―347― 347-350 皮 膚 ・第33巻 ・第3号 ・平 成3年6月

A Case of Lymphangiectasia of Scrotum

Yuji Inoue* Ichiro Katayama** Kiyoshi Nishioka** Motomu Maeda*** Koreaki Matsumoto****

A 53-year-oldmale patient with uncommonscrotal lymphostasisdue to filariasis presented. The scrotal skin showedmultiple reddish keratopapular and albopapular lesion. The entire scrotal skin was excised and wound was resurfaced with split skin grafts. Histologicalexamination of the specimen revealed a dilated dermal lymphaticnetwork. No filaria wormswere seen even under microscopicexamination. Postoperativeresult was successfuland the lymphatic obstructionof the scrotal skin improvedcompletely.

Keywords : lymphoscrotum-filariasis-lymPhangiectasia-surgicalrepairof the lymPhoscrotum

Lymphoscrotum is mainly caused by filariasis ,which phantiasis of the lower extremities. is a disease occurring in tropical and subtropical At the age of 17, he had suffered from recurrent countries. There are two lymphatic dwelling febrile attacks which were accompanied by lymph- parasites of men, Wuchereia bancrofti and Brugia adenitis of the inguinal region. Since then he had malayi . The adult worms of both species usually experienced a periodical (once a year) chyluria reside in the lymphatic channels where they cause and copious milky discharge from the breaking lymphostasis". Here we describe a case of lympho- down of albopapules of the scrotum. He had been scrotum with a peculiar course, in which the skin treated conservatively and his condition had persisted test for filaria was positive and the appearance of for many years. the scrotum was unusual since it had lymphangiectatic In 1983, histological examination of a biopsy of lesions. the reddish papules revealed angiokeratoma scroti, but there were no findings suggesting a diagnosis Case report of filariasis. One year later, he developed non- A 53-year-old man presented with reddish keratotic symptomatic albopapules on the scrotum and more papules on the scrotum in February 1983. He did than half of the reddish papules changed to albopa- not show either signs of lymphoscrotum or ele- pules . His scrotum was edematous and a copious milky discharge from papules which had broked

*井 上 裕 史 open was noted at the base of the scrotum.

Department of Plastic Surgery, Kobe Children's On physical examination, his scrotum was diffusely Hospital. **片 山 一 郎 swollen and covered with numerous white papules ,西 岡 清 Department of Dermatology, Tokyo Medical and 3-5mm in diameter, which were intermingled with Dental University School of Medicine, ***前 田 求 reddish keratotic papules of 1-2mm in diameter

Department of Plastic Surgery, Osaka Police Hospital. (Fig. 1 ) . A thumb-sized mass was palpable in his ****松 本 維 明 left scrotum, and it could not be distinguished from Unit of Plastic Surgery, Department of Dermatology, the left testicle. A walnut-sized non-tender lymphnode Osaka University School of Medicine 平 成3年3月16日 掲 載 決 定 was palpable in his right groin. Physical examination ―348― 皮 膚 ・第33巻 ・第3号 ・平成3年6月

Fig. 3 : Histopathological section of an albopapular lesion

Fig. 1 : Before surgery ; albopapular lesions of the scrotum are seen.

Fig. 4: Six months after reconstruction.

