A multicenter retrospective study of 108 men with urethral hemangioma

Introduction

Male urethral hemangioma is an uncommon vascular lesion and represents a rare but likely under-recognized cause of sex-related hematuria and hematospermia in otherwise healthy men (1-3).

Patients typically present with gross hematuria, urethral bleeding, or blood spotting during the first urination following sexual activity (such as intercourse or masturbation), or even after penile erection without sexual stimulation, symptoms that are frequently misattributed to more common entities such as seminal vesiculitis, prostatitis, or prostatic/seminal vesicle pathology (4-6). As a result, many patients undergo repeated empirical treatments and multiple negative investigations before the true diagnosis is established, leading to prolonged anxiety, sexual avoidance, and impaired health-related quality of life (HRQoL) (4,7).

Current knowledge of urethral hemangioma is derived predominantly from isolated case reports and small case series (8), often with heterogeneous diagnostic pathways, and treatment modalities (1-7,9-27). Various endoscopic techniques, including urethrotomy, monopolar/bipolar coagulation, and different laser platforms, have been described, but most reports include only a few patients and provide limited follow-up (23,27-29). Consequently, the true clinical phenotype, optimal diagnosis and treatment modality, and robust data on recurrence risk remain poorly defined. Moreover, almost all published studies focus on anatomical or clinician-reported outcomes, with virtually no systematic assessment of patient-reported outcomes (PROs) such as global improvement, sexual function, lower urinary tract symptoms (LUTS), anxiety, and HRQoL.

Several key uncertainties therefore persist: (I) whether male urethral hemangioma, especially posterior lesions near the verumontanum, represents a relatively consistent clinical-anatomical entity with a recognizable symptom pattern; (II) the long-term safety and efficacy of standardized endoscopic surgery as a definitive treatment; (III) the true patient-perceived benefit and residual burden after treatment, as captured by structured PROs; and (IV) which baseline factors confer increased risk of poor PROs or recurrence, and thus might inform risk-adapted counselling and follow-up.

In this context, the present two-center retrospective cohort study reports, to our knowledge, the largest series of male urethral hemangioma to date. The aims were: (I) to characterize the clinical and anatomical phenotype of male urethral hemangioma, with particular attention to posterior urethral lesions; (II) to evaluate perioperative and long-term outcomes of a standardized approach combining cystourethroscopy with immediate bipolar endoscopic treatment; (III) to systematically assess PROs, including a prespecified composite PRO endpoint; and (IV) to identify risk factors for poor PRO and disease recurrence using appropriate regression methods for relatively rare events. We present this article in accordance with the STROBE reporting checklist (available at https://tau.amegroups.com/article/view/10.21037/tau-2026-0347/rc).

Methods

Study approval and patient selection

This was a retrospective, multicentered cohort study of consecutive male patients diagnosed with urethral hemangioma and treated endoscopically from January 2014 to June 2025 from Affiliated Hospital of Guizhou Medical University and Army Medical Center of Army Medical University. PROs were collected at the last follow-up by telephone or clinic visit. The study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. This study was approved by the Institutional Review Board of Affiliated Hospital of Guizhou Medical University (IRB: 2025190K). The requirement for written informed consent was waived owing to the retrospective design. Army Medical Center of Army Medical University was also informed of and agreed to the study.

Patients were eligible for inclusion if they met all of the following criteria: (I) age ≥18 years; (II) a clinical history of sex-related hematuria and/or hematospermia (e.g., post-ejaculatory hematuria, hematospermia with sexual activity); and (III) endoscopically confirmed urethral hemangioma treated with transurethral bipolar electroresection and/or electrocoagulation at one of the participating centers. Exclusion criteria were: (I) concomitant or alternative urological diagnoses that could reasonably explain sex-related hematuria (including but not limited to genitourinary malignancies, urolithiasis, seminal vesiculitis, or severe prostatitis) without endoscopic evidence of urethral hemangioma; (II) absence of diagnostic cystourethroscopy or lack of definitive visualization of a urethral hemangioma; and (III) patients not undergoing endoscopic surgical treatment (e.g., managed conservatively or lost before intervention), as well as those with incomplete key baseline or follow-up data.

