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Characteristics and outcomes in bladder Leiomyoma management: a systematic review of case reports and case series from the past 20 years

BMC Urology volume 24, Article number: 252 (2024) Cite this article

Abstract

Introduction

Bladder leiomyomas (LM) are uncommon, non-cancerous growths that originate from the smooth muscle cells of the bladder and constitute 0.5% of bladder tumor cases. This review aims to compile existing data and present a summary of bladder leiomyomas’ characteristics, management, and related outcomes.

Method

We conducted systematic review of studies that investigated bladder leiomyoma. Case studies or series describing individuals with bladder leiomyoma who underwent surgery and the outcomes were included. Four databases were used in our literature search, which was carried out until January 2024: PubMed, Proquest, EBSCOHost, and Google Scholar. We utilized MeSH terms such as “leiomyoma,” “urinary bladder,” and looked for synonyms of “bladder leiomyoma” in free text.

Results

A total of 99 studies with 119 patients were included. Most reported bladder leiomyoma cases were female, accounting for 79.0% of all cases. While symptomatic patients primarily presented with lower urinary tract symptoms (LUTS) (59.7%), hematuria (24.4%), acute urine retention (11.8%), and about 16.8% of cases were incidentally detected. Storage-related symptoms were the most common LUTS (37.0%). Different diagnostic techniques were used, frequently combining CT (Computed Tomography), MRI (Magnetic Resonance Imaging), USG (Ultrasonography), and/or cystoscopy. Bladder leiomyomas were commonly found on the left lateral wall (26.9%) and the bladder neck (17.6%). In more than half of the cases (52.1%) the treatment techniques used were transurethral resection (TURBt/TUR). Fifteen out of 119 cases (12.6%) had recurrence or remain symptomatic. Symptomatic symptoms at first presentation and extended location are frequently found among recurrent or symptomatic cases after first management.

Conclusion

Management of bladder leiomyoma should focus on the relief of symptoms and recurrence and be personalized based on the tumor characteristics, patient symptoms, and surgeon’s expertise. Further investigation is necessary to fully understand the best course of treatment and long-term results for bladder leiomyomas. In particular, prospective trials with bigger participant pools and meticulously controlled factors should be the main emphasis of this research.

Peer Review reports

Introduction

Bladder leiomyomas (LM) are uncommon, benign tumors that develop from the smooth muscle cells of the bladder wall and constitute less than 0.5% of bladder tumour [1, 2]. Despite being benign, these tumors have the potential to restrict the bladder outlet if located near bladder outlet or neck, which can result in symptoms and consequences related to the urinary tract, including sensation of incomplete emptying, weak stream, urine retention, recurrent urinary tract infections, and renal impairment [3,4,5], . The symptoms vary according to the size and location of the tumor. Bladder LM may present as irritative symptoms, hematuria, and flank pain. Bladder tumor may cause disorganized contraction of bladder leading to lower urinary tract symptoms. Tumor near ureteral orifice might cause obstruction leading to ureteral colic, flank pain, and obstructive uropathy. There were also a few patients who had no symptoms at all [6].

Bladder LMs have been treated with a variety of modalities; they include transurethral resection, open surgical excision, and minimally invasive methods like laparoscopy and robotic-assisted surgery [6,7,8]. However, opinions on the best course of action for diagnosis and management are diverse. Furthermore, the majority of the literature on bladder LM therapy consists of case reports and short case series, which makes it challenging to draw definitive conclusions about the features and results of various treatment approaches.

As such, single case reports and small case series are excellent sources for understanding the subtleties of the clinical presentation and therapeutic strategies for this rare illness. To summarize the existing data and offer insights into the characteristics, approaches to treatment, and outcomes related to bladder LMs, a thorough analysis of case reports and case series is necessary. This study aims to better understand the clinical presentation, diagnostic strategy, treatment methods, and results of bladder LMs by methodically locating and examining published cases. This will help guide future research directions and support clinical decision-making.

Method

This systematic review was performed based on Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA) guidelines and registered on PROSPERO (CRD42024506575).

Eligibility criteria

Research studies may be eligible for inclusion if they explore or document cases of patients diagnosed with bladder LM who underwent surgical management with reported post-management outcomes. The literature should be in the form of a case report or case series. Additionally, letters or correspondences providing information about bladder LM, with adequate details, will also be incorporated to prevent overlooking crucial information on this uncommon occurrence. Reports from the same health center and demographic will be excluded to ensure the avoidance of duplicate patient inclusions. Studies that were not reporting bladder LM or no management data were also excluded. Pediatric cases were also excluded due to the rarity in children. No year of publication was applied for identifying studies, however, only studies published in 2000 and over were included. We only included studies using English or Indonesian.

