European Journal of Obstetrics & Gynecology and Reproductive Biology 89 (2000) 135–139
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Review
Urethral diverticula S.J. Bennett MB BS, MRCOG* Senior Registrar, Dept. Obstetrics and Gynaecology, Leicester Royal Infirmary, Leicester, LE1 5 WW, UK Received 25 January 1999; received in revised form 15 June 1999; accepted 9 August 1999
Abstract Urethral diverticula are a common cause of chronic genitourinary symptoms in women. They occur in three percent of women overall with higher frequencies in selected populations of symptomatic women. The peak incidence is between the ages of 25–45 but they affect all ages. The classical presentation is with recurrent urinary tract infections and post micturition dribbling but almost any urinary symptom may be a presenting feature. Reported cure rates following surgery approach 70% for recurrent urinary tract infection and almost 100% for local symptoms such as dyspareunia. However, despite this and the availability of effective diagnostic techniques diagnosis is often delayed. This is partly due to a lack of awareness among clinicians and partly because the condition overlaps the traditional territories of gynaecologists and urologists. 2000 Elsevier Science Ireland Ltd. All rights reserved.
1. Introduction Urethral diverticula have long been recognised as a cause of genitourinary symptoms in women. Following the first case report in 1803 [1] sporadic reports of isolated cases appeared until early this century when the first case series were published. They classically present with a history of repeated urinary tract infection and postmicturition dribbling together with a palpable suburethral mass on vaginal examination and the discharge of pus from the urethral meatus on milking the urethra. However, this classical presentation is only found in a minority of cases, and the diagnosis is as a result often overlooked. Many symptomatic women give long histories of repeated erroneous investigations and ineffective treatments sometimes from specialist urological or gynaecological clinics. The frequency with which the diagnosis of this ’Cinderella’ condition is made seems to be more closely related to the level of clinical awareness than to the true underlying incidence. This is shown by reports of dramatic increases in cases diagnosed at different centres following *Address for correspondence: Ladywell Unit, North Devon District Hospital, Raleigh Park, Barnstaple, North Devon, EX31 4JB. Tel.: 1441271-322784.
initial reports of surgical series. In one report 71 cases had been diagnosed over 60 years, followed by a further 50 in the next 12 months [2]. A high index of suspicion, a careful examination and referral for appropriate investigation in all patients with urinary symptoms will improve the number correctly diagnosed and lead to considerable benefit since most symptomatic cases can be cured by appropriate surgery.
2. Aetiology A number of different theories have been advanced as to the underlying mechanism leading to formation of diverticula. The most popular first proposed in 1890 [3] is that an obstructed paraurethral gland duct becomes dilated with secretions and if secondary infection supervenes a suburethral abscess may rupture into the urethra creating the diverticulum. This theory is supported by the demonstration of small dilatations and cysts in the paraurethral ducts and glands in the normal female urethra [4], from the position of urethral diverticula which coincides with the position of the paraurethral glands. In two thirds of cases diverticula lie dorsolateral to the urethra [5] and the ostia open into the lower two thirds of the urethra in 85% [6],
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S. J. Bennett / European Journal of Obstetrics & Gynecology and Reproductive Biology 89 (2000) 135 – 139
the paraurethral glands lie in a network around the lower urethra and are less common proximally [4]. Finally diverticula are more common in patients with previous gonococcal infection [7]. Cultures from urethral diverticula have not demonstrated the gonococcus [8–11] but since most symptomatic patients will have received many courses of antibiotics prior to diagnosis this does not rule out past infection. An alternative theory is that diverticula are congenital. Suburethral cysts have been reported in the newborn [12] and there have been cases where the diverticulum was the site of opening of an ectopic ureter [2,13]. In yet other cases diverticula have been lined with colonic epithelium [11,14] suggesting that they may arise from cloacogenic rests during development. Other possible mechanisms for congenital diverticula include development from Gartners duct cysts, Mullerian duct cysts, cysts arising from faulty union of primal folds in the embryo or arising directly as congenital dilatations of the paraurethral ducts or glands [4]. Nevertheless relatively few cases are probably congenital since diagnosis before the age of 20 is unusual [2], even allowing for the fact that the onset of symptoms typically occurs 5 years before diagnosis [2,15,16]. A third possibility that they may arise as a result of trauma and infection at childbirth is less probable since significant numbers of nulliparous women are reported in most series [2,16,17] and urethral diverticula are no more common in women of high parity [2].
3. Incidence In the largest study of asymptomatic women 300 such women being treated for cervical carcinoma underwent positive pressure urethrography [18]. Six had one diverticula and three women had two, an overall prevalence of 3%. The size varied from 2–16 mm in diameter. In another series of 129 asymptomatic women 6 diverticula were identified giving a similar figure of 4.7% [8]. In contrast smaller studies of women with recurrent urinary infection have shown prevalence rates of 40% and 16% [5,16].