Baseline data included age, body mass index (BMI), comorbidities (hypertension, diabetes, cardiovascular disease, and anticoagulant/antiplatelet use), main clinical manifestations (hematospermia, post-ejaculatory hematuria, arousal- or erection-related hematuria, blood clots, LUTS), International Prostate Symptom Score (IPSS) at baseline, symptom duration, prior misdiagnosis and corresponding treatment history, preoperative imaging [ultrasound/TRUS, magnetic resonance imaging (MRI)], prior urethrocystoscopy elsewhere, lesion location (distal verumontanum 6-o’clock vs. other sphincter-plane vs. prostatic vs. bulbar/anterior urethra) identified during surgery, lesion number (solitary vs. multiple), and surgical pathology when available. Perioperative variables included operative time, estimated blood loss, length of stay, catheterization duration and postoperative complications.

Outcome definitions

The primary PRO was a composite endpoint prospectively collected by telephone or clinic visit. Telephone follow-up was collected between September 2025 and October 2025. PRO failure was defined as the presence of any of the following: (I) patient global impression of change (PGIC) rated as “minimally improved”, “no change” or worse (PGIC was rated between 1 and 6, in which 1= very much improved, 2= much improved, 3= minimally improved, 4= no change, 5= minimally worse, 6= much worse), (II) overall satisfaction with surgery ≤7 (out of 10) (1–3: not satisfied; 4–7: neutral; 8–10: satisfied), or (III) existence of fear-driven avoidance of sexual activity over the past 3 months. Patients not meeting any of these criteria were classified as PRO success. Other PROs included individual domains at last follow-up: willingness to undergo the same procedure again, presence of post-ejaculatory hematuria or hematospermia, sexual health inventory men (SHIM) score, self-reported change in erectile function, ejaculatory pain, subjective change in ejaculate volume, IPSS, change in LUTS, new-onset urinary incontinence, anxiety related to bleeding/disease, overall HRQoL score, impact on intimate relationship, and patient perception of being cured. The secondary outcome was disease recurrence, defined as recurrent hematuria or hematospermia after an initial period of complete symptom resolution. All symptom recurrence patients were confirmed by cystourethroscopy and underwent repeat endoscopic surgery.

Surgical technique

Diagnosis was based on typical symptoms plus endoscopic confirmation. Urethral hemangiomas typically appear as venous, mass-like protrusions measuring approximately 5–10 mm in diameter, with well-defined margins, most often located at the 6 o’clock position near the external urethral sphincter distal to the verumontanum. The surface color ranges from pink to dark red or bluish-purple, depending on the degree of vascular engorgement and lesion depth.

Several technical points are important during endoscopic inspection. First, gentle, repeated pressure on the urethral mucosa with the tip of the endoscope can help unmask small or partially concealed lesions. Second, maintaining a relatively low irrigation pressure, or intermittently pausing the inflow, reduces mucosal collapse and bleeding obscuration and improves visualization of the sphincter plane. Third, particular attention should be paid to the distal verumontanum and the adjacent external urethral sphincter plane, as these are the most frequent sites of disease.

For endoscopic treatment, bipolar electrosurgical resection or coagulation using low-power, short, and precise bursts is recommended to limit stimulation of the external urethral sphincter and to minimize the risk of postoperative urinary incontinence or urethral stricture. For small, well-localized hemangiomas, focal coagulation with a needle-type electrode can effectively ablate the lesion while limiting collateral thermal injury. In cases with circumferentially distributed dilated vessels, care should be taken to avoid extensive mucosal destruction in order to reduce the risk of subsequent urethral stricture. The detailed surgical technique has been described previously (5).

Statistical analysis

Continuous variables are presented as mean ± standard deviation or median (interquartile range) according to distribution, and categorical variables as number (percentage). To identify factors associated with PRO failure, univariable logistic regression analyses were first performed for clinically relevant covariates (age, baseline LUTS, fear of unknown etiology, lesion location, lesion multiplicity, and recurrence). Variables with P<0.10 in univariable analysis and those considered clinically important were entered into a multivariable logistic regression model. As only 8 patients experienced disease recurrence, Firth penalized Cox proportional regression was used to reduce small-sample bias and mitigate potential separation in the recurrence models. Univariable Firth models were fitted for age, symptom history, presence of LUTS, typical versus atypical lesion location, lesion multiplicity, and anticoagulant/antiplatelet use. Variables of interest were then included in a parsimonious multivariable Firth model. All statistical tests were two-sided, and a P value <0.05 was considered statistically significant. Analyses were performed by Stata 18.0 (StataCorp, College Station, TX, USA).

