Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 33  |  Issue : 1  |  Page : 24-27

Leiomyoma of the bladder neck


1 Department of Urology, Creta InterClinic HHG Group, Heraklion, Greece
2 Department of Pathology, Hygeia Hospital, Athens, Greece
3 Department of Urology, Creta InterClinic HHG Group; Urological Office, Iatriko Kritis (Affidea), Heraklion, Greece

Date of Submission10-Mar-2021
Date of Decision31-Mar-2021
Date of Acceptance12-Apr-2021
Date of Web Publication15-Feb-2022

Correspondence Address:
Kostas Chondros
Urological Office, Iatriko Kritis (Affidea), Eleftherias Sq. 45, Heraklion, Crete
Greece
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/HUAJ.HUAJ_10_21

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  Abstract 


Benign bladder tumors represent a rare entity of bladder neoplasms and include tumors such as leiomyomas. Female preponderance is characteristic, and patients usually present with bladder outlet obstruction, dysuria, hematuria, and urinary tract infections; symptoms that are not disease specific and can be overlooked. Bladder leiomyomas can be easily diagnosed during imaging tests or/and cystoscopy evaluation and they are mainly treated with transurethral surgical excision. We present a case of a female patient with urinary obstruction due to missed on previous ultrasound examinations large leiomyoma located at the bladder neck and expanded into the proximal urethra.

Keywords: Benign bladder tumor, bladder leiomyoma, bladder obstruction, transurethral resection


How to cite this article:
Gorgoraptis P, Papaioannou D, Kazanis I, Chondros K. Leiomyoma of the bladder neck. Hellenic Urology 2021;33:24-7

How to cite this URL:
Gorgoraptis P, Papaioannou D, Kazanis I, Chondros K. Leiomyoma of the bladder neck. Hellenic Urology [serial online] 2021 [cited 2022 May 22];33:24-7. Available from: http://www.hellenicurologyjournal.com/text.asp?2021/33/1/24/337775




  Introduction Top


Benign bladder tumors represent only 1% of all bladder neoplasms.[1] They mainly consist of papillomas, leiomyomas, hemangiomas, neurofibromas, and lipomas. Patients typically present with urinary tract infections, pain, and voiding dysfunction caused by obstruction and rarely gross hematuria. Endoscopic evaluation and transurethral excision are usually mandatory. Women are at higher risk of developing such tumors than men, especially leiomyomas.[2] We present a case of a female patient who was diagnosed with a large bladder leiomyoma located in the right aspect of the bladder neck, expanded to the urethra and causing a valve-like outflow obstruction. The patient remained undiagnosed for several months perhaps due to the unusual tumor location.


  Case Report Top


A 61-year-old female patient presented with lower urinary tract (LUT) symptoms indicating urinary infection. The patient was afebrile, and the urine tests revealed typical cystitis caused by Escherichia coli which was treated with a short-term course of oral fosfomycin due to resistance to other common antibiotics. The patient had a significant surgical history including thyroidectomy, tonsillectomy, appendicectomy, double cesarean incisions, and several uterine curettages and a medical history of hypertension and chronic pulmonary disease. She was also a heavy smoker (approximately 40 pack-years). The clinical examination was normal, and no urethral pathology, vaginal lesions, or pelvic organ prolapse were present. Furthermore, the patient reported several urinary infections in the past 2 years and prolonged voiding dysfunction. However, no bladder pathology was ever demonstrated despite a series of ultrasound evaluations the recent years. Because of the high suspicion of bladder malignancy, the patient submitted to further imaging investigation. A circumstantial ultrasound investigation revealed a 3 cm mass located at the trigone [Figure 1]a. Subsequently, the patient submitted to computed tomography (CT) urography which showed a normal upper urinary tract, no other visceral lesions, and no lymph node involvement. The bladder mass had a size of 2.7 cm × 3.1 cm, and it was located right at the bladder neck approximately 1.5 cm away from both ureteral orifices. The lesion was solid, moderately enhanced after the contrast injection, and created a characteristic filling deficit during the urography [Figure 1]b,[Figure 1]c,[Figure 1]d. No conclusion could be made about the degree of urethral involvement. Urine cytology was also negative for any malignancy. Eventually, the patient was submitted to transurethral resection (TURB) 1 week later, where a large proximal urethral mass was recognized that was attached to the right side, caused an almost complete obstruction of the bladder neck, and had extensive intravesical protrusion [Figure 2]a. There was no involvement of the trigone itself, yet a biopsy of the mucosa was sent separately for pathological evaluation. The tumor was completely excised carefully using a pure bipolar resectoscope with low power settings to minimize the trauma on the urethral sphincter [Figure 2]b. The patient was discharged the next day and 1 week later reported impressive voiding function improvement. The pathological examination showed a benign tumor of smooth muscle cells with no mitotic activity, no significant atypia, and no necrosis, consistent with bladder leiomyoma [Figure 3]. Immunohistochemical evaluation included stains for Cytokeratin, h-Caldesmon, and Ki67 and resulted in a very low proliferation rate (<1%) which corroborated the diagnosis. Finally, the patient was scheduled for endoscopic follow-up after multidisciplinary consultation without any additional treatment. The cystoscopy at 3 months was negative for any signs of residual tumor or recurrence and the mucosa of the urethra, bladder neck, and bladder was normal [Figure 4]a and [Figure 4]b.
Figure 1: Imaging evaluation of the patient. (a) Ultrasonographic image identifying the solid mass at the bladder neck. (b) Coronal plane of the computed tomography scan demonstrating the urethral involvement. (c) Axial plane of the computed tomography urography showing the characteristic filling deficit into the bladder (white arrow). (d) Three-dimensional reconstruction of the urinary tract demonstrating a deficit (white arrow) at the level of the pubic symphysis

