.sysu.edu.cn).
Received for publication April 2, 2008; accepted for publication July 1, 2008.

   Abstract
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 Abstract
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 Results
 Discussion
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Several experiences with laparoscopic management of müllerian duct remnants (MDRs) have been reported to date. This report outlines our experience and reviews the present literature to evaluate the results of laparoscopic excision of MDR. Between April 2003 and December 2007, 4 male patients (age range, 6–20 years) with MDR underwent laparoscopic excision in our center. A literature search revealed 5 additional reports of laparoscopic excision of MDR, comprising 13 patients (age range, 1.5–48 years). For the 4 patients in our center, the operative time was 135–200 minutes (mean, 159 minutes) and the estimated blood loss was 20–100 mL (mean, 48 mL). For the 13 patients in the literature reviewed, the operative time was 105–360 minutes (mean, 190 minutes) and the estimated blood loss was 50–200 mL. All of these 17 patients recovered uneventfully with no complications. A small prostatic diverticulum had been found in 1 case at 6 years postoperatively; no recurrent evidence or voiding dysfunction had been found in the other 16 patients during 3–50 months of follow-up. No patient had required any further operative therapy. Laparoscopic excision of MDR is a safe and effective surgical procedure, associated with minimal invasion, minimal postoperative morbidity, and rapid recovery for the patient.

     Key words: Laparoscopy, prostatic utricle cyst, urogenital abnormality



In the male, the müllerian ducts regress under the effect of müllerian inhibiting factor (MIF) produced by the Sertoli cells of the testicles at about the 10th week of fetal life. A müllerian duct remnant (MDR) is a midline prostatic cystic lesion that results from incomplete müllerian duct regression caused by failure of the effectiveness of MIF or incomplete masculinization of the urogenital sinus. MDR is associated with hypospadias or intersex disorders in 90% of cases (Ritchey et al, 1988). Although most MDRs are asymptomatic, these may be associated with recurrent urinary infections or epididymitis, hematospermia, infertility, dysuria, urinary retention, perineal pain, hematuria, and neoplastic degeneration (Coppens et al, 2002). MDR is usually interchangeably called müllerian duct cyst or prostatic utricle cyst (Schuhrke and Kaplan, 1978; Hendry and Pryor, 1992). Surgical excision is considered to be the best form of treatment. Many open surgical approaches have been described. Laparoscopic approach to excise MDR has been recommended recently (Willetts et al, 2003; Lima et al, 2004). The purpose of this report is to describe our experience with laparoscopic excision of MDR and to review the literature in order to provide a comprehensive assessment of the laparoscopic approach to MDR.


   Method
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Patients

Between April 2003 and December 2007, 4 male patients with MDR underwent laparoscopic excision of MDR in our center. The age range was 6–20 years (mean, 11.8). All of these 4 patients had a history of hypospadias and/or cryptorchidism, and had previously undergone urethroplasty and/or orchiopexy. Ultrasonography, computed tomography, or magnetic resonance imaging revealed cystic lesions of variable size behind the bladder and above the prostate (Figure 1). Retrograde ureterogram showed the lesions of these 4 patients communicating with the urethra (Figure 2). The baseline clinical variables are listed in Table 1.


Figure 1
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Figure 1. Computed tomography scan showing midline round cyst behind the bladder and above the prostate.

 

Figure 2
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Figure 2. Müllerian duct remnant (MDR) detected by retrograde ureterogram and the cystic lesion communicating with the urethra.

 

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Table 1. Four MDR patients in our center with laparoscopic excision
 

A literature search revealed 5 additional reports of laparoscopic management of MDR, comprising 13 patients (age range, 1.5–48 years). The patients' records included the time of the operative procedure, the estimated blood loss, the time of urethral catheter removal, the time the patient was discharged, and any immediate or long-term complications (Table 2).


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Table 2. Thirteen patients in 5 other studies reported with laparoscopic excision of MDR
 

Laparoscopic Technique

General anesthesia was administered by tracheal cannulation. The patient was placed in the supine position with the head about 15° lower. A 5-port transperitoneal technique was used. The primary 10-mm trocar for the laparoscope was placed at 1 cm below the umbilicus. Two 5- or 10-mm trocars were placed 2–3 cm below the umbilicus on the lateral pararectal lines bilaterally. Two 3- or 5-mm trocars were positioned 2–3 cm superior and medial to anterosuperior iliac spines bilaterally. The peritoneal reflection was incised behind the bladder. The prostatic utricle was grasped and carefully dissected free of the surrounding tissues of the retrovesical space. Particular attention was given to avoid injury to the bladder neck, ureters, vas deferens, seminal vesicles, prostate, and rectum. After dissection was complete, the neck of the cyst was ligated with 2 endoscopic loops or Hem-o-Lok clips (Weck Closure Systems, Research Triangle Park, North Carolina) and excised (see Supplemental Figure 1, available online at www.andrologyjournal.org). The excised MDR was removed through a 10-mm port, the pelvic drain was placed through a 5-mm port site, and the ports were closed as usual.


