□ 증례보고 □

Multiple Cavernous Hemangiomas of the , Penis, and

Jun Min Lee, Joon Ho Wang1, Hong Sup Kim 대한비뇨기과학회지 제49권 제1호 2008 From the Departments of Urology and 1Internal Medicine, College of Medicine Konkuk University, Chungju, Korea 건국대학교 의과대학 비뇨기과학교실, 1내과학교실 Although a hemangioma is usually found in childhood, a cavernous 이준민ㆍ왕준호1ㆍ김홍섭 hemangioma of the genitalia is rarely found. Urologists often face difficulty 접수일자：2007년 10월 2일 when making the decision for surgery to treat a genital cavernous 채택일자：2007년 11월 9일 hemangioma. We report here on a case of a cavernous hemangioma that 교신저자: Hong Sup Kim extended into the glans penis, penile shaft and scrotum. Immediately after Department of Urology, surgical excision of the hemangiomas in the penile shaft and scrotum, the College of Medicine, Konkuk worm-like lesions subsided. The protruding lesions of the glans penis were University, 620-5, Kyohyun naturally relieved after 12 months follow up. (Korean J Urol 2008;49:92- 2-dong, Chungju 380-704, Korea 94) TEL: 043-840-8270 󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏 FAX: 043-848-4722 Key Words: Cavernous hemangioma, Penis, Scrotum E-mail: [email protected]

Hemangioma, a benign vascular neoplasm or a congenital were tender and looked like a “bag of worms” (Fig. 1). Scrotal malformation, is a proliferation of endothelial cells. It com- mass was single and separated from penile mass. And the mass monly involves skin and is rarely found in genitalia. It did not transmit light. The dorsal penile masses placed at 0.5 constitutes only 1-2% of all hemangiomas.1 Because of its cm below the corona sulcus extending onto proximal penile rarity, there is no clear guideline for treatment on genital shaft. Bluish tiny painless masses were found diffusively in hemangiomas. Most hemangiomas on penile shaft have been four different areas of glans penis. Lesions of penile shaft naturally healed after conservative therapy while some require caused a slight right penile lateral curve (less than 15 degree) surgical excisions to treat further pain and/or cosmetic pro- during . Clinical and laboratory findings including blem.2 Recently various types of laser and Intralesional blood test, chemical blood test, blood coagulation test and sclerotherapy have been used to remove hemangiomas within urinalysis were normal. the confines of glans penis or small lesion.2 However, due to These hemangiomas were definitely confined within superfi- its impossibility of knowing the invasive depth and the need cial layer at physical examination without any grossly extended of repeated procedures, laser therapy is not recommended as lesions which were beyond genital area. And there was rare a primary surgical treatment to remove multiple or large chance of extension of hemangioma into other organs. Therefore, hemangiomas. Therefore, it is difficult to decide how and which we decided to perform surgical excision of scrotal and penile area to surgically excise. hemangiomas without any further radiologic studies. Under spinal anesthesia, the area between cutaneous tissue CASE REPORT and Buck's was dissected up to proximal penile shaft after circumference incision 1.0 cm below from the corona A 30-year-old male patient visited due to painful palpable sulcus. Two main masses were found on the dorsal portion of masses of the penis and scrotum developed since two years ago. penile shaft and 5-6 other masses were branched out on the His past and family history did not show specific findings lateral portion of penile shaft. The size of two masses of penile related to the palpable masses. On physical examination, shaft were 3.0x2.0x1.0 cm and 2.0x2.0x1.0 cm and its shape palpable masses were detected on scrotum, penile shaft, and looked like strawberry appearance. Penile main masses were glans penis. The palpable masses on scrotum and penile shaft completely removed but others were partially excised because

92 Jun Min Lee, et al：Multiple Hemangiomas of the Penis 93

Fig. 1. Cavernous hemangiomas of the glans penis, penis (A) and scro- tum (B). Physical examination re- veals a “bag of worms” (arrow) on the penis and scrotum.

