Article ID: WMC003428 ISSN 2046-1690

Female Urethral Diverticula: A Review of the Literature

Corresponding Author: Mr. Anthony Kodzo - Grey Venyo, Urologist, Urology Department. North Manchester General Hospital - United Kingdom

Submitting Author: Mr. Anthony Kodzo - Grey Venyo, Urologist, Urology Department. North Manchester General Hospital - United Kingdom

Article ID: WMC003428 Article Type: Review articles Submitted on:31-May-2012, 05:02:46 AM GMT Published on: 31-May-2012, 12:37:04 PM GMT Article URL: http://www.webmedcentral.com/article_view/3428 Subject Categories:UROLOGY Keywords:Female Urethral Diverticulum; Diverticulectomy; -Vaginal Fistula; Voiding Cystourethrogram; Cltrasound Scan; MRI Scan; Virtual Computed Tomography Urethroscopy; Recurrence; Infection. How to cite the article:Venyo A , Gopall A. Female Urethral Diverticula: A Review of the Literature . WebmedCentral UROLOGY 2012;3(5):WMC003428 Copyright: This is an open-access article distributed under the terms of the Creative Commons Attribution License(CC-BY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Source(s) of Funding: None

Competing Interests: None

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Female Urethral Diverticula: A Review of the Literature

Author(s): Venyo A , Gopall A

Abstract diagnosis (over 12 months) have been found to be at high risk of developing postoperative complications. More worryingly, high recurrence rate after surgery and early Background: metastases is the unfortunate outcome of Female urethral diverticulum is a condition which is delayed diagnosis of diverticula-neoplasms. often overlooked and frequently misdiagnosed. It is hoped that as a result of greater awareness of female Conclusions: urethral diverticulum more timely diagnosis of the Female urethral diverticulum is eminently treatable but condition would be made which would result in early there is little standardization of this condition. The appropriate treatment. greatest single improvement in the management of female urethral diverticulum would emanate from more Objective: widespread clinical awareness of the condition and its The objective of this review is to summarize the presentation and the fact that all available presentation, investigation and management of female investigations when used as a result of increased urethral diverticulum. awareness of the condition would lead to early Result of the Literature Review: diagnosis and appropriate early treatment. Delay in Urethral diverticula are more common than is currently the diagnosis of female urethral diverticulum can have being diagnosed. The presenting symptoms can be a significant impact on a patient’s outcome. Patients summarized as , post-micturition Dribbling and with a Dysparaeunia (‘the three Ds’). Patients may also delayed diagnosis (over 12 months) have been found present with other types of lower urinary tract to be at high risk of developing symptoms. More than half of the cases of female postoperative complications. Therefore, by being more urethral diverticula may be palpable on examination. aware of urethral diverticulum With regard to investigation, traditional contrast as a differential diagnosis, clinicians may be able to studies are currently being replaced by advanced- reduce this delay and the imaging procedures including magnetic resonance complications that can occur with the condition. imaging, and virtual computed tomography urethroscopy. These imaging modalities depict a much Introduction greater and better tissue definition; only few studies have directly compared the contrast-based investigative procedures, the various ultrasonography A urethral diverticulum can be described as an techniques, out-pouching of the urethra into the urethrovaginal and cross sectional imaging modalities that are potential space. Those with urethral diverticula are available. With regard to treatment small often misdiagnosed or there is a delay in diagnosis asymptomatic female urethral diverticula may not due to a lack of awareness amongst clinicians about require treatment. There are three options of surgical the condition as well as due to its non specific treatment for urethral diverticulum and these include: presentation. Urethral diverticula are more common in 1. Trans-urethral incision of the urethral women than men and can often present in a urological communication, thus transforming a narrow or gynaecological setting. Complications of the diverticulum into a wide-mouthed diverticulum. condition include recurrent urinary tract infections, 2. Marsupialization of the diverticulum sac into the calculus and malignancy. The latter complication in vagina by incising the urethro-vaginal septum. particular, although uncommon, highlights the 3. Diverticulectomy with or without a reconstructive importance of clinicians including urethral diverticula in surgery. their differential diagnoses. Therefore, the aim of this Delay in the diagnosis of female urethral diverticulum review is to increase awareness of urethral can have a significant impact diverticulum in females. on a patient’s outcome. Patients with a delayed