Search strategy

We employed four databases—PubMed, Proquest, EBSCOHost, and Google Scholar to conduct a literature search up to January 2024. Our search utilized the MeSH term (“urinary bladder” AND “leiomyoma”) OR free text word of “bladder leiomyoma” and its word variants (listed in Supplementary File 1). Two authors (AH and SKL) independently screened research titles to exclude studies that did not meet eligibility criteria, and subsequently, selected studies underwent abstract and full-text reviews (Fig. 1). Any disagreements between the two authors were resolved through discussion with a third and fourth author (JCP and SPH).

Fig. 1
figure 1

PRISMA flow diagram 2020

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Data selection, collection, and extraction

The data were extracted by two independent co‑authors and double‑checked by the third author to ensure the data were matched and organized. The following information was extracted from the selected studies: author, study year, country, study design, duration of follow-up, number of patient(s), age, complaints, diagnostic data, location of the tumor, size of the tumor, presence of comorbidity, management, and outcome of the patient (quality of life, residual symptoms, urodynamic studies, and complications).

Quality assessment

We used The Joanna Briggs Institute (JBI) critical appraisal checklist designed for case series and case reports to assess the quality of the studies.

Results

A total of 1003 studies were retrieved from four databases and screened independently by two co-authors resulting in 99 studies eventually included in this review (Fig. 1) [2, 3, 5,6,7,8,9,10,11,12,13,14,15,16,17,18,19,20,21,22,23,24,25,26,27,28,29,30,31,32,33,34,35,36,37,38,39,40,41,42,43,44,45,46,47,48,49,50,51,52,53,54,55,56,57,58,59,60,61,62,63,64,65,66,67,68,69,70,71,72,73,74,75,76,77,78,79,80,81,82,83,84,85,86,87,88,89,90,91,92,93,94,95,96,97,98,99,100,101]. All included cases are shown in Table 1. The total number of patients included in this review is 119 patients, with ages ranging from 22 to 75 years (median 43) (Table 2).

Table 1 Characteristics of included studies
Full size table
Table 2 Patient’s characteristics
Full size table

Most of the reported cases were female, which comprised 79.0% of total cases. A total of 16.8% cases were found incidentally (asymptomatic), while in symptomatic patients, most experienced LUTS (59.7%), followed by hematuria (24.4%), acute urinary retention (11.8%), abdominal pain (9.2%), flank pain (5.9%), palpable mass (7.6%), pelvic pain or discomfort (5.9%), and abnormal uterine bleeding (3.4%). The most common type of LUTS experienced by the patients was predominantly storage type (37.0%).

Diagnostic modalities used in these reported cases were combinations of either USG, CT scan, MRI, or cystoscopy. All cases underwent pathology examination to determine the final diagnosis of bladder LM. There is no mismatch between preliminary biopsy and final pathology results. Some studies that did not undergo preliminary biopsy directly conducted excisional surgery for final pathology. Bladder LM is composed of smooth muscle fibers mixed with connective tissue without mitotic activity, cellular atypia, necrosis, or infiltration. Most of the bladder LMs were found on the left lateral wall (26.9%), bladder neck (17.6%), bladder trigone (8.4%), and anterior wall (8.4%), with a wide range of sizes ranging from 1.0 to 18 cm of the longest dimension (median 4.1 cm). More than half of the reported cases were managed by TURBt/TUR (52.1%), followed by partial cystectomy (18.4%), open local excision (9.2%), open transvesical enucleation (5.9%), laparoscopic enucleation (2.5%), and laparoscopic cystostomy (2.5%).

A total of 15 out of 119 cases had a recurrence or remained symptomatic, 75 patients did not have recurrence or symptoms, and the rest 29 patients did not have reported information regarding the final outcome of the management (Table 2). Most were symptomatic at the beginning, only 6.7% were incidental findings and extending to more than one location. Ten out of 15 cases underwent TURBt/TUR (66.7%) and residual tumors were frequently found.

Discussion

The present systematic review synthesized data from 99 studies encompassing 119 patients diagnosed with bladder LM over the past two decades. Our findings highlight several key characteristics and outcomes pertinent to the management of this rare condition. Even if the precise etiology is still unclear, several important processes have been suggested with most research derived from uterine LM studies [102,103,104]. The intricate interaction of genetic, hormonal, and environmental variables is responsible for the pathogenesis of LM.

Demographics

Our analysis revealed a predominance of female cases, comprising 79% of the total cohort. The age range of affected individuals varied widely, spanning from 22 to 75 years, with a median age of 43 years. It frequently occurs in middle-aged women and rarely in young men. For pediatric cases, yhe youngest cases were from a 1-year-old boy and 6-year old boy [105, 106].

