Case Report

Diagnosis and Management of Scrotal Superficial Angiomyxoma With the Aid of a Scrotoscope: Case Report and Literature Review Zhao Wang,1 Yong-bao Wei,1 Zhuo Yin,1 Bin Yan,1 Duo Li,2 Ke-qin Zhou,1 Jin-rui Yang1 Clinical Practice Points  Scrotal superficial angiomyxoma is a rare begin tumor

in men, with fewer than 10 cases reported.  We describe a scrotal superficial angiomyxoma treated with the aid of a scrotoscope.

 The scrotoscope is a good tool for diagnosis, treat-

ment, and follow-up in scrotal-content lesions.

Clinical Genitourinary Cancer, Vol. -, No. -, --- ª 2014 Elsevier Inc. All rights reserved. Keywords: Epididymic Cyst, Scrotoscope, Scrotum, Superficial Angiomyxoma, Ultrasound

Introduction Superficial angiomyxoma is rare in male genitalia, especially the scrotum. The scrotoscope has been widely used to diagnose and treat scrotal-content lesions. Here we present a case of scrotal superficial angiomyxoma treated with the aid of a scrotoscope in diagnosis, treatment, and follow-up. To our knowledge, ours is the first study to describe a scrotal superficial angiomyxoma treated with the aid of a scrotoscope.

Case Presentation A 25-year-old man sought care at our department in October 2014 because of a mass in his left scrotum, which he had had for nearly a year and a half. It had been a soybeanlike mass 18 months ago, but its size had recently increased to nearly 6  2 cm. The patient consulted with local physicians in March 2014. Ultrasound revealed a weak and heterogeneous echogenic mass nearly 4.2  1.2 cm above the head of left epididymis. Dotlike vascular flow was also detected (Figure 1A). The physician diagnosed an epididymic head cyst. Open surgery was advised by the doctor to remove the mass. The patient rejected this treatment; 1

Department of Urology Department of Pathology The Second Xiangya Hospital, Central South University, Changsha, China 2

Submitted: Nov 12, 2014; Accepted: Nov 29, 2014 Address for correspondence: Jin-rui Yang, Department of Urology, The Second Xiangya Hospital, Central South University, 139 Ren Min Road, Fu Rong District, Changsha 410011, China Fax: þ86-731-85295634; e-mail contact: [email protected]

1558-7673/$ - see frontmatter ª 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.clgc.2014.11.009

he wanted to have the mass observed for further determination. The mass volume and swollen pain had increased, and he was admitted to our department of urology. The patient’s own history as well as his family history were normal. Physical examination revealed a 6  2 cm mass in the left scrotum, which was soft and cystlike (Figure 1B). Serum lactase dehydrogenase, a-fetoprotein, and b human chorionic gonadotropin were within normal reference ranges. Ultrasound detected a hypoechogenic and irregular mass nearly 6.1  2.1 cm in size in the left scrotum, with dotlike and line vascular flow in the mass. Ultrasound also revealed a clear separation from testis and epididymis. We diagnosed a left scrotum cyst before surgery. We explored the patient’s left scrotum via a minimally invasive incision with the aid of scrotoscope. The testis and epididymis appeared normal under the scrotoscope. We did not detect a mass in the contents of the left scrotum. An irregular red mass with complete capsule was found near the scrotal septal region, processing to the left scrotum. We created a small incision near the left scrotal septal region. The mass was removed with complete capsule through this incision (Figure 1C and D). We then performed the scrotoscopy again to make sure that the mass had been removed completely, with no bleeding and no residual mass. Wound drainage was removed within 48 hours after surgery, and the patient was discharged 3 days after surgery. Histologically, the mass border was clear, with complete capsule (Figure 2A). The mass comprised mainly myxoid stroma, including distributed spindle and stellate cells. Thin-walled vascular vessels also appeared in the mass (Figure 2B). The primary diagnosis was mesenchymal lesion. Immunohistochemical examinations were as

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Scrotal Superficial Angiomyxoma Figure 1 (A) Ultrasound Showing Weak, Heterogeneous Echogenic Mass and Dotlike Vascular Flow. (B) Mass in Left Scrotum. (C) Scrotoscopy of Left Scrotum. (D) Incision of Mass With Complete Capsule

Abbreviations: E ¼ epididymis; SS ¼ scrotal septal wall; T ¼ tumor.

follows: VIMþ, CD34þ, S100, SMA, myogenin, Ki-67, and CK (Figure 2C and D). The final pathologic diagnosis was superficial angiomyxoma.

Discussion

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Superficial angiomyxoma is a rare begin tumor in the adult scrotum, especially near the scrotal septal regions. Superficial angiomyxoma is more common in middle-aged men than in women, and superficial angiomyxoma usually occurs on the head and neck, trunk, and extremities.1,2 Fewer than 10 cases of superficial angiomyxoma have been reported in the scrotum.3,4 To our knowledge, we are the first to diagnose a scrotal superficial angiomyxoma with the aid of a scrotoscope. Preoperative diagnosis of superficial angiomyxoma is difficult. In our case, the patient was diagnosed with a cyst in the epididymis head in a local hospital. In our hospital, ultrasound revealed a hypoechogenic mass with dotlike and line vascular flow; however, it did not permit us to draw any conclusions about the mass. Diagnostic difficulties may exist. First, superficial angiomyxoma is relatively rare, especially in scrotal regions. Second, the scrotal room is small, and sometimes it is difficult to locate the mass clearly, especially when the mass is close to testis or epididymis. Finally, some mesenchymal lesions could manifest as superficial angiomyxoma, including aggressive angiomyxoma. Some cysts of scrotal