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Literature Review of Urethral of dysuria (10 to 80%), (10 to 70%) and post-void dribbling (25%). These include frequency Diverticula in Females and urgency (40 to 100%), chronic or recurrent urinary tract infections (30 to 80%), stress or urge incontinence (4 to 70%) and haematuria (10 to 35%). Epidemiology Less frequent symptoms include urethral pain, pelvic or suprapubic pain, an anterior vaginal wall mass, As females with urethral diverticula are often purulent urethral discharge, urinary hesitancy and asymptomatic and the diagnosis is difficult, it is very urinary retention. [11], [12], [13] Physical examination likely the reported prevalence, estimated as 1 to 6% of may reveal very little. On the other hand a mass on women, is an under representation of those with the the posterior aspect of the urethra may be found. This condition [1]. This number interestingly climbs to 16 to may be tender and urine or purulent discharge may be 40% in females with recurrent urinary tract expressed on palpation [2], [14]. Although uncommon, infections.[2] Urethral diverticula are most common in a hard mass should raise suspicion of a urethral women between 30 and 60 years old and are rare in diverticular neoplasm or calculus. Urethral neoplasms children and neonates [3], [4], [5], [6]. In addition, are more often adenocarcinomas. They appeared to surgery for urethral diverticula has been found to be be more prevalent in black people and in a study of higher in black women [7]. patients who had surgery for urethral diverticulum 6% were found to have a malignancy [15], [16]. It has Anatomy and Aetiology postulated that repeated injury to the diverticular wall leads to the re-proliferation that contributes to the The urethra is lined by transitional cell epithelium at development of neoplasms.[17] Calculus formation the bladder neck, becoming squamous epithelium as has been reported in up to 10% of cases.[11] Such the external urethral meatus approaches. calculi may form due to urinary stasis and salt Paraurethral glands (glands of Skene) are most deposition, however they are more commonly found to commonly located medially and posterolaterally along be migratory. the mid and distal third of the urethra. These glands secrete mucous material which empties into the Differential Diagnosis urethra via paraurethral ducts. There are a number of theories regarding the aetiology of urethral diverticula. Differential diagnoses for periurethral masses include The most prominent is that of an acquired origin, vaginal wall inclusion cyst, Skene's gland abscess, which is supported by the anatomy of the urogenital Gartner's duct cyst, ectopic ureterocele, periurethral system. Urethral diverticula are most commonly found fibrosis, urethrocele, vaginal leiomyoma, at the distal two thirds of the urethra, posterior-laterally, endometrioma and urethral or vaginal neoplasm. [18] at the 3 and 9 o clock positions.[8] These are in very In addition, urethral diverticulum should be considered similar locations to the previously mentioned in the differential diagnosis of patients who continue to paraurethral glands. Therefore, it has been postulated re-present with the urogenital symptoms previously that these paraurethral glands become obstructed and discussed. infected leading to the formation of a sub-urethral abscess. This subsequently ruptures into the urethral Investigation lumen creating the diverticulum.[9] Other theories include trauma, for example, from urethral and vaginal 50 to 60% of urethral diverticula are diagnosed by surgery. Childbirth trauma has also been suggested. physical examination therefore confirmation or further However urethral diverticula are common in investigation is often necessary [6],[13]. Magnetic nulliparous women.[6] Congenital urethral diverticula resonance imaging (MRI) is felt to be the most are believed to be rare. [10] sensitive mode of imaging for the diagnosis of urethral diverticulum [19], [20], [21]. MRI offers higher Presentation resolution, providing detailed information regarding number, location, size, configuration, complications, Females with urethral diverticula may be and communication with the urethra of diverticula. In asymptomatic or they can present with vague addition it has the ability to differentiate urethral urogenital symptoms, thus contributing to the pathology from anatomical variants and identify the challenge of diagnosing the disorder. There are a presence of stones or neoplasms [22]. This modality variety of presentations in addition to the classic triad of imaging therefore provides useful information for