Fig. 3. Thin walled irregular vessels and many red blood cells were lined by endothelial cells (arrow) with encapsulation by fibrous tissue. Fig. 2. Operation findings of the cavernous hemangioma on the penis. The main penile hemangioma (arrow). 3.0x2.0x1.0 cm in size was identified at the mid portion of the penile shaft. After surgery, patient had no pain during erection and the lesions disappeared. The patient was satisfied with the disap- of severe adhesion with Buck's fascia. About 3 cm separate pearance of glans penis lesion after 12 months follow up. vertical incision on scrotal raphe was made. Scrotal main mass was completely removed, and they were not connected to any DISCUSSION large vessel such as pudendal veins. The size of scrotal mass was about 2.0x2.0x1.0 cm and its surface was also bumpy (Fig. Most urogenital hemangiomas appear on kidney and bladder 2). Masses of glans penis were left without surgical operation and it is rare to find hemangiomas on glans penis, penile and after removing masses of penile shaft and scrotum because the scrotum. Only 10 cases of solitary hemangiomas on penis, possibility of onset of glans penis mass due to branched out scrotum, and were reported in domestic literature.3,4 mass of penile shaft was high. The masses of penile shaft and Because of its rarity, there is no clear guideline for treatment. scrotum were completely separated from each other. Most penile hemangiomas are painless and require no treat- On microscopic pathological examination, ectatic blood ment due to a high success of natural healing after conservative vessels containing red blood cells and endothelial cells were therapy. However, treatments may be needed when patients consisted with hemangioma. Encapsulated thickening fibrous suffer from pain, ulceration, heaviness and bleeding. Unlike tissues among vessels showed typically characterized cavernous penile hemangiomas, scrotal hemangiomas strongly require a hemangiomas (Fig. 3). surgical excision because of the risk of massive bleeding, 94 대한비뇨기과학회지：제49권 제1호 2008 incomplete resolution, and gradual extension of adjacent organs. natural relief after surgical excision of main mass. Especially, Up to 50% of scrotal hemangiomas showed that it could be in which the glans penis is an associated lesion, watchful extended into the perineum, thigh or abdominal wall, therefore, waiting for spontaneous resolving is primarily recommended. accurate history talking and physical examinations are Because the glans penis is cosmetically important and not easily necessary to define the hidden lesion.5 In some cases, en bloc managed by surgical procedures, it is not late to decide the resection should be performed to reduce the chance of decrease excision of glans penis lesion when there is no satisfactory in size of testis and the rate of infertility at the diagnosis of result after conservative treatment. scrotal hemangiomas.6 In a case of glans penis hemangiomas, various types of laser REFERENCES

(CO2 laser, Nd:YAG laser, yellow-light lager, etc.) and Intralesional sclerotherapy have been used.2 In the legion 1. Alter GJ, Trengove-Jones G, Horton CE Jr. Hemangioma of penis and scrotum. Urology 1993;42:205-8 confined within glans penis, Nd:YAG Laser is preferred due 2. Casale AJ, Menashe DS. Massive strawberry hemangioma of to good cosmetic outcome due to less scar tissue, less bleeding, the male genitalia. J Urol 1989;141:593-4 and no time wasting by using local anesthesia. However, laser 3. Kang MH, Lee SH, Seok BC, Yang SH. A case of intrascrotal therapy is not enabled to know the exact coverage of invasion hemangioma with spontaneous bleeding. Korean J Urol 1994; depth and may need repeated procedures for a complete 35:1036-8 4. Park JS, Chung DY, Kim SJ, Kim YS, Lee EJ, Park KH. removal of multiple or large hemangiomas. Furthermore, laser Hemangioma of scrotum: a report of 3 cases. Korean J Urol therapy costs more than other treatment options. Thus, laser 1997;38:885-8 therapy is not recommended as a primary surgical treatment of 5. Kaufman DG, Kasznica J, Burbige KA, Hensle TW. Benign multiple or large hemangiomas.7 Intralesional sclerotherapy is scrotal tumors masquerading as expanding varicoceles. Urology inexpensive and can be also performed under local anesthesia. 1987;29:612-5 6. Froehner M, Tsatalpas P, Wirth MP. Giant penile cavernous However, it carries the risk of causing pulmonary embolism, 8 hemangioma with intrapelvic extension. Urology 1999;53:414-5 thrombophlebitis, necrosis and ulceration in adjacent tissues. 7. Chung JH, Koh WS, Lee DY, Lee YS, Eun HC, Youn JH. Therefore, it is restrictively used only to treat small lesion on Copper vapour laser treatment of port-wine stains in brown glans penis along with laser therapy. For large or multiple skin. Australas J Dermatol 1997;38:15-21 hemangiomas surgical excision is recommended, because of no 8. Savoca G, De Stefani S, Buttazzi L, Gattuccio I, Trombetta C, Belgrano E. Sclerotherapy of hemangioma of the glans need of repeated procedure and the possibility of complete penis. Urology 2000;56:153 removal of mass.9 9. Ferrer FA, McKenna PH. Cavernous hemangioma of the scro- In the treatment of multiple genital hemangiomas, we may tum: a rare benign genital tumor of childhood. J Urol 1995; conservatively treat the associated small lesions and wait 153:1262-4