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Figure 2: Intraoperative images of the transurethral resection of the tumor. (a) The bladder neck leiomyoma inside the urethra causing a complete obstruction (white arrow). The tumor was attached to the right side and the resection began on the left side toward the base of the tumor. The yellow arrow is pointing at the normal posterior urethral mucosa. (b) Final endoscopic view after complete resection at the level of the bladder neck

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Figure 3: Histopathological image of the specimen under H and E, ×100. Irregular fascicles of smooth muscle cells with no significant atypia or mitotic activity, covered by normal urothelium indicative of benign bladder leiomyoma and mild chronic cystitis inflammation findings

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Figure 4: Three-month postoperative flexible video-cystoscopy of the patient. (a) 210° flexion image of the bladder neck demonstrating normal mucosal scarring with no signs of residual tumor or recurrence (whitish area at 10,11 and 12 o'clock, upside-down image). (b) Endoscopic view of the previously obstructed bladder neck with normal appearance after complete surgical excision of the leiomyoma

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  Discussion Top


Leiomyomas of the urinary bladder are very rare neoplasms, with only ~250 cases previously reported in the literature.[3] Although rare, they are the most common benign mesenchymal tumors of the bladder. They usually affect patients in their third to sixth decade with a female preponderance (70%).[4] According to their location, leiomyomas are classified as endovesical, intramural, and extravesical. Endovesical leiomyomas are the most common and constitute 63%–86% of the cases, while intramural leiomyomas are present in 3%–7% and extravesical in 11%–30%. The most common symptoms at presentation are obstructive (49%), followed by irritative (38%) and hematuria (11%), while in larger masses, flank pain may present due to ureteric obstruction.[5] LUT leiomyomas should always be considered in the differential diagnosis of an anterior vaginal mass. Ultrasound is most commonly the first imaging step. Magnetic resonance imaging (MRI) and CT may also be performed. The ultrasound will typically detect a smooth-walled solid lesion with numerous internal echoes and homogenous texture of medium echogenicity. Cystoscopy can show a characteristic bladder mass with a smooth and regular mucosa. CT generally shows a smooth-walled bladder filling defect with an attenuation coefficient of 25–50 Hounsfield units.[6] MRI shows an intermediate signal intensity on T1-weighted images, giving good contrast compared with the low-intensity signal of urine. On T2-weighted images, it gives areas of high and low intensity at the same time, giving excellent contrast, as opposed to the intermediate-low intensity of the bladder muscle, facilitating the diagnosis of an extravesical extension.[7] TURB represents the main treatment in almost 90% of cases unless a large intramural or an extravesical leiomyoma is encountered requiring a wider excision such as a partial cystectomy or a transvaginal resection.[5],[8] Surgical treatment is usually effective since bladder leiomyomas have generally a good prognosis, and the recurrence rate is extremely low. However, a residual tumor may be present in 18% of the cases after TURB and these patients will require a re-operation.[5] Therefore, a cystoscopic follow-up schedule is recommended in these patients. Notwithstanding, the knowledge of urinary leiomyomas is limited to only a few case reports or small case series to recommend standard treatment and follow-up options.

