0022-5347/97/1582-0539$03.00/0 THEJOURNAL OF UROL~CY Copyright 0 1997 by AMERICAN UROL~CICAL ASSOCIATION, INC.
Vol. 158,539-540, August 1997 Printed in U S A
Case Reports URETHRAL HEMANGIOMA: CASE REPORT TETSUO HAYASHI, KAZUMASA IGARASHI
From the Departments of Urology, Nissan Kouseikai Tamagawa Hospital, Tokyo, and University Hospital Mizonokuchi, Teikyo University School of Medicine, Kanagawa, Japan
KEY WORDS:urethra, hemangioma, urinary tract Urethral hemangiomas are extremely rare tumors that are seldom described in the literature. To our knowledge the first case was reported in 1895,' and the last review revealed 20 cases in 1991.2 We present a case of this unusual entity. CASE REPORT
A 30-year-old man was referred to our hospital because of gross hematuria and acute urinary retention. All laboratory examinations were within normal limits except for urinalysis, which revealed numerous red blood cells. Ultrasonography and excretory urography were unremarkable. Cystourethroscopy revealed an approximately 5 mm. solitary sessile lesion on the membranous urethra between the verumontanum and external sphincter (part A of figure). Beneath the normally glistening mucous membrane the bluish-red discoloration was irregular in contour and looked like varicosities. At its apex hemorrhage could be seen clearly. The bladder was free of hemangioma and physical examination did not show any hemangiomas on the body surface. The lesion was resected endoscopically and localized transurethral fulguration was attempted. Histological examination showed cavernous hemangioma with a large number of endothelial lined spaces containing red blood cells in the subepithelial tissue (part B of figure). With subsequent healing there has been no evidence of recurrence, incontinence or dysuria due to urethral stenosis. The patient remained well 6 months postoperatively. DISCUSSION
Hemangioma of the urinary tract is a relatively unusual entity that is seldom described in the literature. To our Accepted for publication January 2, 1997.
knowledge since the ease reported in 18951 fewer than 30 cases have been reported.'" We present a case of urethral hemangioma successfully Mgurated endoscopically. In male patients urethral hemangiomas usually present in the distal urethra. Reported ages at clinical presentation ranged from 3 to 68 years old (mean 22). Previous reports stated that urethral hemangiomas were commonly seen in the third decade of life but recently several pediatric cases have also been described.' Since the lesion is believed to be congenital, arising from the embryonic rest of unipotent angioblastic cells that fail to develop into normal blood vessels, early presentation should be expected. The clinical presentation is bloody urethral discharge or frank urethral bleeding. Isolated hematuria or urinary retention may also occur, depending on the sites and numbers of the lesion. Urethral bleeding is common in hemangioma in the distal urethra and hematuria is common in the membranous or proximal urethra. Urethral hemangiomas can be localized in a small or extensive area and can occur as single or multiple lesions. As with hemangiomas of the bladder, extension is oRen further than is grossly evident, with lesions usually occurring in the distal urethra. The management of urethral hemangioma must be individualized but opinions differ regarding the most effective treatment. The tumors are treated with local resection, electrocoagulation and excision of a minor or wider part of the urethra. Histologically, these lesions are composed of thin endothelial lined spaces. The delicate nature of the vascular channels makes it difficult to fulgurate the lesions endoscopically. Although benign in nature, these lesions tend to recur unless completely eradicated.3 For these reasons investigators suggest complete urethral excision and subsequent ure-
A, cystourethrosco y reveals single 5 mm. sessile lesion on urethral mucosa between verumontanum and external sphincter. B, tumor composed of thin engothelid lined spaces containing red blood cells, consistent with cavernous hemangioma. H Q E, reduced from X 100. 539
thral reconstruction. 1-3 Endoscopic management is recommended only for small and isolated lesions.I3 Oral steroid therapy can give good short-term results. Radium insertion and urethral injection with silver nitrate have been tried but are not recommended because of the risk of fibrosis and stricture formation.l Conversely, endoscopic sclerotherapy has also been attempted, resulting in control of bleeding, resolution of the hemangioma and no stricture formation. With selective arteriography urethral bleeding from the hemangioma has been terminated by embolization. Although neodymium:YAG laser irradiation has been described for treatment of bladder hemangioma, we have found no
report of its use for urethral hemangioma. In our case a transurethral approach with fulguration has afforded gradual but complete control of the lesion. REFERENCES
1. McClea, L.E.:Angioma of the male urethra: review of the literature and report of a case. Urol. Cutan. Rev., 52: 204, 1948. 2. Jahn, H. and Nissen, H. M.: Haemangioma of the urinary tract: review of the literature. Brit. J. Urol., 68:113,1991. 3. Scholl, A. J. and Braasch, W. F.: Primary tumors of urethra. Ann. Surg., 76 246, 1922.