Results

Population characteristics

In Table 1, a total of 108 men were included (mean age 41.4±8.7 years; median symptom duration 23 months), with low comorbidity burden and universal presentation with hematospermia plus post-ejaculatory hematuria. Lesions were predominantly solitary (87.0%) and located at the distal verumontanum 6-o’clock position (73.1%), and perioperative outcomes were favorable with minimal blood loss, short hospital stays, and catheterization for a median of 2 days. During a median follow-up of 55 months, symptoms resolved within 6 weeks in all patients, recurrence occurred in 8 (7.4%) cases (6/8 at atypical locations), and all recurrent cases were successfully managed with repeat endoscopic surgery. Overall complication rate was only 2.7%.

Pathological confirmation was obtained in 20 patients, mainly during the early phase of our team’s experience with this condition. As our clinical experience accumulated, we recognized that this lesion is benign and that routine biopsy or pathological confirmation did not provide additional benefit for diagnosis or treatment planning, while it could increase unnecessary financial burden. Therefore, pathology was not performed routinely in later cases unless clinically indicated.

PROs at last follow-up

In Table 2, at last follow-up, PROs showed marked clinical benefit, with 97.2% of patients willing to undergo the same procedure again, a median PGIC of 1 and high satisfaction scores, and essentially complete elimination of post-ejaculatory hematuria and hematospermia. Sexual function and LUTS were generally preserved or improved (median SHIM 19; median IPSS 4), avoidance of sexual activity due to fear of re-bleeding was rare (1.8%), and nearly all patients perceived themselves as cured with high overall HRQoL scores [8 (7, 9) score].

Risk factors for PRO failure

According to this definition, 17/108 patients (15.7%) were classified as PRO failure. As shown in Table 3, in univariable logistic regression, older age (OR 1.077, 95% CI: 1.014–1.145; P=0.02) and baseline LUTS (OR 4.444, 95% CI: 1.450–13.419; P=0.009) were associated with higher odds of PRO failure, whereas fear of unknown etiology, lesion location, multiplicity, and recurrence were not significantly associated. In the multivariable model, only baseline LUTS remained an independent predictor of PRO failure (OR 3.400, 95% CI: 1.089–11.114; P=0.04).

Risk factor for disease recurrence

Given the limited number of recurrence events (n=8), Firth penalized Cox regression was used to explore risk factors for disease recurrence. In univariable Firth models, age, symptom history >2 years, baseline LUTS, typical versus non-typical location, and anticoagulant/antiplatelet use were not significantly associated with recurrence. In the multivariable Firth model including lesion location (typical vs. non-typical) and multiplicity, multiple lesions emerged as an independent predictor of recurrence (HR 5.732, 95% CI: 1.044–31.468; P=0.04), whereas typical versus non-typical location was not associated with recurrence (HR 0.421, 95% CI: 0.080–2.212; P=0.31) (Table 4).

Discussion

Urethral hemangioma is an uncommon, benign vascular lesion of the lower urinary tract and a rare but often under-recognized cause of sex-related hematuria and hematospermia. Because of limited awareness among clinicians, diagnosis is frequently delayed or missed. In this largest series to date with median 55 months of follow-up, male urethral hemangioma demonstrated a highly characteristic clinical and anatomical pattern centered on the distal verumontanum 6-o’clock region. Our findings confirm that cystourethroscopy performed under anesthesia followed by immediate bipolar endoscopic resection and/or coagulation represents a safe and effective one-step diagnostic-therapeutic strategy. This approach achieved rapid symptom control, durable long-term efficacy with a recurrence rate of 7.4% within a median of nearly 5 years, and high patient-reported satisfaction.