   Results
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 Abstract
 Method
 Results
 Discussion
 References
 
For the 4 patients in our center, the mean operative time was 159 minutes (range, 135–200 minutes) and the mean estimated blood loss was 48 mL (range, 20–100 mL). All patients recovered uneventfully with no complications. The urethral catheters were removed and the patients were discharged home 6–8 days after operations. The final pathology reports were MDR. A small prostatic diverticulum had been found in 1 case at 6 years postoperatively. No recurrent evidence or voiding dysfunction had been found at 3–50 months of follow-up (Table 1).

For the 13 patients in the reviewed literature, the operative time was 105–360 minutes (mean, 190 minutes) and the estimated blood loss was 50–200 mL. There have been no major or minor complications. A small prostatic diverticulum was found in 1 case at 6 years postoperatively (McDougall et al, 2001); there was no recurrent evidence found in the other 12 patients at 2–48 months follow-up. No patient required any further operative therapy (Table 2).


   Discussion
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 Abstract
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MDR is usually interchangeably called müllerian duct cyst or prostatic utricle cyst. The prostatic utricle is usually a tubular-shaped structure communicating with the urethra that is frequently observed in younger patients in association with hypospadias or intersex disorders. The müllerian duct cysts are round and variable in size, are associated with normal external genitalia, and usually do not communicate with the urethra (Schuhrke and Kaplan, 1978; Ritchey et al, 1988; Hendry and Pryor, 1992). The distinction between these 2 cysts is considered not necessary, because the symptoms, anatomical location, and primary treatment are the same (Dik et al, 1996).

MDR is not common. Moore (1937) reported a nearly 1% incidence of these cysts at autopsy, and Slocum (1954) identified enlarged utricles in 4% of newborns and 1% of adult males. Clinical symptoms reported include recurrent infections or epididymitis, irritative problems (frequency, dysuria, and urgency), obstructive problems (hesitance, a decreased urinary flow rate, prolonged voiding, urinary retention, and stone formation) or less frequently terminal hematuria, urethral discharge, perineal and abdominal pain, a palpable mass, constipation, impotence, azoospermia, and infertility (Ritchey et al, 1988; Lima et al, 2004). Furthermore, a 3% incidence of neoplasia arising in the MDRs has been reported, with a peak incidence at around the fourth decade of life. Adenocarcinoma and squamous cell carcinoma are the 2 reported neoplastic cell types occurring in MDRs (Youngson, 1990; Gilbert et al, 1992).

Many surgical techniques have been proposed to treat MDR. Endoscopic cyst catheterization and aspiration, cyst orifice dilation, and resection of cyst roof have been reported; these managements have the advantage of minor invasion but are associated with high recurrence rate (Schuhrke and Kaplan, 1978). To date the use of endoscopic treatment has been limited to unroofing infected and obstructed cysts. Complete excision is recommended to prevent recurrent infection or future neoplastic changes. Many different open surgical approaches have been described to excise MDR, including transperitoneal (Tank and Hatc, 1986), retropubic or suprapubic extravesical (Ikoma et al, 1986), transvesical transtrigonal (Okur and Gough, 2003), posterior sagittal transanorectal (Siegel et al, 1995), anterior sagittal transanorectal (Rossi et al, 1998), perineal (Ikoma et al, 1986), and posterior pararectal (Kuhn et al, 1994) approaches.

Because MDR is often strongly adherent to the prostate and seminal vesicles and posterior bladder, traditionally open surgical approaches are often technically challenging and associated with a high risk of injury to the prostate, vas deferens, seminal vesicles, bladder, urethra, ureters, rectum, external sphincters, and pelvic nerve (Krstic et al, 2001). The perineal and posterior pararectal approaches have been described as effective, but they are difficult procedures that may have significant associated morbidity with a high risk of injury to the external sphincters or the reflex arc between the rectum and striated muscle sphincters (Okur and Gough, 2003). The transperitoneal, retropubic, or suprapubic extravesical approach is generally criticized for its poor exposure when dissecting the distal extent of the MDR in the bladder neck region; these difficult procedures may cause a high risk of damage to the nerves, urethra, ureters, and vas deferens (Lima et al, 2004); furthermore, excision of the lesion is incomplete in 58% of the MDR cases (Schuhrke and Kaplan, 1978). For the transvesical transtrigonal approach, splitting the posterior bladder wall might at least transiently interfere with the function of the trigonal musculature; mild vesicoureteral reflux occurred in 25% of Ikoma's reported cases (Ikoma et al, 1986). The posterior or anterior sagittal transanorectal approach requires thorough bowel preparation before and fasting for 1 week after surgery. If any infection supervenes, a rectourethral fistula will inevitably occur, necessitating a diverting colostomy (Yeung et al, 2001). All of these procedures are accompanied by limited anatomical visualization and potential significant morbidity from iatrogenic damage of adjacent structures. Accomplishing complete removal without damaging the normal structures is a demanding task (Table 3).