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surgical planning. Voiding-Computed-Tomography (CT) A review found excision of the diverticulum to have an urethrography and virtual urethroscopy are overall symptomatic cure rate of 70% with a further-imaging techniques, that provide high recurrence rate of 10 to 20%. [13] From the small diagnostic accuracy. However, they may not always number of reports published, marsupialization be available [23], [24]. If such imaging is not available, procedures have achieved essentially 100% ultrasound is an alternative choice. [11](25, 26, 27, symptomatic cure rate and have very minimal 28) The entire length of the urethra and surrounding complications if limited to distal diverticula. Common tissues can be visualised in the absence of ionizing post operative complications include recurrence of radiation, it can provide detailed information regarding diverticlum (1 to 25%), urethrovaginal fistula (1 to 8%), the diverticulum and identify differential diagnoses. In (1 to 16%), urethral strictures (0 to addition, a study found ultrasound scan to be more 5%) and recurrent urinary tract infections (7 to 31%) sensitive than more traditionally used imaging such as [13], [14], [35]. A high recurrence rate is associated cystoscopy and voiding cystourethrogram with delay in the diagnosis and treatment of female (VCUG).[26] Urethroscopy, balloon positive pressure urethral diverticulum. [36], [37]. urethrograpy, VCUG and retrograde positive pressure urethrography, although historically used, are no Discussion longer recommended for routine investigation of urethral diverticulum. VCUG and retrograde positive pressure urethrography rely on contrast medium Lee and Fynes [2] stated that appropriate entering the lumen of the diverticulum therefore they investigations play a vital role in the diagnosis of are only effective if the opening is sufficiently patent. urethral diverticula and these investigations should Hence they have a low sensitivity, VCUG more so provide the surgeon with information in relation to the than retrograde positive pressure urethrography [2], location, number, size, configuration and [29], [30]. communication of the urethral diverticulum. The investigations should also be able to identify any Management associated malignancy or calculi. Golomb and associates [29] stated that it is equally as important to Patients with mild symptoms can be managed accurately diagnose large urethral diverticula, which conservatively. This usually involves prophylactic would require extensive dissection, as it is to identify antibiotics and monitoring. Digital decompression post difficult-to-diagnose small or non-communicating voiding, periodic needle aspiration and urethral dilation urethral diverticula. Experiences gained by a number has also been suggested. [31] However, these of authors regarding the investigation of female measures only provide symptomatic relief and do not urethral diverticula are summarized as follows: target the underlying cause. There is also little documentation regarding the outcome of conservative Urethroscopy: management in urethral diveticulum. For symptomatic Saito [38] stated that urethroscopy allows direct visual patients there are a number of surgical techniques that inspection of the urethra, and thus identification of the can be used. The approach chosen depends mainly location of the position of the ostia; however, although on the location of the diverticulum along the urethra [2], frequently performed it quite often fails to be helpful, [32]. If the middle or proximal third of the urethra is especially if the urethral diverticulum is collapsed and affected, the treatment of choice is urethral the ostium is not visible. Lee and Fynes [2] stated that diverticulectomy. Transvaginal excision of the urethral urethroscopy is not of much use in assessing the size, diverticulum is considered definitive surgical treatment. or shape of the divericulum, as well as the fact that [33] If the diverticulum is found at the distal third of the any concurrent inflammation may further obscure the urethra, marsupialization into the vagina is an option. visualization. Marsupialization is avoided in the more proximal diverticulum as there is an increased risk of Micturating cysto-urethrogram [Voiding complications. Proximal incision of the urethra can cystourethrogram (VCUG): lead to incontinence due to injury to urethral Lee and Fynes [2] stated that micturating sphincters. Transurethral procedures have also been cysto-urethrogram had been considered the proposed [2], [34]. In order to reduce complications investigation of choice for the identification of urethral acute infection should be treated before surgery. diverticula with a sensitivity of 65%. However, Colomb and associates [29] are of the opinion that micturating Surgical Outcomes cysto-urethrogram is equivocal and additional imaging

