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Journal of Indian Association of Pediatric Surgeons
     Journal of Indian Association of Pediatric Surgeons
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Table of Contents   
CASE REPORT
Year : 2023  |  Volume : 28  |  Issue : 1  |  Page : 62-64
 

Recurrent hemangioma of the penile urethra – An unusual etiology of gross hematuria managed successfully with holmium laser coagulation


1 Department of Urology, Jawaharlal Institute of Post Graduate Medical Education and Research, An Institute of National Importance, Government of India, Puducherry, India
2 Department of Pediatric Surgery, Jawaharlal Institute of Post Graduate Medical Education and Research, An Institute of National Importance, Government of India, Puducherry, India

Date of Submission12-Jul-2022
Date of Decision12-Sep-2022
Date of Acceptance12-Sep-2022
Date of Web Publication10-Jan-2023

Correspondence Address:
Bikash Kumar Naredi
Department of Pediatric Surgery, Jawaharlal Institute of Post Graduate Medical Education and Research, An Institute of National Importance, Government of India, Jipmer Campus Road, Gorimedu, Puducherry - 605 006
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.jiaps_96_22

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   Abstract 


Urethral hemangioma is a benign uncommon vascular tumor. It presents with hematuria and macroscopic urethrorrhagia. Urethrocystoscopy is the main diagnostic modality. We share the case of a 14-year-old male child who presented with recurrent massive hematuria and was treated with urethra-cystoscopic holmium laser coagulation.


Keywords: Hematuria, holmium laser, urethral hemangioma


How to cite this article:
Pal AK, Naredi BK, Manikandan R, Kalra S, Jindal B, Koya N. Recurrent hemangioma of the penile urethra – An unusual etiology of gross hematuria managed successfully with holmium laser coagulation. J Indian Assoc Pediatr Surg 2023;28:62-4

How to cite this URL:
Pal AK, Naredi BK, Manikandan R, Kalra S, Jindal B, Koya N. Recurrent hemangioma of the penile urethra – An unusual etiology of gross hematuria managed successfully with holmium laser coagulation. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 Feb 2];28:62-4. Available from: https://www.jiaps.com/text.asp?2023/28/1/62/367396





   Introduction Top


Hematuria occurs in approximately 1.5% of children.[1] Besides multiple etiologies such as urinary tract infection, trauma, stone disease, instrumentation, and rarely, tumors, vascular malformations like urethral hemangiomas are one of the rare causes of hematuria. They usually present with hematuria and macroscopic urethrorrhagia. The treatment is variable and ranges from intralesional steroid injection and transurethral procedure to open surgery, removal, and reconstruction. Herein, we present the case of a 14-year-old male child who presented with recurrent hematuria and was treated with urethra-cystoscopic holmium laser coagulation.


   Case Report Top


A 14-year-old male child presented in our hospital with complaints of hematuria for 3 days. The episodes of hematuria occurred without any precipitating factors and were self-limited. There was no associated history of prior trauma, lower urinary tract symptoms, and bleeding diathesis. He was normal till 3 months back when he started developing hematuria for the first time. He went to another hospital, where cystoscopy and bipolar coagulation of distal urethral hemangioma was done. He remained symptom-free for 1 month; then, he started developing hematuria again. On examination, there was visible blood from the external urethral meatus. He was lethargic and had severe pallor but was vitally stable. Abdominal and external genitalia examinations were unremarkable. His hemoglobin was 6.6 g/dl and he was transfused one unit of packed cells. His other hematological, biochemical, and coagulation profiles were normal. Mid-stream urine analysis revealed more than 10 red blood cells with 2–4 white blood cells. The urine culture was sterile. Focused ultrasonography of the penis showed prominent vascular spaces on the right side of the corpus spongiosum in the periurethral region extending from the tip to the base of the penis. Corpora cavernosa showed no lesion. Magnetic resonance imaging (MRI) fat-saturated proton density image taken in the axial plane of the penis showed hyperintense thickening in the penile urethra at the proximal third of the penis at the 10–12 O'clock position [Figure 1]. After discussion with the patient, the plan of urethra-cystoscopy and laser coagulation under general anesthesia was made. Hemangiomatous vascular malformation was noted in the anterior urethra at the 8–12 o'clock position of the anterior urethra extending from the mid-penile region to 2 cm distally just before the bulbar urethra. The bladder and posterior urethra were normal. Laser coagulation was done using a holmium laser with 12 Hz and 1.2 joules [Figure 2]. Foley's catheterization was done after securing hemostasis. In the postoperative period, urine was clear. On removal of the catheter after 5 days, there was no hematuria. Urethrocystoscopy performed after 1 month showed no sign of hemangioma. He has completed 1 year of follow-up. He is symptom-free with normal micturition and erectile function.
Figure 1: MRI showing fat-saturated proton density image taken in the axial plane of the penis showed hyperintense thickening in the penile urethra at the proximal third of the penis at 10–12 o'clock position. MRI: Magnetic resonance imaging

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Figure 2: Urethroscopy image showing the urethral hemangioma (a), The holmium coagulation of the lesions (b and c) and the postcoagulation picture of the urethra (d)

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   Discussion Top


Hemangiomas were initially described around 1895. Urethral hemangioma is a benign uncommon vascular tumor, with cavernous hemangioma being the most familiar type. Its origin is still a mystery, although it has been suggested that it originates from unipotent angioblastic stem cells that fail to develop into normal blood vessels.[2] They are commonly found in the skin and liver. Only 2% of them occur in the genitourinary system. The kidney, ureter, bladder, and prostate are the common locations, and there are exceedingly rare in the urethra. Histologically, they appear as thin-walled vascular spaces with endothelial linings. Besides congenital theory, there are other hypotheses such as herniation of the cavernous bodies' constituent tissue, anomalous revascularization after a trauma or chronic irritation, local varicosities, and vascular malignancy. They may be associated with cutaneous hemangiomas, as well as may be part of Klippel–Trenaunay syndrome in 3%–6% of cases.[3]