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Table 3. Disadvantages of traditional surgical management of MDR
 

McDougall et al (1994) reported the first case of laparoscopic treatment of MDR. Laparoscopic technique offers a clear and close view of the deep pelvic structures that cannot be obtained with an open approach, enabling urologists to precisely perform dissection with less blood loss and reduce the complication rate of injury to the surrounding structures because of the 12- to 15-fold magnification of the surgical field. The common advantages of laparoscopic approach also include minor incision, less postoperative pain, and earlier return to full activity. From 1994 to 2007, the literature search revealed 5 reports comprising 13 patients with laparoscopic excision of MDR. All of these 17 patients (including the 4 in the present report) recovered uneventfully with no complications. No patient has required any further operative therapy.

In conclusion, laparoscopic excision of MDR is feasible for surgeons with advanced laparoscopic techniques. When compared with open surgical approaches, this procedure can afford minimally invasive access to the retrovesical space, provide a clear view of the deep pelvic structures, reduce the incidence of injury to adjacent structures, and decrease the time of recovery.


   References
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 Abstract
 Method
 Results
 Discussion
 References
 
Coppens L, Bonnet P, Andrianne R, de Leval J. Adult müllerian duct or utricle cyst: clinical significance and therapeutic management of 65 cases. J Urol. 2002; 167: 1740 –1744.[CrossRef][Medline]

Dik P, Lock TM, Schrier BP, Zeijlemaker BY, Boon TA. Transurethral marsupialization of a medial prostatic cyst in patients with prostatitis-like symptoms. J Urol. 1996; 155: 1301 –1304.[CrossRef][Medline]

Gilbert RF, Ibarra J, Tansey LA, Shanberg AM. Adenocarcinoma in a müllerian duct cyst. J Urol. 1992; 148: 1262 –1264.[Medline]

Hendry WF, Pryor JP. Müllerian duct (prostatic utricle) cyst: diagnosis and treatment in subfertile males. Br J Urol. 1992;69: 79 –82.[CrossRef][Medline]

Ikoma F, Shima H, Yabumoto H, Mori Y. Surgical treatment for enlarged prostatic utricle and vagina masculina in patients with hypospadias. Br J Urol. 1986; 58: 423 –428.[Medline]

Krstic ZD, Smoljanic Z, Micovic Z, Vukadinovic VJ. Surgical treatment of the Müllerian duct remnants. Pediatr Surg. 2001; 36: 870 –876.[CrossRef]

Kuhn EJ, Skoog SJ, Nicely ER. The posterior sagittal pararectal approach to posterior urethral anomalies. J Urol. 1994; 151: 1365 –1367.[Medline]

Lima M, Aquino A, Dòmini M, Ruggeri G, Libri M, Cimador M, Pelusi G. Laparoscopic removal of müllerian duct remnants in boys. J Urol. 2004;171: 364 –368.[CrossRef][Medline]

McDougall EM, Afane JS, Dunn MD, Shalhav AL, Clayman RV. Laparoscopic management of retrovesical cystic disease: Washington University experience and review of the literature. J Endourol. 2001; 15: 815 –819.[CrossRef][Medline]

McDougall EM, Clayman RV, Bowles WT. Laparoscopic excision of müllerian duct remnant. J Urol. 1994; 152: 482 –484.[Medline]

Moore RA. Pathology of the prostatic utricle. Arch Pathol. 1937;23: 517 –518.

Okur H, Gough DC. Management of müllerian duct remnants. Urology. 2003;61: 634 –637.[CrossRef][Medline]

Ritchey ML, Benson RC, Kramer SA, Kelalis PP. Management of müllerian duct remnants in the male patient. J Urol. 1988;140: 795 –799.[Medline]

Rossi F, De Castro R, Ceccarelli PL, Dòmini R. Anterior sagittal transanorectal approach to the posterior urethra in the pediatric age group. J Urol. 1998; 160: 1173 –1177.[CrossRef][Medline]

Schuhrke TD, Kaplan GW. Prostatic utricle cysts (müllerian duct cysts). J Urol. 1978; 119: 765 –767.[Medline]

Siegel JF, Brock WA, Peña A. Transrectal posterior sagittal approach to prostatic utricle (müllerian duct cysts). J Urol. 1995;153: 785 –787.[CrossRef][Medline]

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