4. Presentation It is difficult to estimate the frequency of various presenting symptoms because most series are surgical and biased as to the signs or symptoms which are considered to warrant investigation. Patients with ’atypical’ symptoms may not be investigated and hence remaining undiagnosed and patients with chronic symptoms may experience long symptom free remissions [2]. This explains the discrepancies in incidences of various presenting symptoms in different publications. Given that three percent of the
population have asymptomatic diverticula it is possible that most cases produce no symptoms at all. With these provisos the commonest reported symptoms are urinary frequency and urgency in 40–100%, dysuria in 30–70%, recurrent urinary tract infection in 30–50%, post micturition dribbling in 10–30%, dyspareunia in 10–25%, and haematuria also in 10–25%. Other less common symptoms include stress or urge incontinence, suprapubic pain, hesitancy, perineal pain, pelvic pressure, vaginal or urethral discharge, urethral tenderness, incomplete voiding, urinary retention and terminal dysuria or haematuria [2,4,7–9,15,17,19–21]. The frequency of the classical finding of a palpable suburethral mass with expression of pus from the urethral meatus on milking the urethra appears to have fallen recently from 79% of cases in a 1958 series [2] to only 2–6% in the series from the 1970’s [7,16]. It is uncertain whether this represents changing diagnostic habits or is a true reflection of a change in presentation with time, perhaps related to increased use of antibiotics.
5. Investigations Many diagnostic techniques are imprecise but the gold standard for many years has been positive pressure urethrography using a double balloon catheter. This was first described in 1956 [22] and involves passing a modified blind ending catheter into the bladder. The catheter balloon is inflated to occlude the upper end of the urethra, and a second sliding balloon is then moved along the catheter to abut the external urethral meatus and inflated to occlude the lower end of the urethra. Contrast medium is then passed along the lumen of the catheter and exits from side ports within the urethral portion to delineate the urethra and any connections. Using this technique urethral diverticula, outpouches, fistulae and periurethral glands can all be demonstrated [5]. The development of this technique was a major advance in diagnosis with a reported accuracy of 90% [15,23], and many authors advocate it as the screening method of choice [2,15]. However some patients find it uncomfortable and the results may be difficult to interpret if the opacified catheter overlies small diverticula or their urethral connections [5]. One alternative, cystourethroscopy, appears in general to be much less precise than conventional radiographic techniques [2,5,11,19,24] Indeed some reported series take as their starting point symptomatic patients with negative urethroscopic findings [16]. In other hands however, diagnostic accuracies of 71–100% have been reported [9,15,25–27] and there is general agreement that this investigation has a role in the preoperative assessment of patients with proven diverticula to assess the level of the diverticular orifice. Another alternative, voiding cystourethrography has a reported diagnostic accuracy between 65–100% [7,23,25].
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Some authors advocate it as the primary procedure reserving double balloon urethrography for patients with a strong clinical suspicion but negative voiding cystourethrography [20,25]. Other authors have taken this a stage further and have abandoned all other radiological diagnostic methods in favour of voiding cystourethrography [24]. Of the non invasive imaging modalities ultrasound scanning avoids radiation exposure is quick, painless, cheap, readily available and allows both good visualisation of diverticula and examination of any contents such as stones, polyps or potential malignancy. Pulsed colour doppler can also be used for further investigation of suspected malignancies [28] and ultrasound is particularly effective for distinguishing between multiple diverticula and a single large septated one [29]. The earliest technique using transabdominal scanning [30] has been discarded because of the poor image quality. Endolumenal scanning [31] with the probe in the urethra can be done intraoperatively to assist surgery but is at present only a research technique. Transrectal [32,33], translabial [28], transperineal [29] and endovaginal [29,34] scanning all have their advocates and can all effectively demonstrate urethral diverticula including some missed by conventional investigations [31,34]. Ultrasound may become the primary investigation of choice but to date reported series are all small, no large comparison with voiding cystourethrography or positive pressure urethrography, has been reported and at best ultrasound can rarely either identify the neck of the diverticula or differentiate between diverticula and other cystic structures such as Gartners duct cysts, inclusion cysts or abscesses of Skenes glands. Where an ultrasound abnormality is identified therefore, further investigations will be required. Magnetic resonance imaging (MRI) can also demonstrate diverticula [26,27,35] and often distinguish them from other paraurethral cysts, but it is less reliable at differentiating multiple diverticula from septated ones [26,27]. Like ultrasound MRI cannot reliably identify diverticular ostia. A completely fresh diagnostic approach using the urethral pressure profile was suggested in 1981. A notch or depression in the normal bell shaped pressure profile curve is associated with a diverticulum [36] and may correspond to the area of damage of the supporting paraurethral tissues around the ostium. Attempts have been made to try to utilise this as a means of distinguishing between diverticula opening distal to the site of maximal urethral pressure (the urethral sphincter) and those opening either at the same level or proximally. The assumption being that the former group would be suitable for marsupialisation whereas in the latter group this would risk causing postoperative incontinence. In practice the usefulness of urethral pressure profiles is limited by the fact that the pressure depression usually involves a considerable portion of the urethra (29–58% of the total and 41–64% of the
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functional length) [25], that biphasic profiles have been demonstrated in patients without diverticula [25], and may occur in only 72% of patients with proven diverticula [20]. At present urethral pressure profilometry has little place in preoperative assessment of patients with known diverticula nor is it a useful screening test. Nevertheless identification of a biphasic profile in a patient undergoing routine urodynamic investigation certainly justifies further investigation.