Urethral hemangiomas mainly affect males. Depending on the site and size of the lesion, the presentation of urethral hemangiomas varies. As urethral cavernous tissue is half-wrapped around the glans and the cavernous body of the penis, there is limited growth of the hemangioma. Thus, often urethral hemangioma is asymptomatic. Painless gross hematuria is the most common presentation of hemangiomas of the anterior urethra. They might cause acute urinary retention with blood clots or hemospermia when located in the proximal urethral. Bigger-sized lesions can produce obstructive urinary symptoms and even bulge throughout the external urethral meatus.[4] In sexually active adult males, they can present with hematuria which often aggravates after penile erection, even with visible blood dripping from the urethral meatus. In our case, the 14-year-old child presented with recurrent gross hematuria.

The diagnostic procedure of choice for urethral hemangioma is urethrocystoscopy. It helps in identifying the site and appearance of the lesions, as well as rules out other causes of urethral bleeding. Therapeutic intervention can be planned in the same setting. Ultrasound and Doppler study can be used as the initial screening test. MRI and MR angiography can delineate the vascular supply of the penis by producing excellent soft-tissue contrast. Although not routinely used, it can be used when the diagnosis is uncertain. In cases of an extensive lesion, it helps to know the extent of the lesion.[5]

Asymptomatic urethral hemangioma does not require treatment. However, various therapeutic options such as pharmacotherapy, physical therapy, endoscopic treatment, and surgery are available [Table 1]. Pharmacotherapy includes oral propranolol, oral or injection of glucocorticoid, and local injection of pingyangmycin. Direct current copper needles, sclerotherapy, arterial embolization, radiation, and microwave therapy can be used as physical therapies. Surgery treatment can be done with local excision, the purse-string closure method, etc.[4] Endoscopic, less invasive photoablation treatment with different lasers such as Nd: YAG, argon, Potassium titanyl phosphate (KTP), and holmium have been proven successful.[3] The surgical excision of Urethral hemangioma (UCH) is a complex operation depending on the location, and there is a very high chance of stricture formation. Radiation treatment can cause radiation orchitis and affect reproductive function. The laser energy gets well absorbed in hemoglobin and produces less scatter, and thus less chance of necrosis of the adjacent tissue and postoperative stricture formation.[6] It can be done as an office procedure and can be repeated if required. In our case, holmium laser ablation was done, which led to successful management. Conservative management by gentle catheterization, wrapping the penis with a bandage, and oral propranolol 0.1 mg/kg in three divided doses is done in cases of acutely bleeding and hemodynamically unstable patients. In cases of recurrent lesions and lesions of a more extensive nature, open surgery and reconstruction are contemplated.[7]
Table 1: Previously reported cases of urethral hemangiomas and their management

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Urethral hemangioma may recur after management. In our case, the child presented with recurrent hematuria, but after the holmium laser treatment, he has been symptom-free after 1 year of follow-up.


   Conclusion Top


Urethral hemangiomas are extremely rare vascular malformations that usually present with gross hematuria. Urethrocystoscopy is the primary diagnostic modality. Laser photocoagulation can be used as a successful management strategy to control bleeding.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the parents have given their consent for their child's images and other clinical information to be reported in the journal. The parents 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.
Fitzwater DS, Wyatt RJ. Hematuria. Pediatr Rev 1994;15:102-8.  Back to cited text no. 1
    
2.
Abbinante M, Crivellaro S, Guaitoli P, Mastrocinque G, Ammirati E, Frea B. Cavernous hemangioma of the spongious body of the urethra: A case report. Urologia 2012;79:211-3.  Back to cited text no. 2
    
3.
Varea-Malo R, Campos-Juanatey F, Portillo Martín JA, Castillo Carvajal L. Multiple urethral hemangiomas associated with urethral stricture: An uncommon aetiology for urethral bleeding. Case Rep Urol 2019;2019:9071327.9.  Back to cited text no. 3
    
4.
Masood A, Hussain I, Khan UU, Masood Y, Umair M, Rehman OF. Hemangioma of penile urethra-Treatment with intralesional triamcinolone. Urol Case Rep 2021;35:101494.  Back to cited text no. 4
    
5.
White JT, Baverstock RJ. Eruption of blood: Arteriovenous malformation of the penile urethra. Can Urol Assoc J 2017;11:E32-4.8.  Back to cited text no. 5
    
6.
Lauvetz RW, Malek RS, Husmann DA. Treatment of extensive urethral hemangioma with KTP/532 laser. Lasers Surg Med 1996;18:92-5.  Back to cited text no. 6
    
7.
Firinci B, Mansıroglu AK, Caglar O, Sipal S, Yigiter M. Urethral cavernous hemangioma in a prepubertal girl: A rare pediatric case. Pediatr Int 2020;62:498-9.  Back to cited text no. 7
    
8.
Yong F, Juan L, Jinhuan W, Haohua Y, Wei C, Jiacong M, et al. Urethral cavernous hemangioma: a highly misdiagnosed disease (a case report of two patients and literature review). BMC Urol 2019;19:13.8.  Back to cited text no. 8
    
9.
Itesako T, Eura R, Okamoto Y, Tatarano S, Yoshino H, Nishimura H, et al. Oral propranolol in a child with infantile hemangioma of the urethra. Urology 2018;122:165-8.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1]



 

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