Historically, urethral hemangioma has been described only sporadically in the literature (4-7,10-27,30) (Table S1, Figure 1). Since the first case reported by Klotz in 1895 (31), fewer than 150 cases have been documented worldwide, mostly as isolated reports employing heterogeneous diagnostic and therapeutic approaches. Leary et al. [1974] (9) first recognized urethral varices as the source of hematospermia in selected patients, followed by Cattolica et al. [1982] (32) who proposed that large-volume bleeding originates from rupture of dilated abnormal veins during erection. Two decades later, Hayashi et al. [1997] (33) established posterior urethral hemangioma as a distinct clinicopathological cause of sex-related hematuria. Despite these observations, awareness remains low, and previous studies have not systematically defined its clinical phenotype, long-term outcomes, or patient-centered effects. Building upon our previous institutional experience of 39 cases published in 2020 (4), the present study expands this knowledge base through a multi-center series with over 100 patients and long-term observation.

Figure 1 Reported cases of urethral hemangioma by year (2000–2025).

The etiology of urethral hemangioma remains uncertain and likely multifactorial, but several mechanisms have been proposed. Some authors consider it a congenital vascular malformation originating from unipotent angioblastic stem cells, whereas others suggest that it represents an acquired vascular dysplasia triggered by chronic mechanical irritation, local hypoxia, or inflammation during repeated sexual activity (6,7,34). Associations with congenital syndromes such as Klippel-Trenaunay have been described but are rare and were absent in our cohort (35-37). Anatomically, the lesion most often arises in the posterior urethra, particularly at the 6-o’clock plane distal to the verumontanum, where rich venous plexuses and mechanical stress converge during erection and ejaculation.

There are three key clinical features for suspicious of urethral hemangioma: (I) first-void hematuria following erection, ejaculation, or sexual stimulation; and (II) bright-red blood, normally separate from semen; (III) hematuria associated with this condition often recurs or persists stubbornly but is absent in the absence of sexual activity. In severe cases, it can be substantial enough to form clots, potentially leading to urinary retention or bladder tamponade (4,26,27). In our experience, these features are strongly correlated with the diagnosis of urethral hemangioma.

Despite these recognizable features, urethral hemangioma is often under-diagnosed. In our cohort, approximately 80% of patients had received empirical antibiotic therapy and 16% experienced diagnostic delays of more than five years. Routine imaging, including ultrasound, TRUS, and MRI, showed limited diagnostic yield, and even prior cystourethroscopy performed elsewhere was often negative. These findings emphasize the importance of direct visualization under optimal conditions with appropriate skills (4,5).

Not all urethral hemangiomas, however, are symptomatic. In the series by Saito et al., 9 of 20 patients (45%) were incidentally identified during urethroscopy for other indications (27). This suggests that asymptomatic lesions may be more common than previously recognized, and routine inspection of the distal verumontanum and surrounding sphincteric region during cystourethroscopy, particularly in patients with persistent hematospermia or unexplained hematuria, may improve detection rates.

For symptomatic patients, endoscopic evaluation under anaesthesia is recommended. This one-step diagnostic-therapeutic strategy, allowing immediate bipolar resection or coagulation once the lesion is identified, avoids repeated investigations and expedites definitive cure. During the procedure, meticulous inspection under direct vision should focus on the distal verumontanum and the external sphincteric plane. Some authors have suggested that pre-procedure sexual activity may enhance vascular engorgement and lesion visibility, whereas others advocated intraoperative induction of artificial erection. In our experience, such measures are unnecessary when careful endoscopic technique and appropriate irrigation control are employed (15,23). Although Doppler ultrasonography, MRI, or angiography have been explored for localization, their diagnostic value remains limited due to the small lesion volume and minimal blood flow in the flaccid state (6,27).