6. Treatment Traditional treatment is surgical excision but even under optimal conditions with small turgid and distally located lesions such operations may be both frustrating and challenging. Infection may have obliterated planes of dissection, intraoperative bleeding can make visualisation difficult and tearing of fragile tissue may lead to incomplete excision and recurrence. Dissection is hampered if the sac ruptures or if the diverticulum is flaccid due to free passage of the contents through the ostium into the urethra. If the diverticulum opens proximal to the urethral sphincter development of a postoperative urethrovaginal fistula will cause urinary incontinence. Nevertheless surgery offers a 70% cure rate for symptomatic patients [2,16] and there is little alternative conservative management. The standard diverticulectomy operation utilises a vertical midline incision in the vaginal wall overlying the diverticulum through which the diverticulum is freed by a combination of sharp and blunt dissection, and removed. The urethral ostium is closed with interrupted sutures and the paraurethral tissue closed over the suture line before closing the vaginal epithelium. The paraurethral tissues and vaginal epithelium may be sutured in an overlapping fashion or alternatively, an inverted U vaginal incision placed so that the diverticulum lies behind the flap [11] may reduce the risk of postoperative fistula. An alternative approach avoiding incising the vaginal skin at all uses a semilunar submeatal incision to approach the diverticulum by dissection along the urethra [21]. Reported complications with this approach are very low and if confirmed this may become the operation of choice. The commonest complications are recurrent symptoms due to incomplete excision or development of new diverticula (10–20% of cases [2,9,15,17]), postoperative stress incontinence (up to 5% of cases [11,15,19]) and urethrovaginal fistula (up to 5% of cases [7– 9,11,15,17,19]). Urethrovaginal fistula distal to the urethral sphincter whilst undesirable does not of course affect continence. Other reported problems include postoperative urethral strictures [9,19] and inadvertent opening of the bladder [15]. These problems have led to many different operations and modifications such as insertion of a Foleys catheter into the diverticulum via a transvaginal stab incision [13]
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to aid in intraoperative dissection or preoperative injection of silastic via an intra urethral catheter similarly [37]. None have shown conspicuous improvements in success or complication rates. Conservative surgical measures such as transurethral dilation of the diverticular ostia and incision and drainage via the vagina are ineffective. However two particular conservative procedures are useful. The first approach is to enter the cavity of the diverticulum via a transvaginal stab incision, irrigate with antiseptic and pack with oxycel before closing the vaginal incision with interrupted sutures [38]. The cavity becomes obliterated with fibrosis and despite no attempt being made to close the ostium does not cause postoperative fistula. This method has been recommended where extensive ramifications of a diverticulum in the trigonal area make traditional surgery unduly risky [10]. The second useful conservative approach is masupialisation, this is a refinement of an older operation which involved division of the urethra from the external meatus to the diverticulum followed by diverticulectomy and urethroplasty [6]. Marsupialisation involves the insertion of one blade of a pair of straight bladed scissors into the urethra and dividing the posterior urethral wall and anterior vaginal wall down to the diverticular ostium. The diverticular wall is then divided in the midline together with the overlying vaginal epithelium. A continuous locking suture is then employed to oppose the epithelial margins of the diverticulum and the vaginal skin to create a saucering of the diverticulum [24,39]. This technique is quick, avoids any difficult dissection, has low complication rates and is applicable to the majority of cases where diverticula arise from the distal portion of the urethra. It can be applied equally well to simple or loculated diverticula but is contraindicated if the diverticula arise from the proximal urethra or are multiple. Whilst the outcome of surgery is not closely related to the precise technique, it is highly dependant on accurate preoperative assessment [15]. Approximately 10% of diverticula have multiple ostia [8,9,19], all of which will need to be closed, and multiple diverticula are found in 26% of cases [23]. A precise assessment of the number of diverticula, their relationship to the bladder neck and the urethral sphincter is essential to plan the most appropriate surgical approach.
7. Conclusion The natural history of urethral diverticula as well as their true prevalence and incidence remains uncertain but they are probably more common than generally supposed and should be suspected whenever a patient presents with chronic genitourinary symptoms with no obvious cause. A high index of suspicion will expedite early diagnosis. The most accurate diagnostic tests appear to be positive pres-
sure urethrography and micturating cystourethrograms although developments in ultrasound may soon supersede these. There is an urgent need for proper studies to compare these diagnostic tests, to evaluate the treatment options available and measure long term outcome. Whilst surgical treatment appears to be effective, preliminary workup must assess the size, site, and number of diverticula present, to allow an appropriate surgical approach and reduce complications. Surgery is most effective at relieving local symptoms such as dyspareunia and urethral discomfort but less effective for chronic urinary symptoms. In view of the risks of surgery asymptomatic diverticula encountered incidentally should be left untreated.
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