Blue rubber bleb nevus

* We present a case of spermatic cord cavernous hemangioma. A 32-year-old man presented with a circumscribed, painless mass in the left side of the spermatic cord. An orchiectomy of the left testicle was performed. A 3 x 3 x 2.5-cm mass was present in the spermatic cord area. Histologic examination and immunohistochemical study showed a benign vascular tumor composed of vascular spaces of varying size. Although cavernous hemangioma can occur in any location, the spermatic cord is an extremely rare site, and, to our knowledge, only a few cases have been previously reported.

(Arch Pathol Lab Med. 2002;126:357-358)

Benign and malignant neoplasms of the spermatic cord are rare.1-3 Lipoma, adenomatoid tumor, angiomyxoma, papillary cystadenoma, and leiomyoma are the most commonly occurring benign neoplasms of the spermatic cord. To our knowledge, only a few cases of spermatic cord hemangioma have been reported in the literature.4

REPORT OF A CASE

A 32-year-old man presented with a painless peritesticular mass that had been present for several months, with no significant change in size. The patient was otherwise healthy, and the results of the remainder of his physical examination were unremarkable. An orchiectomy of the left testicle was performed.

MATERIALS AND METHODS

Sections of the tumor were fixed in 10% neutral-buffered formalin. Three-micrometer-thick sections were stained with hematoxylin-eosin and immunohistochemically stained by the streptavidin-biotinylated horseradish peroxidase complex method, as directed by the manufacturer. The antibodies used were vimentin (1:200, Dakopatts, Glostrup, Denmark) and CD34 (1: 100, Becton Dickinson, San Jose, Calif).

PATHOLOGIC FINDINGS

The surgical specimen measured 7.5 x 6 x 3.5 cm and included a testis with an adjacent circumscribed blue paratesticular mass measuring 3 x 3 x 2.5 cm. On cut section, the mass was composed of homogenous, spongy tissue with no evidence of necrosis. Microscopic examination showed irregular, thin-walled vascular channels of varying size, lined by a flattened endothelium without atypia or mitotic activity (Figure, A). The endothelial cells demonstrated positive immunoreactivity for vimentin and CD34 (Figure, B).

COMMENT

Hemangiomas are the most common benign neoplasm of soft tissues The vessels comprising the lesion so closely resemble their normal counterpart that it is difficult to distinguish clearly among neoplasm, hamartoma, and vascular malformation. Cavernous hemangioma, a subtype of hemangioma, can occur in mesenchymal tissue in any part of the body. Most hemangiomas are superficial lesions that have a predilection for the head and neck region. The spermatic cord is an extremely unusual site for this tumor, where it is thought to arise from the pampiniform plexus.6 The color and surface appearance of the lesion relate to the location. Superficial lesions are blue, whereas deep lesions may impart little or no color to the overlying skin. Cavernous hemangiomas are composed of large, dilated, blood-filled vessels lined by flattened endothelium. The vessels may be arranged in a roughly lobular arrangement or may have a diffuse haphazard pattern. The walls are occasionally thickened by adventitial fibrosis. Several syndromes may be associated with cavernous hemangioma. Thrombocytopenic purpura, complicating giant hemangioma, is known as Kasabach-Merritt syndrome.7 A distinctive form of a cavernous hemangioma of the skin, associated with similar gastrointestinal lesions, has been labeled the blue rubber nevus syndrome.8 Cavernous hemangioma of the spermatic cord is a slow-growing, benign tumor that must be distinguished from other paratesticular masses with similar clinical features. Ultrasound and other diagnostic imaging techniques are not helpful in the preoperative evaluation of these lesions.

References

1. Vesga-Molina F, Zabala-Egurrola A, Acha-Perez M, et al. Lipoma of spermatic cord: report of a case. Arch Esp Urol. 1994;47:69-70.

2. Madrigal B, Veig M, Vava A, et al. An aggressive inguinal angiomyxoma in a male patient. Arch Esp Urol. 1999;52:785-788.

3. Geenen RW, Bevers RF, Gielis C, et al. Papillary cystadenoma located in the spermatic cord. J Urol. 1997;158:546.

4. Madrid-Garcia FJ, Garcia S, Parra L, et al. Hemangioma of spermatic cord: presentation of a case with review of the literature. Arch Esp Urol. 1998;51:499502.

5. Enzinger F, Weiss S. Soft Tissue Tumors. 3rd ed. St Louis, Mo: Mosby-Year Book Inc; 1995:579-588.

6. Ergan S, Bran T, Soyka A, et al. Angio architecture of the human spermatic cord. Cell Tissue Res. 1997;288:391-398.

7. Pampin C, Devillers A, Treguier C, et al. Intratumoral consumption of indium-111 -labeled platelets in a child with splenic hemangioma and thrombocytopenia. J Pediatr Hematol Oncol. 2000;22:256-258.

8. Rodrigues D, Bourroul MZ, Ferrer AP, et al. Blue rubber bleb nevus syndrome. Rev Hosp Clin Fac Med Sao Paulo. 2000;55:29-34.

Pavel Liokumovich, MD; Mehrdad Herbert, MD; Judith Sandbank, MD; Michael Schvimer, MD; Leah Dolberg, MD

Accepted for publication August 6, 2001.

From the Department of Pathology, Assaf Harofeh Medical Center, Zerifin, affiliated with Tel-Aviv University, Tel-Aviv, Israel (Drs Liokumovich, Herbert, Sandbank, and Schvimer); and Department of Pathology, Misgav Ladach General Hospital, Jerusalem, Israel (Dr Dolberg).

Reprints: Pavel Liokumovich, MD, Department of Pathology, Assaf Harofeh Medical Center, Zerifin 70300, Israel (e-mail: david1001@ newmail.co.il).

Copyright College of American Pathologists Mar 2002
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