Case report

Heart

"°dVessels

© Springer-Verlag 1998

Heart Vessels (1998) 13:152-154

Isolated periarteritis nodosa of the spermatic cord presenting as a scrotal mass: report of a case Kaori K a m e y a m a 1, Shigeru K u r a m o c h i 1, N a o h i k o Kamio 1, Yoshikiyo A k a s a k a 2, Isao Higa 3, and Jun-ichi H a t a 1 1Division of Diagnostic Pathology, Keio University Hospital, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan 2Second Department of Pathology, Toho University School of Medicine, Tokyo, Japan 3Department of Urology, Hiratsuka City Hospital, Kanagawa, Japan

Summary. We report a 57-year-old man who visited a local hospital because of a fist-sized swelling and pain in his right scrotum. Surgically resected tissue revealed necrotizing arteritis in a small spermatic artery. As systemic symptoms for angiitis were not present, this is thought to be the second case of isolated angiitis occurring in the spermatic cord. Key words: Periarteritis nodosa - Vasculitis Spermatic cord

Introduction Isolated periarteritis nodosa commonly occurs in the skin, but may also be found in the appendix, gallbladder, breast, uterus, central nervous system, testis, and epididymis. Occurrence in the spermatic cord is extremely uncommon. We describe herein a rare case of isolated periarteritis nodosa of the spermatic cord without systemic involvement.

Case report A 57-year-old Japanese man visited Hiratsuka City Hospital (Hiratsuka City, Kanagawa, Japan) because of a fist-sized swelling and pain in his right scrotum. The mass had appeared 2 weeks prior to his hospital visit. There was no history of injury, but he had had acute left epididymitis two months before. The patient did not smoke or drink alcohol. There was no family history of urogenital disease. He had undergone thoracoplasty for

Address correspondence to: K. Kameyama

Received December 3, 1998; accepted December 4, 1998

pulmonary tuberculosis at the age of 38 years. He had no evidence of hepatitis B or human immunodeficiency virus infection. On admission, his clinical data were within normal limits, except for an inflammatory reaction. He had no signs or symptoms of systematic vasculitis. His urine contained Pseudomonas aeruginosa. A neoplasm of the testis, epididymis, or spermatic cord was suspected. At operation, however, the bilateral testes were normal, and only the right spermatic cord was swollen. The patient underwent right vasectomy with a routine circumcision. Macroscopically, the surgically resected right spermatic cord showed a small locular mass measuring 5 mm in diameter. The specimen was fixed in 10% formalin and embedded in paraffin for light microscopic examination. For a conventional histochemical study, paraffin sections were stained with hematoxylin and eosin (H-E), elastic H-E, elastica-van Gieson, PTAH, periodic acid-Schiff, alcian-blue, and acid-fast stainings. Histopathologically, the section revealed necrotizing arteritis in a small (approximately 100 ~tm in diameter) spermatic artery (Fig. 1). The artery was infiltrated by lymphocytes, plasma cells, and scanty eosinophils. The muscle coats were disrupted by these cells. Fibrinoid necrosis was observed in a portion of the wall. The internal elastic lamina of the artery was partially disrupted by intimal cellular proliferation and showed luminal narrowing. There was no evidence of granuloma, fibrosis, or a healed lesion. Giant cells were not present (Fig. 2). Only the artery in the segmental site was affected, and veins adjacent to the arteritic lesions and other smaller vessels throughout the specimen were intact. The vas deferens was not involved and showed no significant changes. Paraffin sections were also stained by antibodies for IgG, IgM, and IgA (Dako, Kyoto, Japan) using indirect immunohistochemical techniques. There was no positive staining for any of these markers in any acute arteritic lesion, and the acid-

K. Kamcyama et al.: Periarteritis nodosa of the spermatic cord

Fig. 1. Low-power view of the specimen. The walls of the spermatic arteries (large arrow) are infiltrated with inflammatory cells. The vas deferens (small arrow) is intact (elastica H-E stain, ×10)

fast staining was negative. After a 3-year follow-up, the patient remains asymptomatic.

Discussion Vasculitis of the testis or epididymis is found on pathological examination in patients with systemic arteritis, such as polyarteritis nodosa, rheumatoid arteritis, Henoch-SchOnlein purpura, thromboangiitis obliterans, and tuberculosis. As our patient had no evidence of such systemic vasculitis, we diagnosed this case as isolated periarteritis nodosa of the spermatic cord. To our knowledge, there has only been one case of isolated periarteritis nodosa of the spermatic cord, reported by Takai et al. [1]. Their patient presented with arteritis and subsequent lipid granuloma of the surrounding tissue in the spermatic cord.

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Fig. 2. High-power view of Fig. 1. The spermatic cord artery shows fibrinoid necrosis of a portion of the wall, destruction of the media by inflammatory cells, and luminal stenosis due to profound intimal proliferation (elastica H-E stain, × 100)

The differential diagnosis was thromboangiitis obliterans (Buerger's disease), tuberculosis, or Pseudomonas aeruginosa infection. Thromboangiitis obliterans of the spermatic cord has been reported several times. Our patient was not a smoker, his coagulation system was normal, and histologically, venous thrombosis and neutrophil microabscesses were absent. Therefore, the present case was unlikely to be thromboangiitis obliterans. He had had pulmonary tuberculosis 30 years prior to his hospital visit. It is possible that tuberculosis may contribute to intrascrotal changes, but granuloma, caseous necrosis, and acid-fast bacilli were absent in the sections. Pseudomonas aeruginosa was detected in his urine, but no neutrophil infiltration or evidence of bacterial infection was observed around the vasculitis. Generally, vasculitis is considered to be an autoimmune disease, and isolated periarteritis nodosa may also be related to a localized immunological mecha-

154 nism. In other organs, several types of immunoglobulin have been detected in the affected vessels, but in this case, they were not immunohistochemically detected at the arteritic lesion. Although isolated arteritis has an excellent prognosis [2], patients should be investigated for evidence of a systemic disease and kept under medical surveillance, as the etiology of this condition remains unknown.

K. Kameyama et al.: Periarteritis nodosa of the spermatic cord

References 1. Takai K, Kanemura M, Kitahara K, Hara M, Hoshino Y, Mizuguchi K (1986) Arteritis and fatty degeneration in the spermatic cord resembling periarteritis nodosa: a case report (in Japanese with English abstract). Hinyokika Kiyo 32:615-618 2. Womack C, Ansell ID (1985) Isolated arteritis of the epididymis. J Clin Pathol 38:797-800