Diagnosis

Notably, a considerable proportion of cases (16.8%) were incidentally detected. Among symptomatic patients, lower urinary tract symptoms (LUTS) were the most commonly reported clinical presentation, affecting 59.7% of cases (Table 2). Urinary retention occurs usually in bladder outlet tumours or large LMs.

It might be difficult to distinguish bladder LMs from bladder cancer for a variety of reasons. Clinical distinction is challenging since both illnesses frequently present identical symptoms, such as hematuria, lower urinary tract symptoms (LUTS), and pelvic pain. Moreover, mass lesions within the bladder in both entities may be detected by radiological imaging techniques, confounding the diagnosis even more. Thus, diagnostic evaluation typically involves a combination of imaging modalities such as ultrasonography (USG), computed tomography (CT) scan, magnetic resonance imaging (MRI), and cystoscopy to ascertain what lesion the clinicians are dealing with. In the end, histological analysis of tissue taken from a biopsy or surgical resection is required for a definite diagnosis of bladder LMs or bladder cancer, highlighting the significance of careful pathological assessment in clinical decision-making. Unlike uterine LM, bladder LM was never reported to transform into malignant leiomyosarcomas [107].

Urinary bladder usually investigated using USG through transabdominal route. On USG, LM usually characterized as having smooth-walled homogenous hypoechoic solid mass with thin echogenic surface [6]. A LM may show up on a CT scan as a homogeneous, pedunculated mass that lacks distinguishing characteristics from other bladder neoplasms. The visual characteristics of bladder LMs on MRI are determined by the presence of collagen and smooth muscle, just like uterine LMs. They often have variable degrees of dynamic enhancement and a uniform T2-hypointense and T1-hypointense look that resembles muscle tissue [108]. Furthermore, they might exhibit a “T2-weighted black-out” effect, which would show up as low signal intensity regions on diffusion-weighted MRI, hypointense areas on high b-value diffusion-weighted MRI, and hyperintense areas on ADC maps. In urothelial carcinoma, it frequently manifests as a strongly enhancing mass with limited dissemination that arises from the wall of the bladder. Non-muscle-invasive urothelial carcinomas are usually present as sessile or flat lesions that are only visible following a cystoscopy as erythema or anomalies of the mucosa. Interestingly, there is usually no thickening or abnormality in the surrounding bladder wall associated with these lesions. On occasion, papillary frond surfaces can show signs of calcification. Leiomyosarcoma typically appears as big, polypoid masses, usually near the bladder dome. It can be difficult to differentiate from a bladder LM since both lesions usually show low signal intensity on T2-weighted MRI. However, characteristics including poorly defined borders, invasion into neighboring structures, and necrotic patches can help differentiate LM from leiomyosarcoma. As with the distinction between leiomyosarcomas and uterine LMs, testing for diffusion limitation can help separate bladder leiomyosarcomas from bladder LMs. The diagnostic accuracy of bladder LM by any imaging modalities is not known due to rarity of cases, however, investigation by Ozden et al., showed that CT has 100% detection rates for bladder tumor larger than 5 mm, while USG has detection rate ranged from 90 to 100% for tumor larger than 5 mm, except for anterior wall region (with detection rate only 60-66.6%) [109]. Tumors with size less than 5 mm also difficult to be detected with USG (0–75% detection rate) or CT (0-66.6% detection rate). Other than diagnosis, USG can be applied to assess if there is invasion to bladder wall. Diffusion-weighted MR can reach sensitivity 95% and specificity 85% for bladder cancer detection [110].

In practice, USG appearance of malignant tumor or any mass-like benign lesions might be similar, it is difficult to differentiate them [111]. At CT, LM is indistinguishable from other bladder neoplasm [108]. Regardless the result of USG, CT, or MRI, whenever bladder tumor is suspected, cystoscopy and biopsy should be conducted as gold standard diagnostics. If bladder mass is found, the differential diagnoses include urothelial carcinoma, malignant mesenchymal mass (such as leiomyosarcoma, lymphoma of bladder, or angiosarcoma), other benign masses (such as paraganglioma, hemangioma, lymphangioma), inflammatory lesions (such as pseudotumor, poplypoid cystitis) [108].

Treatments

To the best of our knowledge, the lack of established studies identifying a superior treatment can be attributed to the rarity of cases and the variability observed among tumors, including variations in size, location, and accessibility [5]. Transurethral resection of bladder tumor (TURBT) is presently utilized as the primary treatment option, owing to its safety and un-eventful procedure, particularly in small, endovesical tumors that are easily accessible [88]. The genesis of TURBT can be traced back to 1806 [112], with subsequent refinement in 1910 by Beer [113], who introduced the utilization of electrocoagulation for the management of bladder tumors. After these historical developments, TURBT has been deemed sufficient for the management of endovesical tumors when performed by experienced operators, following confirmation through imaging modalities [5, 114].