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studies are usually necessary. The attached video have been used in the investigation of the female illustrates a cysto-urethrogam which demonstrates a urethra and these include endoluminal [21], small urethral diverticulum. endovaginal [44] endorectal [45] and external coils. Some of the advantages of magnetic resonance Double balloon urethrogram (DBU): imaging include: accuracy of the diagnosis, the fact Lang and Davis [39] stated that VCUG for some-time that patients do not require catheterization and are not was considered the gold standard investigation, required to void for the study, there is no radiation however, it has the potential for urethral injury and exposure involved and the procedure can be there is significant discomfort to the patient and performed in three breath-hold sequences. [46]. additionally it is an invasive procedure which is associated with radiation. Fortunato and associates Virtual computed tomography urethroscopy: [31] found that DBU has the highest accuracy. Computed tomography (CT) urethrography has the Nevertheless, Neitlich and associates [40] reported advantage of being non-invasive and can identify the that other studies have shown greater urethral anatomy and pathology of the extraluminal organs diverticula detection with other modalities of better than urethroscopy. Chou and associates [47] investigation such as magnetic resonance imaging reported a case study in which a horseshoe-shaped (MRI). Jacoby and associates [30] reported a diverticulum with a clearly identified orifice in the prospective study which had compared DBU with mid-urethra. The diverticulum was missed on VCUG in 32 patients with urethral diverticula. They urethroscopy and voiding cystourethrogram. Kim and reported that sensitivity which was confirmed by associates [48] reported two cases with improved surgery of 100% with DBU and 44% with VCUG ability to detect the ostia compared with MRI (P=0.002). They observed that the cases that were missed by VCUG were those of smaller urethral Treatment: diverticula (12.4 mm compared with 24.2 mm; P=0.018). Non Surgical Treatment: Fortunato and associates [31] stated that there is no Urodynamics: need or indication for surgical treatment in cases of Urodynamics is useful for the detection of any asymptomatic urethral diverticula and patients with associated stress incontinence [13], [41], as well as only mild symptoms. They recommended that these any associated lower urinary tract obstruction [42]. two groups of patients should be followed and treated Radiographic pictures that are taken during voiding symptomatically with antibiotics; and a high index of when performing voiding cystourethrogram or suspicion for carcinoma should be present in the event video-urodynamic studies may show urethral of development of any insidious signs. diverticula as some urethral diverticula empty at the end of voiding and would be missed in the event of Surgical Treatment: taking only filling and post-micturition films [43]. Patients with symptoms related to their urethral diverticula require treatment. Scarpero and associates Ultrasound scan: [46] recommended that all cases of acute suppuration Siegel and associates [28] compared voiding and inflammation should require pre-operative short cystourethrogram with the ensuing ultrasound scan course of antibiotics. Greenberg and associates [49] techniques: trans-vaginal, endo-urethral, and also recommended that in the case of presence of trans-perineal. They reported that both modalities of significant infection, incision and drainage may be investigation voiding cysto-urethrogram and required prior to formal surgery; in cases of urinary ultrasound scan detected 13 out of 15 urethral stress incontinence or an open bladder neck in diverticula. The ultrasound scan identified the neck of association with urethral diverticulum a decision is the diverticula in all detected cases but the voiding required whether or not to perform concomitant sling cystourethrogram identified the neck of the surgery. Some authors [13], [41], [50], [51], have diverticulum in only 2 cases. In addition ultrasound reported successful concomitant surgery. However, scan was able to identify nearby disorders which Patel and Chapple [32] tend to treat the urethral included infected peri-urethral cysts and leiomyomas diverticulum initially and later on re-assess the which were missed by voiding cysto-urethrogram. appropriateness for sling insertion. Patel and Chapple [32] observed that in many instances there was no Magnetic Resonance Imaging: need for a sling procedure later. Aspera and Various Magnetic Resonance Imaging techniques associates [11] stated that there are three options of