The pathophysiology of these lesions remains unknown but four theories have been proposed: (1) Hormonal disturbances cause these tumors to develop; (2) dysontogenesis, for example, embryonic rests of tissue residing in the bladder that transforms into leiomyomas; (3) perivascular inflammation leading to a metaplastic transformation of the bladder vascular supply; and (4) bladder musculature infection leading to inflammation and the development of these benign tumors.[9] More studies and research are needed to elucidate the mechanism of their growth. Leiomyomas of the urinary bladder share common histopathological features with leiomyomas of the uterus, i.e., round gray-white nodules with a spiral appearance of smooth muscle fibers gathered in small fascicles and separated by varying amounts of fibrous connective tissue, and fewer than two mitotic figures per high-power field.[8] Interestingly, bladder leiomyomas during pregnancy increase more obviously in size and recur more frequently.[10],[11] This observation suggests that bladder leiomyomas may be dependent on hormonal changes as uterine leiomyomas. The use of gonadotropin-releasing hormone (GnRH) may be another choice for the treatment of bladder leiomyomas as in uterine leiomyomas. GnRH analogs can be effective for uterine leiomyomas because they can influence hypothalamus–pituitary–gonadal axis and play a role as medical oophorectomy. GnRH treatment has been predicted to be effective for bladder leiomyomas as well, since bladder leiomyomas have also been reported to be associated with hormonal changes.[12],[13]

In conclusion, bladder leiomyoma is a rare nonmalignant tumor that can present with typical LUT symptoms and/or hematuria. Imaging and endoscopic evaluation are mandatory since the tumor can be effectively managed with transurethral resection only.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Campbell EW, Gislason GJ. Benign mesothelial tumors of the urinary bladder: Review of literature and a report of a case of leiomyoma. J Urol 1953;70:733-41.  Back to cited text no. 1
    
2.
Silva-Ramos M, Massó P, Versos R, Soares J, Pimenta A. Leiomyoma of the bladder. Analysis of a collection of 90 cases. Actas Urol Esp 2003;27:581-6.  Back to cited text no. 2
    
3.
Khater N, Sakr G. Bladder leiomyoma: Presentation, evaluation and treatment. Arab J Urol 2013;11:54-61.  Back to cited text no. 3
    
4.
Cornella JL, Larson TR, Lee RA, Magrina JF, Kammerer-Doak D. Leiomyoma of the female urethra and bladder: Report of twenty-three patients and review of the literature. Am J Obstet Gynecol 1997;176:1278-85.  Back to cited text no. 4
    
5.
Goluboff ET, O'Toole K, Sawczuk IS. Leiomyoma of bladder: Report of case and review of literature. Urology 1994;43:238-41.  Back to cited text no. 5
    
6.
Bryckaert PE, Ceccaldi PF, Bancheri F, Staerman F. Pelvic pain caused by bladder leiomyoma: Diagnostic and radiologic difficulties. Prog Urol 2002;12:1299-301.  Back to cited text no. 6
    
7.
Maya MM, Slywotzky C. Urinary bladder leiomyoma: Magnetic resonance imaging findings. Urol Radiol 1992;14:197-9.  Back to cited text no. 7
    
8.
Knoll LD, Segura JW, Scheilhauer BW. Leiomyoma of the bladder. J Urol 1986;136:906-13.  Back to cited text no. 8
    
9.
Teran AZ, Gambrell RD. Leiomyoma of the bladder. Int J Fertil 1989;34:289-92.  Back to cited text no. 9
    
10.
Núñez Mora C, Julve Villalta E, Hardisson Hernáez D, Jiménez de León J, Picazo García ML, Hidalgo Togores L, et al. Leiomioma vesical durante el embarazo [Bladder leiomyoma during pregnancy]. Arch Esp Urol 1999;52:510-3.  Back to cited text no. 10
    
11.
Kulkarni JN, Kamat MR, Chinoy RF. Bladder leiomyoma in pregnancy: A case report. Tumori 1992;78:414-6.  Back to cited text no. 11
    
12.
Furuhashi M, Suganuma N. Recurrent bladder leiomyoma with ovarian steroid hormone receptors. J Urol 2002;167:1399-400.  Back to cited text no. 12
    
13.
Matsuo H, Maruo T. GnRH analogues in the management of uterine leiomyoma. Nihon Rinsho 2006;64 Suppl 4:75-9.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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