In cases where tumors exhibit larger dimensions or entail extensive involvement of the bladder wall, the consideration of segmental resection via either open surgical or laparoscopic approaches may be done, ensuring comprehensive lesion eradication beyond submucosal levels [5, 48]. Minimally invasive techniques upon segmental resection, including laparoscopic approaches for managing bladder LMs, have been documented as early as 2002 [115]. Moreover, a transvesical method can be used to enhance visualization and manipulation of the lesion. This approach, augmented by the integration of robotic-assisted techniques and guided imaging on occasion, offers advantages such as improved precision and reduced risk of damage to surrounding structures when executed by a trained surgeon [8, 78].

The TURBT procedure exhibits the shortest duration, however, it is imperative to consider the recurrence rate and its constraints regarding the extent of the lesion [88]. The minimum length of stay ranged from a single day to a maximum of eight days among patients undergoing TURBT, while for open surgery, it extended from one day to two weeks, and for laparoscopic approaches, it varied from 10 days to three weeks across the studies included within our analysis. The duration of stay should not be the main concern, more invasive approach is indeed causing the longest hospitalization. Choosing the exact surgery for complete removal is the most important.

Our study presents the first comprehensive systematic review of case reports concerning to LM of the bladder, encompassing a cohort of 99 studies involving 119 patients across 38 countries. Furthermore, it encompasses essential data concerning precise anatomical localization, dimensional measurements, therapeutic modalities, and symptomatic presentations, with detailed consideration of age and gender demographics on a global scale.

In summary, our thorough review offers a significant understanding of the clinical characteristics, approaches to diagnosis, therapeutic strategies, and results of bladder LMs. Our results highlight the large age range of affected individuals, the preponderance of female cases, and the variety of clinical presentations, with lower urinary tract symptoms being the most frequently reported. MRI, CT scan, ultrasonography, and cystoscopy are among the imaging modalities commonly used in diagnostic evaluation. Nonetheless, a significant percentage of individuals have recurrence or persistent symptoms, emphasizing the necessity of ongoing observation and monitoring. Due to overlapping symptoms and radiological features, differentiating bladder LMs from bladder cancer can be difficult, underscoring the significance of histological investigation for conclusive. Our opinions for surgical management are that it should focus on the patient’s condition and tumour’s characteristics. Smaller lesions could be managed by TURBt, while larger lesions might require open surgery if full tumour removal cannot be achieved.

Several limitations are inherent in our study. Firstly, the studies included in our analysis predominantly consist of case reports or case series, rendering them vulnerable to biases, particularly selection and publication bias. Moreover, the typically small patient sizes inherent in these study designs may constrain the reliability and generalizability of the findings to larger populations with controlled variability. Consequently, the interpretation and utilization of our results within reviews necessitate careful consideration.

Conclusion

For an understanding of the best course of treatment and long-term results for bladder LMs, more investigation is necessary. This should primarily take the form of prospective trials with bigger sample sizes and controlled variability. Good sensitivity imaging and pathology examination should be done to exclude leiomyosarcoma. Since there is no standard intervention, management of bladder LM should focus on the relief of symptoms and recurrence and be personalized based on the surgeon’s experience, the patient’s condition, and tumour characteristics.

Data availability

The datasets used and/or analysed during the current study are available from the corresponding author on reasonable request.

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Authors and Affiliations

  1. Department of Surgery, Division of Urology, School of Medicine and Health Sciences, Atma Jaya Catholic University of Indonesia, Jakarta, Indonesia

    Johannes Cansius Prihadi, Antoninus Hengky, Stevan Kristian Lionardi & Sean Peter Haruman

  2. Department of Surgery, Division of Urology, Atma Jaya Hospital, Jakarta, Indonesia

    Johannes Cansius Prihadi

  3. Center of Health Research, Atma Jaya Catholic University of Indonesia, Jakarta, Indonesia

    Antoninus Hengky

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JCP: Resources, Supervision; JCP & AH: Conceptualization, Investigation; AH, SKL, SPH: Data Curation, Methodology, Formal Analysis, Project Administration, Validation, Visualization, Software, Writing – Original Draft, Writing – Review & Editing.

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Prihadi, J.C., Hengky, A., Lionardi, S.K. et al. Characteristics and outcomes in bladder Leiomyoma management: a systematic review of case reports and case series from the past 20 years. BMC Urol 24, 252 (2024). https://doiorg.publicaciones.saludcastillayleon.es/10.1186/s12894-024-01624-3

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BMC Urology

ISSN: 1471-2490

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