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surgical treatment for urethral diverticulum and these years.[14] This delay in diagnosis can have a include: significant impact on a patient’s outcome. Patients 1. Trans-urethral incision of the urethral with a delayed diagnosis (over 12 months) have been communication, thus transforming a narrow found to be at high risk of developing postoperative diverticulum into a wide-mouthed diverticulum. complications. More worryingly, high recurrence rate 2. Marsupialization of the diverticulum sac into the after surgery and early metastases is the unfortunate vagina by incising the urethra-vaginal septum. outcome of delayed diagnosis of diverticular 3. Diverticulectomy with or without a reconstructive neoplasms [15], [36], [37]. Therefore, by being more surgery. aware of urethral diverticulum as a differential Patel and Chapple [32] stated that they have not found diagnosis, clinicians may be able to reduce this delay trans-urethral incision to be necessary they instead of and the complications that can occur with the condition. trans-urethral incision allow infected diverticula to settle with antibiotics. Patel and Chapple [32] stated References that marsupialisation of a urethral diverticulum is one of the most common causes of the development of urethro-vaginal fistulae in view of the fact that 1. Antosh D D, Gutman R E. Diagnosis and diverticula usually extend through all layers of the management of female urethral diverticulum. Female urethra. Because of the aforementioned reasons Patel Pelvis Med Reconstr Surg. 2011; 17(6): 264 – 271 and Chapple [32] have adopted their standard 2. Lee J W, Fynes M M. Female urethral diverticula. management plan to perform a diverticulectomy via Best Pract Res Clin Obstet Gynaecol. 2005; 19(6): the vaginal approach in the prone position, adopting a 875 – 893 technique to that which was reported by Leach and 3. Smith N A. Treatment approach to female urethral associates [52]. diverticulum. Rev Col Bras Cir. 2011; 38(6): 440 – 443 Complications of trans-vaginal urethral 4. Silk M R, Lebowitz J M. Anterior urethral diverticulectomy: diverticulum. J Urol. 1969; 101(1): 66 – 67 Porpiglia and associates [37] stipulated that the risk 5. Andersen M J. The incidence of diverticula in the factors for the development of post-operative female urethra. J Urol. 1967; 98(1): 96 – 98 complications emanating from trans-vaginal urethral 6. Davis H J, Telinde R W. Urethral diverticula: an diverticulectomy include: assay of 121 cases. J Urol. 1958; 80(1): 34 – 39 1.Delayed diagnosis (> 12 months), 7. Burrows L J, Howden N L, Meyn L, et al. Surgical 2. Size (> 4 cm) procedures for urethral diverticula in women in the 3. Complex configuration (e.g. horseshoe shape) United Stated, 1979 – 1997. Int Urogynecol J Pelvic Patel and Chapple [32] stated that common Floor Dysfunct. 2005; 16(2): 158 – 161 complications arising from trans-vaginal 8. Mackinnon M, Pratt J H, Pool T L. Diverticulum of diverticulectomy include: , the female urethra. Surg Clin (1.7-16.1%), urethro-vaginal fistula (0.9 – 8.3%), North Am. 1959; 39(4): 953 – 962 urethral stricture (0-5.2%), recurrent urethral 9. Leach G E, Bavendam T G. Female urethral diverticula (1-25%), and recurrent urinary tract diverticula. Urology 1987; 30(5): 407 – 415 infections (0-31.3%). Aspera and associates [11] 10. Jiledar Rawat, Tanvir Roshan Khan, Sarita Singh, stated that the discovery of a urethral diverticulum Madhukar Maletha, Shivnarain Kureel Congenital pursuant to a presumably successful diverticulectomy anterior urethral valves and diverticula: Diagnosis and may occur as a result of a new diverticulum, or, management in six cases. African Journal of alternatively as a result of recurrence. They postulated Paediatric Surgery 2009; 6(2): 102-105 that recurrence of a urethral diverticulum may be as 11. Aspera A M, Rackley R R, Vasavada S P. result of incomplete excision, active infection, difficult Contemporary evaluation and management of dissection, inadequate or excessive suture line tension, the female urethral diverticulum. Urol Clin North Am. residual dead space or due to other technical factors. 2002; 29(3): 617 – 624 Aspera and associates [11] stated that repeat 12. Bennett S J. Urethral diverticula. Eur J Obstet diverticulectomy could be carried out if necessary. Gynecol Rep Biol. 2000; 89(2): 135 – 139 Conclusions: 13. Ganabathi K, Leach G E, Limmem P E, et al. Urethral diverticula are more common than is currently Experience with the management of urethral being diagnosed. A recent study found that the mean diverticulum in 63 women. J Urol. 1994; 152 (5 Pt1): time from onset of symptoms to diagnosis was 5.2 1445 – 1452

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