skin test with filarial antigen (FTS-3, kindly Fig. 2: Lymohangiography show enlarged lymph nodes and dilated lymphatic channels in donated by Prof. S. Satoh, Nagoya City University) the right superficial and deep inguinal was positive. Lymphography showed enlarged lymph system (Immediately after injection). inguinal lymph nodes and dilated lymphatic channels otherwise was unremarkable. Laboratory studies, (Fig.2) . There was lymphostasis and saccular including chest X-ray, electrocardiogram,blood areas of retention of the contrast media which cell counts, urinalysis, serum biochemistry, im- corresponded to the albopapules in the scrotum. munoglobulins and renal function tests, were all He was given diethylcarbamazine for several weeks, normal except for slight eosinophilia (4.8%) and then, the scrotal skin was completely removed at chylomicronemia (73mg/dl). Direct blood films the level of the cremasteric fascia and the right obtained at night were negative for organisms.The perispermaticvesselswereligated.Thedenuded ―349― 皮 膚 ・第33巻 ・第3号 ・平 成3年6月 scrotum was covered with a meshed split skin could have coexisted as a senile changes of the graft obtained from his left thigh .The palpable scrotal skin. They became albopapules when the mass in the scrotum proved to be a hydrocele, lymphstasis became severe. Then might have which was treated by Winkelmann' s operation 2). developed in a similar manner to the albopapules Histological examination of the scrotal skin which occur after an abnormal interaction between showed many dilalted vessels in the papillary and lymphatic and blood vessels, although the pathogenetic mid dermis (Fig.3). relationship of angiokeratoma and filariasis is still Endothelial cells of the dilated vessles were obscure. He never developed elephantiasis of the nagative for Factor VIII-related antigen3). periodical episodes of chyluria and the copious The enlarged lymph nodes from his right groin milky discharge from the albopapules might have showed dermatopathic lymphadenopathy histologically saved his scrotal skin. He has now been free from and no parasites were demonstrated. symptoms for more than seven years. The surgical The postoperative course was good and no treatment we used seems to offer one of the best recurrence has been seen in the seven years since choices for preventing recurrent edema 4). surgery (Fig.4). The cosmetic result was good. Despite the fact that the scrotum was constantly peduncular, as is Discussion seen with full relaxion of the cremaster muscle in Lymphatic obstruction, which is now accepted to a normal scrotum, and that the skin was hairless occur as a result of an reaction to the parasite, is and relatively smooth it was fairly normal looking. one of the significant sequelae of filariasis. Obstruction The meshed split skin grafts produced a good of lymphatic drainage may affect the breasts,the result both funcionally and cosmetically. extremities, the scrotum,or the labia. Involvement of the genitalia is rarely described in the literature 4). References Our patient showed a positive skin test to the 1. Edward KK, Marietta V: A new surgical filarial antigen,but we could not demonstrate the technique for lymphedema of the penis and parasites in either his blood or his lymph nodes. scrotum, Pediat Surg, 15: 239-240, 1980 He had had a filarial infection in his teens and 2. Chester BM: Surgical Anatomy, 6th ed, WB the parasite might have been cleared by the diethyl- Saunders, Philadelphia, 1984, 931 carbamazine therapy. The resultant lymphatic 3. Clive RT: Immunochemical methods in the obstruction could easily caused his scrotal symptoms. study of lymphoma and related conditions, He developed both angiokeratoma-like papules Histochem Chitochem, 26: 496-512, 1978 and albopapules on his scrotum. The angio- 4. Sakti D, Daniel T, Argan D: Surgery of male keratoma-like papules might be an early skin genital lymphedema, Urol, 129: 1240-1242, manifestation of lymphoscrotum. Alternatively, they 1983 ―350― 皮 膚 ・第33巻 ・第3号 ・平 成3年6月

陰 嚢 に み ら れ たLymphangiectasiaの 一例

井上裕史 片山一郎 西岡 清 前田 求 松本維明

53歳,男 性 に み られ た フ ィラ リア症 に 起 因す るlymphscrotumか ら陰嚢 全 体 にlymphangiectasia を きた した1例 を報告 した。病 理 組 織像 では 拡 張 した真 皮 リンパ 管網 が み られ た。 治療 は陰 嚢 皮膚 を 全 切 除 して網 状 に した分 層 皮膚 を移植 し,機 能 的 に も外観 も良 好 な結 果 が 得 られ た 。

キー ワ ー ド:リ ンパ 陰嚢 ― フ ィラ リア症― リンパ 管拡 張 症 ― 外科 的 修 復