Various interventions have been reported for the treatment of urethral hemangioma, including oral medications or intralesional injections (23,25), open surgery for large or complex lesions (30), and several forms of endoscopic intervention, such as bipolar electrosurgical resection, needle electrode coagulation (23), and laser ablation (18,28). Among these, bipolar electrosurgical resection and coagulation have become the preferred options because of their safety, precision, low cost, and rapid recovery. In our study, all 108 patients underwent bipolar endoscopic management, achieving prompt symptom resolution in 100% and a long-term recurrence rate of only 7.4%. These outcomes compare favorably with earlier reports, which were limited by small cohorts, heterogeneous technologies, and short-term follow-up. Importantly, most recurrences occurred at anatomically distinct sites rather than within the original treatment area, suggesting the development of new vascular foci rather than incomplete ablation. Multiple lesions were identified as the only independent predictor of recurrence, underscoring the importance of comprehensive intraoperative inspection and vigilant postoperative follow-up for patients with diffuse disease.

Beyond anatomical and surgical success, our analysis provides the first comprehensive evaluation of PROs in male urethral hemangioma. Nearly all patients achieved durable symptom resolution, high satisfaction (median 9/10), and preservation of sexual and urinary function. PRO failure occurred in 15.7% of cases and was independently associated with baseline LUTS, suggesting that pre-existing voiding dysfunction influences postoperative perception of improvement.

Although this figure may appear relatively high compared with the excellent objective control of bleeding and the low recurrence rate, it likely reflects the impact of progressive age-related decline over the extended follow-up. With longest observation of 12 years, many patients inevitably developed age-associated changes in erectile or voiding function, which could reduce satisfaction or PGIC scores even without anatomical recurrence. In addition, the onset of comorbidities such as benign prostatic enlargement or cardiovascular disease may compound LUTS and sexual fatigue. Accordingly, a proportion of observed PRO failure should be regarded as an expected consequence of long-term physiological ageing rather than a direct indicator of treatment inadequacy. Prospective, age-adjusted PROs assessment will be valuable to distinguish true disease-specific treatment failure from global functional decline.

This study has several limitations that warrant consideration. First, its retrospective design may introduce selection bias, as only surgically treated patients were included and individuals with mild or spontaneously resolving symptoms were not captured. Second, bipolar electrosurgery was the sole technique used across two centers, precluding direct comparison with laser-based or other emerging modalities. Third, PROs were collected through self-reported questionnaires at follow-up visits or by telephone, raising the possibility that minor or asymptomatic recurrences may have been overlooked or recall bias. Moreover, although this represents the largest cohort to date, the study was confined to two tertiary institutions, thus, the generalizability of our proposed diagnostic-therapeutic strategy requires validation in larger, multicenter, prospective settings. Future work should aim to establish standardized diagnostic criteria, incorporate objective functional measurements, and develop longitudinal PRO instruments specific to urethral vascular lesions. However, at this stage, increasing public awareness especially among urologists is still the priority.

In summary, this study consolidates male urethral hemangioma as a distinct urethral entity, demonstrates the safety and effectiveness of a one-step endoscopic strategy, introduces PROs into the assessment framework, and identifies lesion multiplicity as the main predictor of recurrence. Together, these findings form the most comprehensive evidence base to date for the diagnosis, treatment, and long-term follow-up of this uncommon condition.

Conclusions

Cystourethroscopy followed by immediate endoscopic treatment is a safe and effective one-step diagnostic-therapeutic strategy for men with typical urethral hemangioma symptoms with low long-term recurrence and high patient satisfaction. Baseline LUTS and lesion multiplicity offer clinically relevant risk stratification for PROs and recurrence.

Acknowledgments

None.

Footnote

Reporting Checklist: The authors have completed the STROBE reporting checklist. Available at https://tau.amegroups.com/article/view/10.21037/tau-2026-0347/rc

Data Sharing Statement: Available at https://tau.amegroups.com/article/view/10.21037/tau-2026-0347/dss

Peer Review File: Available at https://tau.amegroups.com/article/view/10.21037/tau-2026-0347/prf

Funding: This study was supported by the High-level Talent Recruitment Program of the Affiliated Hospital of Guizhou Medical University (No. gyfygcc-2024-01).

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://tau.amegroups.com/article/view/10.21037/tau-2026-0347/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. The study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. This study was approved by the Institutional Review Board of Affiliated Hospital of Guizhou Medical University (IRB: 2025190K). The requirement for written informed consent was waived owing to the retrospective design. Army Medical Center of Army Medical University was also informed of and agreed to the study.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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