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contents manifest as superficial angiomyxoma as well. Further, various imaging modalities have their limits: preoperative computed tomography reveals a low-density mass with heterogeneity; magnetic resonance imaging (MRI) reveals a soft tissue mass with clear margin and heterogeneous signal intensity inside; 18F-fludeoxyglucose (FDG)epositron emission tomography is characterized by mild local FDG uptake5,6; and radiologic examinations reveal only some basic characteristics about the mass, such as size and texture. Shafik7 and Gerris et al8 first introduced the scrotoscope in 1988. The scrotoscope has a number of indications for assessing scrotal lesions, such as testis biopsy, hydrocelectomy, and orchidectomy.7-9 Direct observation with minimal invasive incision is one of its advantages; it is also possible to ensure that the lesions have been completely removed by means of scrotoscopy. Yang and Huang10 introduced the scrotoscope to our department in 1990 to diagnose and guide electronic incision of scrotal lesions. In terms of distinguishing scrotal tumors, the scrotoscope has advantages over ultrasound.10 In this case, we used the scrotoscope to examine the whole left scrotum, and we detected the mass near the septal region. After removing the mass, we examined the left scrotum again to make sure that no bleeding was present, to ensure complete resection of the mass, and to exclude the presence of injury to the testis and epididymis. We made only 2 small incisions, one for the scrotoscope and the other for removing the mass. Making a large incision for exploration and for

Zhao Wang et al Figure 2 Histologic Examination and Immunohistochemistry. (A) Mass Border Clear and Well Coated (Original Magnification, 340). (B) Massive Spindle and Stellate Cells Distributed in Myxoid Stroma With Thin-Walled Vascular Vessels (Original Magnification, 3100). (C) CD34 Positive (Original Magnification, 3100). (D) VIM Positive (Original Magnification, 3200)

pulling out the scrotal contents for examination is thus avoided through minimally invasive scrotoscopy. The disease recurrence rate may be as high as 30% to 40% in local regions.1,11 Incomplete incision of the tumor is an important reason for recurrence.12,13 The scrotoscope is a tool to make sure that scrotal lesions have been completely removed. Scrotoscopy, in combination with ultrasound or MRI, may also be used to monitor lesion recurrence.

Conclusion Scrotal superficial angiomyxoma is a rare benign tumor. Ultrasound, computed tomography, MRI, and even FDGepositron emission tomography are limited in their ability to permit diagnosis of scrotal superficial angiomyxoma. The scrotoscope is a good tool to locate the mass in the scrotum and to make sure that it is completely removed. Recurrence occurs as a result of incomplete tumor incision; the scrotoscope is a good method to monitor recurrence.

Disclosure The authors have stated that they have no conflicts of interest.

References 1. Calonje E, Guerin D, McCormick D, et al. Superficial angiomyxoma: clinicopathologic analysis of a series of distinctive but poorly recognized cutaneous tumors with tendency for recurrence. Am J Surg Pathol 1999; 23:910-7. 2. Satter EK. Solitary superficial angiomyxoma: an infrequent but distinct soft tissue tumor. J Cutan Pathol 2009; 36(suppl 1):56-9. 3. Fetsch JF, Laskin WB, Tavassoli FA. Superficial angiomyxoma (cutaneous myxoma): a clinicopathologic study of 17 cases arising in the genital region. Int J Gynecol Pathol 1997; 16:325-34. 4. Nakamura M, Tokura Y. Superficial angiomyxoma on the scrotum of a child. Pediatr Dermatol 2011; 28:200-1. 5. Kim HS, Kim GY, Lim SJ, et al. Giant superficial angiomyxoma of the vulva: a case report and review of the literature. J Cutan Pathol 2010; 37:672-7. 6. Nishio J, Iwasaki H, Aoki M, et al. FDG PET/CT findings of superficial angiomyxoma. Clin Nucl Med 2014; 39:367-70. 7. Shafik A. The scrotoscope. A new instrument for examining the scrotal contents. Br J Urol 1990; 65:209-10. 8. Gerris J, Van Camp C, Van Neuten J, et al. Scrotal endoscopy in male infertility. Lancet 1988; 1:1102. 9. Bin Y, Yong-Bao W, Zhuo Y, et al. Minimal hydrocelectomy with the aid of scrotoscope: a ten-year experience. Int Braz J Urol 2014; 40:384-9. 10. Yang J, Huang X. [Comparative study of the diagnostic preciseness of scrotoscope and B-ultrasonography on scrotal lesions]. Zhonghua Wai Ke Za Zhi 1996; 34:1735. 11. Allen PW, Dymock RB, MacCormac LB. Superficial angiomyxomas with and without epithelial components. Report of 30 tumors in 28 patients. Am J Surg Pathol 1988; 12:519-30. 12. Ali N, Child CS, Michaelides M, et al. Recurrence of a rare skin tumour: superficial angiomyxoma in the eyelid. Can J Ophthalmol 2011; 46:205-6. 13. Basak S, Rogers S, Solomonsz AF. Superficial angiomyxoma of the vulva: a case report of a rare cutaneous tumour. J Obstet Gynaecol 2011; 31:360-1.

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Diagnosis and Management of Scrotal Superficial Angiomyxoma With the Aid of a Scrotoscope: Case Report and Literature Review.

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